Series: Volume 4, Issue 2 2014

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Articles Guest Articles

A new paradigm for assessment of learning outcomes among Australian medical students: in the best interest of all medical students?

Truism: a claim that is so obvious or self-evident as to be hardly worth mentioning, except as a reminder or as a rhetorical or literary device.

Assertion: a proposition that is repeatedly restated regardless of contradiction.

“Medical education in Australia is a world-class system, and produces doctors of the highest capability.”

Truism or assertion? I suggest more assertion than truism. I ask you to consider: “how do we know if this statement is true?” How do you know how good you are; whether you have met the necessary learning outcomes from your medical program?

Introduction: the need for change

Most of us involved in medical education would agree that – broadly speaking, across the sector – what we do in Australian medical education is indeed world class. However, most of us would also say that we could always do better, and that we should always be trying to improve the system.

Despite having a rigorous accreditation system, developed and delivered by the Australian Medical Council (AMC), we do not have explicit measures – across the system – of the outcomes of the educational process at our medical schools. [1] This gap first became apparent to me when preparing my school for AMC accreditation a few years ago. We were asked to provide data on the outcomes of the education (including what our graduates had gone on to achieve) and I found this challenging. We did source some data from Royal Colleges that suggested our graduates performed at a similar level on College exams to other school’s graduates, and we had some data on rural practice, but overall the picture is patchy. Does this gap in the outcomes data matter? I think it does – I suggest that this is in fact a major issue for the sector to consider, and I suggest that medical students need to engage with this issue. Luckily I believe we have a simple solution, and one that is within our grasp.

Some unanswered questions

As a medical dean, I wanted to know – in a quantitative and systematic way – how my students and my school perform. Specific questions I asked myself were:

How do my students and my school perform against a defined national or international standard?

How do my students and my school perform against other medical schools (nationally and globally)?

How can I gather this type of data and use it to improve the educational experiences of our students so that they become even better doctors?

How can I provide quantitative reassurance to my university, the profession and to society that we are doing what we can to fulfil our social responsibility, in terms of graduating competent doctors.

I was not, and am not, particularly interested in our performance in a ranking or league table sense.

Current state of play

Currently in Australia each medical school designs and delivers its own examinations. There is some, but limited collaboration in the design, delivery, evaluation, and quality assurance of the exams in Australian medical schools. Certainly there is no externally focussed data or reporting that affirms the quality and outcomes of our medical degree programs. Firstly, we do not have a national standard against which to assess our students; we do have very clear AMC standards but they do not include examination against a defined set of national competencies. Nor do we have an explicit statement of what knowledge, which clinical skills, and which professional competencies, our graduates are expected to display. Although there are projects underway, some under the auspice of Medical Deans Australia and New Zealand (MDANZ), there is no explicit set of expectations.

Not a national licensing exam

I do not argue for a national licensing exam. [2] Indeed I argue against a national licensing exam. [3] And, the broad view of medical deans seems to be that a national exam would be very expensive, time-consuming and could risk undermining the flexibility and diversity that exists within Australian medical programs. Thus, although the USA and Canada, by way of example, have rigorous national licensing exams, medical deans in Australia are not keen to go this route. Many of us see suggesting a national licensing exam as being overly simplistic, a knee-jerk reaction, not necessary to fix the existing gaps, and potentially damaging.

So, what is it we really need? If we are to provide a level of quantitative reassurance to society, to the profession (in the form of the Medical Board of Australia, and the AMC, for example), to our universities and – very importantly –  to our students, we do need a more collaborative approach to assessment of medical student competency, and we need some common exam questions, and hence some data that can be used to compare performance across students and schools.

Nothing new under the sun: collaborations already underway

This is not a new idea, and indeed there is an impressive collaboration of medical schools working within AMSAC (Australian Medical Schools Assessment Collaboration) that has been doing this since 2009. [4]  In 2012, 11 of the 19 medical schools in Australia (comprising 2492 students) took part in AMSAC. The “AMSAC exam” comprised 49 multiple choice questions in 2012, including 19 testing ‘structure’ and 30 testing ‘function’. Even with this relatively modest number of questions a substantial amount of high quality data can be generated that provides significant insight into the performance of the collaborating schools.

Other collaborations are in place across Australia, each with a slightly different focus. For example the ACCLAiM collaboration is focussing on the OSCE exam, developing common OSCE stations and common approaches to marking, among a group of schools. The IDEAL collaboration is a global network of schools that all contribute to a very large database of exam questions. Other “item banks” that have been used, and could be used, include the AMC items used in the examinations taken by international medical graduates.

In contrast to AMSAC, which focuses on assessment of the biomedical sciences around the midpoint of undergraduate medical training, the AMAC (Australian Medical Assessment Collaboration) group has its focus on testing knowledge and application of knowledge at the end of medical school training. Funded initially by the Australian Learning and Teaching Council [5] and now by the Office of Learning and Teaching with the Department of Industry, Innovation, Science, Research and Technology, AMAC now includes the majority of medical schools and has already piloted an ‘end of course common exam’. AMAC’s focus is on developing a strong collaborative culture among Australian medical schools, who will share a commitment to working together on assessment. Figure 1 shows one of the data outputs from AMAC, demonstrating how performance of the collaborating schools varied in the pilot trial. In the future I anticipate that some schools will choose to share a common exam, or part of an exam, while others will (at least initially) work within the group on identifying, developing and quality assuring individual items for the item bank.

Strengthening our medical schools

It is vital to point out here that the underpinning philosophy is one of cooperation and collaboration. This is about schools working together to strengthen their own assessment capacity and capability, and to help others do the same. It is not at all about withdrawing responsibility for assessment from schools, nor about undermining each school’s capacity to change its assessment practice. By making appropriate use of common exam questions, schools can measure and benchmark performance. These data can inform schools of areas of weakness and strength, and hence lead to curriculum development. I suggest this approach is actually an essential part of a broad quality assurance process that should underpin Australian medical education.

This is not about league tables – and such a counter-productive approach can easily be avoided by making comparisons between schools completely anonymous, which is the way that AMSAC functions (Figure 1).

Shall we go global?

In 2012, medical schools at The University of Queensland (UQ) and The University of Sydney, both delivered the International Foundations of Medicine (IFOM) Clinical Sciences Exam (CSE) to final year medical students, as a required formative assessment. A detailed report on the UQ experience has been submitted for publication. The IFOM CSE is a 160 question multiple-choice exam that tests knowledge and application of knowledge across most of the clinical disciplines. It is effectively an international version of United States Medical Licensing Exam (USMLE) and as such provides a “global standard” against which we can test ourselves. Now, let’s be careful about language here: I am not suggested that the USMLE is the global standard, but it is a global standard, and indeed the IFOM is being designed and developed to be one explicit global standard that students and schools can make use of (should they wish). Of course, good practice would have us formally blueprint any exam against our own curriculum, and this is not possible for the IFOM. The exam is produced by the National Board of Medical Examiners (NBME) [7] in the USA, under strict security constraints, and while a high level blueprinting is done by the international committee that oversees IFOM development, local level blueprinting is not possible. Further research and evaluation is needed to explore how important this is.

So, having delivered IFOM CSE once, we now have high quality data that shows how our medical students performed against one global standard, against the USMLE, and against our colleagues at The University of Sydney. All the information we have gathered is new, is insightful, and is stimulating a range of thoughtful conversations. Of course the data is not definitive, it is not a “magical, gold standard” but it is important data that is giving us important pause for thought.

Peering into the future

So, we are on a journey – a journey that I firmly believe is in the best interests of students, medical schools and all our stakeholders, most importantly your future patients. Just 2-3 years ago while several innovators were working on some of the collaborations described here, the importance of sector-wide change was not on the Deans’ agenda; now it is. What might it look like in the future?

The ideal scenario that would develop over the next few months and years is as follows:

A formal, voluntary, collaboration between as many medical schools in Australia and New Zealand as possible, run under the auspice of MDANZ as the peak body representing these medical schools

  • A formal, inclusive, governance structure would be in place, with appropriate representation of all members
  • A proper business plan to support the collaboration would be developed and managed through the governance structure
  • The outputs of the collaboration would be used by each medical school in a way that it sees fit, and the activities and outputs could include:
    • Annual meeting on assessment practice and strategy
    • A common clinical sciences exam of 100-200 multiple choice questions, covering all core clinical sciences that schools that wish to use a common exam would take up
    • An item bank of MCQs and OSCE stations: schools might choose to use some common OSCE stations in their own clinical exams
    • A range of innovation projects to develop new assessment practices
  • Analysis and statistical support would be provided to allow schools and students to understand how they are performing in comparison with a defined national or global standard
  • Anonymous reports would be available to schools that provide benchmarking data, which could be used in accreditation reports to reassure the AMC, MBA, and society about learning outcomes.

Importantly, students need to be a part of this process. Medical students are deeply engaged in all aspects of medical education in Australia, and rightly so. Surely it is in the students’ best interests to know that their schools are working to improve their educational experience, and their educational outcomes all the time?

Support for this publication/activity has been provided by the Australian Government Office for Learning and Teaching. The views expressed in this publication/activity do not necessarily reflect the views of the Australian Government Office for Learning and Teaching.

References

[1] Australian Medical Council (website) http://www.amc.org.au (accessed Feb 2012).

[2] Koczwara B, Tattersall MHN, Barton MB et al. Achieving equal standards in medical student education: is a national exit examination the answer? Med J Aust 2005; 182: 228–230.

[3] Harden RM. Five myths and the case against a European or national licensing examination. Medical Teacher 2009; 31: 217–220.

[4] Wilson I, O’Mara D. The Australian Medical Schools Assessment Collaboration: What do the differences mean? ANZAHPE Conference, Alice Springs NT, June 2011; Presentation 4331.

[5] Department of Education, Science and Training. Australian Medical Education Study. Commonwealth of Australia 2007, Canberra ISBN 978-0-642-77859-8.

[6] Wilkinson D, Edwards D, Coates H, Canny B, Pearce J, Schafer J, Papinczak T, McAllister L. The Australian Medical Assessment Collaboration: developing the foundations for a national assessment of medical student learning outcomes. Project report )www.olt.gov.au/project-developing-foundation-national-assessment-medical-student-learning-outcomes-2010) ISBN 978-1-922125-33-0.

[7] National Board of Medical Examiners (website) http://www.nbme.org/Schools/iFoM/index.html (accessed Feb 2012)

Categories
Original Research Articles Articles

Melioidosis in the Torres Strait Islands: an 11 year audit 2001-2012

Melioidosis is an infection of concern to global health. It is caused by the intracellular gram-negative bacterium Burkholderia pseudomallei, which is found in the soil and fresh waters of endemic regions. This study identified the average annual incidence of melioidosis in the Torres Strait region between 2001-2012, and compared this to one other similar study, which identified the average annual incidence between 1995-2000. Patient demographics, clinical presentation, outcomes and risk factors were compared to other available studies. In this retrospective study of melioidosis in the Torres Strait, 31 cases were identified over an 11-year period, representing an annual incidence of 37 cases per 100,0000 population. Of these cases, 84% recovered, 16% required intensive care unit (ICU) admission, 3% had a relapse and two patient deaths occurred. The mortality rate was 6.4%. Pneumonia accounted for fifteen presentations (48%) and splenic abscesses for ten presentations (32%), with nine patients presenting with septic arthritis of a joint (29%). Other presentations included hepatic (19%), prostatic (19%), renal (10%), skin (6%), pancreatic (3%), scrotal (3%) and spinal abscesses (3%). Four presented with bacteraemia alone (13%) and one case presented with urethritis (3%). Risk factors included diabetes mellitus (68%), excessive alcohol intake (35%), renal disease (12%), autoimmune disease (6%), malignancy (4%) and the use of immunosuppressive medication (2%).

Introduction

Melioidosis is an infection caused by the intracellular gram-negative bacterium Burkholderia pseudomallei, which is found in the soil and fresh waters of endemic regions. [1] Endemic regions include Southeast Asia and Northern Australia, with peaks of infection occurring during the wet seasons. [2] Melioidosis is of global public health significance, and may be thought of as an emerging infection across tropical regions. [3]

The Torres Strait is a tropical region comprised of 274 islands between the Cape York Peninsula of mainland Australia and Papua New Guinea (PNG). According to the 2006 Australian Bureau of Statistics (ABS) census data, the region has a total population of 7,624, with 82.5% identifying as Indigenous. [4] Half of this population is clustered within the central island group located closest to Thursday Island (TI), which is the commercial and governmental centre of the region. Hospital services are also centralised at TI, however, the closest tertiary referral centre for the Torres Strait region is the Cairns Base Hospital, located 800km south of TI.

Melioidosis is endemic in the Torres Strait, with the most recent average annual incidence reported to be 42.7 cases per 100,000. [5] This is significantly greater than other centres such as Darwin, where the annual incidence was noted to be 19.6 cases per 100,000 between 1986 and 2008. [6] However, during periods of extreme climate, such as during years of significant heavy rainfall, this incidence dramatically increases. This was observed in Darwin between 2009 and 2010, during which the annual incidence increased to 50.2 cases per 100,000, as a result of a heavy wet season. [7] The variability in annual incidence highlights the significant relationship between the transmission of melioidosis and certain environmental factors, such as rainfall level. [1,2]

The transmission of melioidosis most commonly occurs through percutaneous inoculation, and less commonly through inhalation, aspiration and ingestion. [2] A range of host and environmental factors must also exist for an individual to be infected. This includes reduced host immunity and the significant environmental exposure to the pathogen which occurs in endemic regions. [1] This was demonstrated in the study by Kanaphun et al. conducted in northeast Thailand, in which serological studies of 80% of the population exhibited positivity for antibodies against B. pseudomallei by four years of age. [18] There is clear significant environmental exposure in populations of northeast Thailand, yet only 20% of these children developed a symptomatic infection. [1,18] In addition, of the adults infected with symptomatic melioidosis, over 80% displayed reduced host immunity, with most affected by diabetes mellitus or renal failure. [8] In comparison, studies conducted in Australia demonstrated that most individuals were affected by excessive alcohol consumption and diabetes mellitus. [9]

The clinical syndrome associated with the infection of B. pseudomallei is diverse and can affect a variety of organs. Both domestic and international literature overwhelmingly demonstrated the lung as the most commonly affected organ, with pneumonia being the most common clinical presentation of melioidosis. [7,9] Other clinical presentations include symptoms of septicaemia such as fever, malaise, pain in the joints or abdomen, which may be the result of abscess formation in the liver, prostate, kidney, skin or pancreas.

The incubation period varies, as B. pseudomallei can remain dormant for a prolonged period of time. This makes it difficult to establish the exact period of infection. In most cases, a diagnosis of melioidosis is made through positive cultures demonstrating the growth of B. pseudomallei. Serological evidence can also be used to demonstrate past infections, or the presence of rising titres can provide a diagnosis in the absence of positive cultures. [2]

Recurrence of melioidosis can occur in 15% of individuals within ten years of the primary infection, with 50% of these occurring within the first twelve months. [10] Overall, 25% of individuals with recurrence will die. [10] Risk factors for recurrence include severity of initial infection, treatment regime and compliance, and short treatment duration. [10]

Within Australia, it was noted that the mortality rate was similar across the Torres Strait, North Queensland and Darwin. The most recent study conducted in the Torres Strait demonstrated a 22% mortality rate, [5] whilst a larger study in Darwin exhibited a similar mortality rate of 19%. [6,9]

The literature demonstrates the importance of melioidosis to the Torres Strait region. Its seasonal, wet, tropical location and its burden of chronic disease make it a prime location for B. pseudomallei. Furthermore, the most recent examination of this condition in the area is ten years old, highlighting the need for more recent data. This study aims to retrospectively examine all melioidosis cases between the year 2001 and 2012, in order to understand the current burden, risk factors and disease pattern of melioidosis in the Torres Strait.

Methods

This study aimed to conduct a retrospective audit of all patient data between 2001 and 2012, with diagnosis of melioidosis confirmed by isolation of B. pseudomallei. Patients who had been coded as having a diagnosis of a melioidosis infection within this period were identified. All patients who had a positive culture or serology for melioidosis were identified through Queensland Health Pathology. Electronic records were accessed for confirmation of diagnosis and to collect patient medical history, social history and medication lists. Electronic data was accessed through the Queensland Health Electronic Discharge Summary (EDS) program and via clinical notes in Best Practice. AusCare was accessed to confirm positive blood or swab cultures.

Positive serological diagnoses without supporting positive cultures were excluded. Patients with negative pus or blood cultures were excluded. Patient records were de-identified and analysed for demographical data, risk factors, clinical presentation and outcomes. All cases identified were acquired within the Torres Strait region.

Patient transfer to a tertiary hospital for further management and treatment did not result in exclusion of the patient. Once stabilisation was achieved in tertiary centres, patients returned to the TI Hospital to complete treatment, and were not listed as an additional case in the study.

Annual incidence rates were calculated using the 2006 ABS population census data of the Torres Strait region. [4] Recognised risk factors for melioidosis were utilised to aid in analysis of patient records. Data were compared to previous studies from both the Torres Strait and other similar regions within Australia, to determine similarities and differences across these areas.

Patient occupational data was not included in this study, as they were not reflective of environments which would cause significant increased exposure to B. pseudomallei.

Results

Melioidosis was confirmed in 31 cases by isolation of B. pseudomallei from any clinical sample. Of the 31 cases, 28 cases were confirmed by blood culture and two cases were confirmed by swab culture of pus, one from a septic ankle and the other from an epidural abscess. These two cases did not culture B. pseudomallei in serological samples. This represented an average annual incidence of 37 cases per 100,000 of melioidosis within the Torres Strait region.

The majority of individuals affected were male (65%), of Torres Strait Islander decent (Figure 1), with a median age of distribution between 40-49 years (Figure 2). Most patients presented from outer islands (71%), in particular Badu Island. Ninety percent of presentations occurred during the wet season months of the Torres Strait, between January and May.

Many patients had more than one risk factor, and diabetes mellitus was by far the most common, present in 21 cases (68%). Excess ethanol intake (35%) and renal disease (12%) were also identified as significant risk factors in this study. Autoimmune disease (6%), malignancy (4%) and the use of immunosuppressive medication (2%) were considered minor risk factors (Figure 3). Significant risk factors were defined as those that represented a higher percentage in the population as extrapolated from data. Obesity, heart disease and COPD were not identified as significant risk factors in this study.

Of all cases, pneumonia was the most common presentation (48%), closely followed by splenic abscesses (32%) and septic arthritis of a joint (29%). Other presentations included hepatic (19%), prostatic (19%), renal (10%), skin (6%), pancreatic (3%), scrotal (3%) and spinal abscesses (3%). Four presented with bacteraemia alone (13%), and one case presented with urethritis (3%) (Figure 4).The majority of patients (84%) recovered with a total of five ICU admissions (16%), two patients had long- term disability and there were two deaths, giving a mortality rate of 6.4%. Two of the 31 cases occurred in children, one in a five-week old infant with the outcome of death, and one in a five year old child who was not of Aboriginal or Torres Strait Islander origin, who presented with pneumonia and septicaemia. One case of relapse was identified in a 30-year old male from Badu Island who was non-adherent with treatment following discharge from ICU at the Cairns Base Hospital.

Discussion

To date, this study is the largest retrospective study of melioidosis within the Torres Strait. It highlights that melioidosis continues to be a disease of significance in the region, despite a larger public profile in recent years. [3] The average annual incidence reported in this study is 37 cases per 100,000 between 2001 and 2012, less than that reported in a previous study examining 1995 to 2000, in which a total of 24 cases were reported, giving an annual incidence of 42.7 cases per 100,000. [5] The incidence of melioidosis in the Torres Strait remains one of the highest in Australia, excluding the seasonal peak in Darwin between 2009 and 2010. [6] The finding of this study is high compared to international figures, such as those reported in Thailand, where the annual incidence of melioidosis is 5.5 per 100,000. [10]

The high incidence reported here could be attributed to a number of environmental and population factors unique to the Torres Strait region. Climate is a major factor, as the Torres Strait region has very high rainfall during the seasonal wet months from December to May. [5] This reinforces the seasonal distribution of melioidosis, as 90% percent of cases identified occurred within these months. This strong association between the incidence of melioidosis and the rainfall patterns highlights an opportunity for public health intervention to be focussed on the annual wet period in the Torres Strait. In addition, melioidosis incidence was highest on TI (26%), closely followed by Badu Island (23%), which creates additional target locations for public health intervention.

In alignment with the literature, our study demonstrated an over-representation of the Indigenous population presenting with melioidosis. Out of the 31 cases, 28 identified as Indigenous (93.5%), and only three as non-Indigenous. This could be attributed to the generally poorer health status of Indigenous Australians, and the higher rates of chronic illness such as diabetes mellitus, resulting in this population being more susceptible to melioidosis infection. [11] The higher incidence of melioidosis may also be attributed to the cultural differences in the Indigenous population of the Torres Strait. This population has been anecdotally noted to rarely wear shoes, and to spend much of their time in outdoor activities such as fishing. This results in a significant higher environmental exposure to B. pseudomallei.

The incidence of diabetes mellitus within the Torres Strait is one of the highest in Australia, with one third of the population affected. [11] Diabetes mellitus is considered to be one of the most significant risk factors for melioidosis infection, both in Australia and abroad. [10] This was confirmed in this study, with 68% of the case patients identified as having type 2 diabetes mellitus. In comparison to studies in Thailand and Malaysia, 60.3% and 70.4% of their case participants, respectively, were identified as having diabetes mellitus. [12,13] Alternatively, in Darwin, a 20-year prospective melioidosis study from 1989 to 2009, which included 540 cases, identified 39% with underlying type 2 diabetes mellitus. [3] Similarly, a 10-year prospective study in northern Australia identified 37% with diabetes mellitus. [4] Other important risk factors for melioidosis identified were excessive alcohol use (35%) and renal disease (12%). These have been previously identified however alcohol use was considered to be a more significant risk factor for melioidosis in Australia, and renal disease a more significant risk factor in South East Asia. [7,10]

Melioidosis presentation in this study commonly included pneumonia, septic arthritis, and hepatic, splenic and prostatic abscesses. Of these, pneumonia was the most common form of presentation (48%), which reinforces aerosol inoculation as an important transmission route. This finding was similar to that found in numerous studies [3,4]; however, genitourinary infections (3%) were not as common in our study. Genitourinary infections represented 15% of presentations in studies conducted in northern Australia [4] and 14% of presentations in studies conducted in Darwin. [3] Further differences existed in cases presenting with bacteraemia. In our study, 13% of cases presented with bacteraemia alone, compared with 46% of cases presenting with bacteraemia in a north Australian 10-year study. [3] The mortality rate in our study was 6.4%, which was also lower than the north Australian study which reported a mortality rate of 19%. [3] International mortality rates are significantly higher than in Australia, and were reported to be 63% in a Malaysian study and 49% in a Thailand study. [10]

For future follow up and extension of this study, another similar audit would be beneficial to complete for 2012-2022. A future audit would create continuity of data, and would provide further analysis of melioid disease patterns. Melioid disease patterns may be observed to decrease in incidence as a result of increased public awareness, improved access to health care and improved infrastructure, as most of the outer islands currently consist of unpaved roads. Alternatively, the suggested effects of climate change on weather patterns and increased rainfall could lead to an increase in melioidosis incidence, due to the strong environmental links. [1,15-17]

Another type of study that would be beneficial for the Torres Strait region would be to investigate the levels of B. pseudomallei exposure patterns. This would involve environmental sampling, to determine the concentration of B. pseudomallei present in the soils, waters and grasses across different islands. [14] This data could then be cross-referenced with our study data, which identified specific islands as having a higher number of clinical cases. Environmental studies could provide and explanation for the higher incidence of melioidosis present on particular Torres Strait islands. For example, if a decreased concentration of B. pseudomallei was found in the soils, waters and grasses of Badu Island, the health status of the population may be considered as a more weighted risk factor for melioidosis relative to environmental exposure.

Finally, a cost analysis study of the financial burden of melioidosis could be completed. Our study identified that the majority of patients required long stay admissions at TI Hospital and tertiary centres, and that a significant proportion of cases (16%) required ICU stay. This financial burden of melioidosis on the public health system needs to be addressed, as it may provide further incentive to fund greater public health programs aimed at the primary prevention of melioidosis.

Acknowledgements

We acknowledge and appreciate the support and input from all the staff at the TI Hospital. We would like to particularly thank medical records and the pathology department at TI Hospital.

Conflict of interest

None declared.

Correspondence

K Rac: kathrinrac@gmail.com

[1] Wiersinga WJ, van der Poll T, White NJ, Day NP, Peacock SJ. Melioidosis: insights into the pathogenicity of Burkholderia pseudomallei.  Nature Review: Microbiology 2006;4:272-282.

[2] Wiersinga WJ, Currie BJ, Peacock SJ. Melioidosis. The New England Journal of Medicine 2012; 367: 1035-1044.

[3] Dance DAB. Meliodosis as an emerging global problem.  Acta Tropica 2000;74:115-119.

[4] Australian Bureau of Statistics. National regional profile: Torres Strait Islands [Internet]. 2007 [cited 2012 April 24]. Available from:  http://www.censusdata.abs.gov.au

[5] Faa AG, Holt PJ. Melioidosis in the Torres Strait Islands of far North Queensland. Communicable Diseases Intelligence 2002;26:279-283.

[6] Parameswaran U, Baird RW, Ward LM, Currie BJ. Melioidosis at Royal Darwin Hospital in the big 2009-2010 wet season: comparison with the preceding 20 years.  Medical Journal Australia 2012; 196(5):345-8.

[7] Currie BJ, Fisher DA, Howard DM, Burrow JN, Selvanayagam S, Snelling PL, Anstey NM, Mayo MJ. The epidemiology of melioidosis in Australia and Papua New Guinea” Acta Tropica 2000;74:121-127.

[8] Malczewski AB, Oman KM, Norton RE, Ketheesan N. Clinical presentation of Melioidosis in Queensland, Australia. Royal Society of Tropical Medicine and Hygiene 2005; 99(11):856-60.

[9] Currie BJ, Fisher DA, Howard DM, Burrow JN, Lo D, Selva-Nayagam S, Anstey NM, Huffam SE, Snelling PL, Marks PJ, Stephens DP, Lum GD, Jacups SP, Krause VL. Endemic Melioidosis in tropical northern Australia: a 10 year prospective study and review of the literature.  Clinical Infectious Diseases 2000; 31:981-6.

[10] Limmathurotsakul D, Chaowagul W, Chierakul W, Stepniewska K, Maharjan B, Wuthiekanun V, Day NP, Peacock SJ. Risk Factors for Recurrent Melioidosis in Northeast Thailand. Clinical Infectious Disease 2006; 43:979-986.

[11] McDermott RA, McCulloch BG, Campbell SK, Young DM. Diabetes in the Torres Strait of Australia: Better clinical systems but significant increase in weight and other risk conditions among adults, 1999-2005. Medical Journal of Australia 2007; 186:505-508.

[12] Leelarasamee A. Meliodosis in Southeast Asia. Acta Tropica 2000; 74:129-132.

[13] Hansan DZ, Suraiya S. Clinical characteristics and outcomes of bacteraemic melioidosis in a teaching hospital in a north-eastern state of Malaysia: a five year review. Journal of Infections in Developing Countries 2010; 4(4):430-435.

[14] Currie BJ, Ward L, Cheng AC. The epidemiology and clinical spectrum of melioidosis: 540 cases from the 20 year Darwin prospective study. Neglected Tropical Diseases 2010; 30:4-11.

[15] Suppiah R, Collier MA, Kent D. Climate change projections for the Torres Strait region [Internet] 2011. [Cited 2012 April 14]. Available from: http://www.mssanz.org.au/modsim2011/F5/suppiah.pdf

[16] Costello A, Abbas M, Allen A, Ball S, Bellamy R, Friel S, Groce N, Johnson A, Kett M, Lee M, Levy C, Maslin M, McCoy D, McGuire B, Montgomery H, Napier D, Pagel C, Patel J, de Oliveira JA, Redclift N, Rees H, Rogger D, Scott J, Stephenson J, Twigg J, Wolff J, Patterson C. Managing the health effects of climate change. The Lancet 2009; 373:1693-1733.

[17] Kaestli M, Schmid M, Mayo M, Rothballer M, Harrington G, Richardson L, Hill A, Hill J, Tuanyok A, Keim P, Hartmann A, Currie BJ. Out of the ground: aerial and exotic habitats of melioidosis bacterium Burkholderia pseudomallei in grasses in Australia. Environmental Microbiology 2012; 14(8):2058-70

[18] Kanaphun P, Thirawattanasuk N, Suputtamongkol Y, Naigowit P, Dance ABD, Smith MD, White NJ. Serology and Carriage of Pseudomonas pseudomallei: A prospective Study in 1000 Hospitalized Children in Northeast Thailand.  Journal of Infectious Diseases 1993; 167(1): 230-233.
Social phobia in children – risk and resilience factors

Categories
Letters Articles

Lessons learned from internship

Many medical students this year have asked me about what it is like to become an intern. The truth is, nothing you learn at medical school can fully prepare you for the transition to internship. In fact, 42% of newly qualified doctors feel their medical training does not adequately prepare them for starting work. [1] However, it’s not all drama and chaos as shows like House would make you believe. Most internship work is spent on paperwork, requesting investigations and simple procedures like inserting cannulas and taking bloods.

From day one, interns are often rostered for after-hours work, something medical students often have very little exposure to. All of a sudden, new interns may find themselves looking after several wards overnight. Even though some, like me, are interested in critical care medicine, it can still be a challenging thought that over a hundred patients’ lives are entrusted to your care. My first after-hours shift will always stick in my mind, having given me many valuable lessons that I have taken through internship. This is that night in my life:

It is 5pm and most of the doctors have already left. I turn on my pager, secretly hoping it will not beep. Two minutes into the shift, the pager sounds and anxiety kicks in. The nurse on the other side of the phone requests, “Doctor, can you please dose this patient’s warfarin?” It feels strange to not have any other doctors nearby, and my first thought is to ‘phone a friend’. However, I hold off, remembering that the answer lies in the hospital protocol for warfarin, found on all the computers. It reminds me that there is always an abundance of resources and guidance available to us as medical students and interns – if we are willing to ask and look for them.

For the next hour, the tasks are manageable. I re-chart medication charts and get a request to insert a cannula into an elderly lady for intravenous fluids. The team struggled to put the last one in, and her newest one has fallen out during a shower. The lady is thin with fragile veins, and after three painful attempts, the cannula still isn’t in. She is tired of being poked and prodded, and I’m feeling frustrated. I decide to take a break and come back later.

The nurses then page urgently for a doctor. A patient has slipped and knocked his head, and now lies on the floor with a pool of blood beside him. When I arrive at the ward, I find a nurse beside the patient saying, “Everything’s going to be OK, the doctor’s here now,” as if a miracle is about to happen. I do not feel like anyone’s miracle worker, but as one of the first responders and because more senior help had not arrived yet, the nurses look to me for further instructions. My mind freezes, but kickstarts to life again when the basics of ‘ABC’ spring to mind. I feel incredibly grateful for the medical school hammering the ABC approach for such situations. I begin to assess and treat the patient. His airway is patent, cervical spine protected, breathing and circulation maintained. We apply pressure to the wound and perform an ECG and glucose. The few minutes waiting for help to arrive seem to last forever. When more help arrives, we give a huge sigh of relief. I notice that all this time, the patient’s wife has been waiting outside and has been growing extremely worried. As the appropriate members are treating the patient, I take the opportunity to go to her, explain what is happening, and reassure her that her husband is being cared for. One of my consultants once told me that as a junior doctor, one of the best things to do in such situations is to communicate with the patient’s family.

Just when I think that there has been all the excitement I’d need in one night, the pager beeps again. A patient is spiking a high fever, and the nurse is requesting antibiotics. I check through the patients notes first and note that she has been spiking fevers in the last few days, cultures are negative, and the treating team thinks it may be viral. A septic screen has been done, and it was previously decided paracetamol should be sufficient  I reassess her and decide that she does not look too ill at this stage. She has been stable over the last few days.  I choose to leave her without antibiotics, as it does not seem likely that they will be beneficial. The next day, I will check on the patient and be relieved to see that the treating team did not decide to prescribe any antibiotics either.

Before the end of the shift, I go back to visit the elderly lady who still needs a cannula. If I fail, I’ll need to call the duty anaesthetist, and I feel bad because it is getting pretty late in the night. I discuss with the patient, and she agrees for me to have one last opportunity to try. I aim for a small vein in her left hand, and by some stroke of luck, the cannula goes in and flushes smoothly. I breathe a sigh of relief and thankfulness. It reinforces to me that sometimes, just when we are feeling down and tired from trying, we can come back to the task and succeed.

Every day in the hospital, you learn something new. After completing my internship, I am able to reflect back on how much I have learnt in the past year. Completing medical school makes you a doctor, but that is far from the end of the journey. If I may offer some advice, it would be to stay calm in unfamiliar situations, stick with what you have been taught, and never be afraid to ask for help.

Conflict of interest

None declared.

References

[1] Cave J, Woolf K, Jones A, Dacre J. Easing the transition from student to doctor: how can medical schools help prepare their graduates for starting work? Med Teach. 2009 May;31(5):403-8.

Categories
Case Reports Articles

Adult Onset Still’s Disease – a diagnostic dilemma

Introduction

ASOD is characterised by fever, an evanescent skin rash, polyarthralgia, hepatosplenomegaly, leucocytosis, liver enzyme elevation and a high serum ferritin level. [1,2,3] It is a difficult diagnosis to make, as there is no pathognomonic test for the disease and it is a great mimicker of other conditions, such as autoimmune disorders and haematological malignancies.

Despite being a separate clinical entity to JIA and rheumatoid arthritis, there is evidence to suggest that AOSD as well as JIA are triggered by viral infections. [2,3,4] The following case demonstrates a young man who was diagnosed with AOSD following an infection with Epstein Barr Virus. This is impetus for a discussion of the interplay between AOSD and a viral aetiology, and the innate and adaptive immune responses in guiding effective therapy.

Case Presentation

In 2012, a previously healthy 20 year old male presented with a sore throat, malaise, tender cervical lymphadenopathy and fever, consistent with infectious mononucleosis. He was transferred to a secondary referral hospital where paired EBV serology was positive for an active infection despite a negative monospot test. The patient’s travel history and past medical history were unremarkable apart from regular alcohol binge drinking.

After being discharged, he began to experience intermittent fevers and night sweats. In addition to this, he had ongoing malaise and was forced to stop work as a mechanic. Weight loss of 10kg occurred during a two month period, along with a persisting microcytic anaemia, with a haemoglobin level of approximately 8.0 g/dL.

His polyarticular pain was distributed mainly to his ankles, knees, shoulders and wrists, and associated with morning stiffness and visible swelling. The pain was partially responsive to regular ibuprofen. He also complained of intermittent pleuritic chest pain. Over a course of two months, his weight stabilised and night sweats improved, but his anaemia and polyarthralgias persisted.

Approximately two months after his initial diagnosis of infectious mononucleosis, the patient represented to hospital with severe polyarthralgias and was unable to walk. During this admission, he was afebrile but had some mild tender cervical lymphadenopathy with no hepatosplenomegaly. A pleural rub was auscultated. He had a salmon-coloured non-blanching rash on the medial aspect of both legs that felt like a ‘sunburn’; this was biopsied. Although the diagnosis of ASOD had previously been considered, the patient was investigated for other causes for these symptoms. The results of these investigations are presented in Table 1. His investigations included a bone marrow and trephine biopsy, which revealed a markedly hypercellular bone marrow. The skin biopsy of the rash on his legs showed a leucocytoclastic vasculitis with perivascular neutrophilic invasion, but negative staining for complement. This finding is non-specific to the condition and can occur due to drug reaction, immune-complex deposition or be idiopathic. [5] As test results did not indicate another likely cause for his symptoms, the patient was commenced on treatment for ASOD and was referred to a rheumatologist.

Case Discussion

This case illustrates the unique clinical and laboratory picture of AOSD, with its intermitting and remitting fevers, polyarthralgias, myalgias, lymphadenopathy, transient macular rash and pleuritis. It is likely that the patient had a degree of pleuritis, as suggested clinically with a pleural rub and on CT imaging. Serositis manifesting as pleuritis, pleural effusions or pericarditis can be encountered in ASOD, but is rare. [3,6] The rash is fleeting and may only last for hours or days, and skin biopsies generally reveal a non-specific perivascular inflammation. [1] Our patient’s thrombocytosis and markedly elevated serum ferritin are reactive changes. The serum ferritin level has been suggested as a predictive marker for AOSD as it is invariably elevated and often higher than levels found in other autoimmune or inflammatory diseases, with a five-fold increase in serum ferritin being 41% specific and 80% sensitive as a diagnostic test. [9] The markedly high ferritin level in AOSD has been attributed to hyper-production by the reticuloendothelial system or hepatocyte damage, and is unrelated to iron metabolism. [8] The patient’s blood results illustrated a microcytic anaemia, although the iron studies point towards an inflammatory reaction.

The leukocyte count appears to correlate well with the activity of illness. The underlying mechanism of this is probably bone marrow granulocyte hyperplasia, as demonstrated on bone marrow biopsy in our patient. It is not uncommon to see marked reductions in red cell counts, weight loss and hypoalbuminaemia in active disease. [8]

In our patient, causes of fever of unknown origin with or without rash were considered, such as endocarditis, haematological malignancies and systemic vasculitides. The single cytopaenia, normal LDH and bone marrow biopsy excludes leukaemia, lymphoma and myelodysplasia. It is unlikely he had a protracted course of EBV due to the nature of his symptoms and degree of anaemia, in addition to the negative EBV IgM serology. Given the recent heavy rainfall, migrating polyarthritic conditions such as Ross River and Barmah Forest viruses were considered in the differentials.

The diagnosis of ASOD is made after taking into account the patient’s medical history and risk factors for other infectious agents, environment and relevant infectious diseases epidemiology. Although being a diagnosis of exclusion, there are two commonly used clinical criteria in practice, that being Yamaguchi (Table 2), which has been shown to be most sensitive (93.5%) followed by Cush’s (80.6% sensitivity). [7,8]

In regards to the aetiology of AOSD, there have been numerous case reports of AOSD following viral infection, [4,10] with one citing an older female patient diagnosed with AOSD after EBV infection. [2] Other implicated viruses include rubella, mumps, cytomegalovirus, parainfluenza, human herpes virus 6, echovirus, parvovirus B19, and bacterial infections like mycoplasma pneumoniae, chlamydia pneumonia, yersinia enterocolitica and borrelia. [2,8] Although relevant to our patient, the link between infections and AOSD has not been robustly established from an aetiological perspective, [10] and probably only forms part of the multifaceted pathogenesis, that being a dysregulated immune system combined with susceptible HLA loci. However, no consistent associations between AOSD and particular HLA loci have been elucidated, although HLA-B17, HLA-B18, HLA-B35 and HLA-DR2 have been implicated. [6]

It does appear that pathogenesis of the condition overlies autonomous activity of both innate and adaptive immune systems. Patients with AOSD often show hypercomplementaemia, and serum levels of IL-1β, IL-6, IL-18, TNFα, IFN Ɣ and macrophage-colony stimulating factor (M-CSF) have been found to be considerably higher than compared with controls. [6,7,11] These cytokines also appear to share a role in increasing the production of ferritin. [1,12] IL-18 is predominantly secreted by macrophages and has been implicated in hepatotoxicity [13] and joint disease, [7] and may be the cause of liver enzyme derangement characteristic of AOSD. Serum IL-18 levels also appear to correlate significantly with serum ferritin levels. [8] Furthermore, IL-18 may be seen as the part of the bridge between activation of the innate and adaptive immune systems in AOSD, as it facilitates the Th1 response and induces other cytokines like IL-1 β, TNFα and IFNƔ. [6] Pro-inflammatory cytokines such as IL-6, TNFα and IFN Ɣ also increase the expression of Toll-like receptors (TLR), and high circulating levels of cytokines leads to a higher sensitivity of TLR to anti-microbial or viral peptides, thus creating a self-perpetuating cycle of inflammatory response and augmentation. [14]

On the adaptive immunity side of the pathogenesis, the role of T cells in pathogenesis has been well documented. [11,14] Dysregulated production of a particular subset of T helper cells, called Th17 cells, that secrete IL-17 have been implicated in the development of autoimmune diseases. [15] Significantly higher levels of Th17 cells and serum IL-17 levels were found in both AOSD and SLE patients, and there was a parallel decrease with clinical remission. [10] IL-17 stimulates monocytes to produce IL-6 and IL-1β, which are also principle cytokines involved in the differentiation of CD4+ T cells into Th17 cells. [6] These therefore augment and maintain the inflammatory cascade. [16]

Non-steroidal anti-inflammatory drugs (NSAIDS) had previously been the first line medication for ASOD, despite only being effective monotherapy in less than 15% of patients. [10] The benefits of corticosteroids are higher when patients have more pronounced joint disease, with a response rate of two thirds of the patient population. [10]

Highlighting the implicated cytokines, namely IL-1β, IL-6 and TNFα, [17,18] will guide the use of targeted therapies such as the disease modifying anti-rheumatic drugs (DMARDS). There have been favourable results with corticosteroids, and more than two thirds of patients require corticosteroids after NSAIDs are attempted as symptom relief. [6] The use of DMARDS are indicated where the condition is refractory to corticosteroids without signs of remission, or in combination as corticosteroid-sparing agents. This includes methotrexate, which has indirect actions on TNFα and IL-6. Although there is a lack of robust evidence regarding TNF in the pathogenesis of ASOD compared to rheumatoid arthritis, the use of etanercept and infliximab have shown significant improvement in disease in several case series. [10] Of particular note, there is increasing evidence to suggest that anakinra, an IL-1 receptor antagonist, is well tolerated, and several case series have yielded positive results in ameliorating the disease at a haematological, biochemical and cytokine level. An excess of IL-1β inducing factor has been demonstrated in JIA, a condition that also shares similar pathogenesis to that of AOSD. [6,19]

The clinical course of AOSD is heterogeneous, with patients falling into one of three clinical patterns. The first group which affects about 60% of patients [8] is a monocyclic systemic group with only one episode of systemic manifestations, with complete remission within one year of the onset of symptoms. The second group is polycyclic systemic, whom experience more than one episode which is followed by partial or total remission. The third group is a chronic articular group, with persistent polyarthritis lasting longer than 6 months. [6] In the chronic group, the average duration of disease is 10 years, the symptoms appear to be less permanent than other rheumatological diseases and the disease shows less propensity to interfere with social functioning or time off from work despite disability and the need for long-term medication. [20]

Patient outcome

The patient improved satisfactorily with regular ibuprofen and prednisolone 20 mg daily and was discharged after day 7 with a tapering steroid dose.

He was able to resume work, but continued to experience mild intermittent polyarthralgias with no other significant systemic symptoms. Six months post-admission, deterioration in arthritic symptoms prompted the addition of methotrexate.

Key points

  • Adult Onset Stills Disease is a rare systemic inflammatory disorder that mainly affects people aged 16-35 years old.
  • It is a difficult diagnosis to make, and one that must be questioned continually, as it is a mimicker of other disorders, including other causes of fever of unknown origin, infectious diseases and malignancy.
  • It is characterised by both clinical and laboratory manifestations like fever, evanescent rash, polyarthritis and polymyalgias, microcytic anaemia, leucocytosis, thrombocytosis and marked hyperferritinaemia.
  • Treatment is based on clinical course and is similar to that of rheumatoid arthritis. A more targeted biological disease modifying therapy should be chosen with consideration of likely pathogenic pro-inflammatory cytokines.

Consent declaration

Consent from the patient was gained for the writing and distribution of this article for education purposes.

Acknowledgements

Thank you to Dr. Hedley Griffiths, Consultant Rheumatologist.

Conflict of interest

None declared.

Correspondence

S Ooi: soo@deakin.edu.au

References

[1] Mehrpoor G, Owlia M. Adult – onset Still’s disease: A review. Indian J Med Sci. 2009;63(5):207-21.

[2] Schifter T, Lewinski U. Adult onset Still’s disease associated with Epstein-Barr virus infection in a 66-year-old woman. Scand J Rheum. 1998;27(6):458-60.

[3] Mert A, Ozaras R, Tabak F, Bilir M, Ozturk R, Ozdogan H, et al. Fever of unknown origin: a review of 20 patients with adult-onset Still’s disease. Clin Rheum. 2003;22(2):89-93.

[4] Wouters J, van der Veen J, van de Putte L, de Rooij D. Adult onset Still’s disease and viral infections. Ann Rheum Dis. 1988;47(9):764-7.

[5] Koutkia P, Mylonakis E, Rounds S, Erickson A. Leucocytoclastic vasculitis: an update for the clinician. Scand J Rheum. 2001;30(6):315-22.

[6] Kontzias A, Efthimiou P. Adult-onset Still’s disease: pathogenesis, clinical manifestations and therapeutic advances. Drugs. 2008;68(3):319-37.

[7] Bagnari V, Colina M, Ciancio G, Govoni M, Trotta F. Adult-onset Still’s disease. Rheumatol Int. 2010;30(7):855-62.

[8] Kádár J, Petrovicz E. Adult-onset Still’s disease. Best Pract Res Cl Rh. 2004;18(5):663-76.

[9] Lian F, Wang Y, Yang X, Xu H, Liang L. Clinical features and hyperferritinemia diagnostic cutoff points for AOSD based on ROC curve: a Chinese experience. Rheumatol Int. 2012;32(1):189-92.

[10] Mavragani C, Spyridakis E, Koutsilieris M. Adult-Onset Still’s Disease: From Pathophysiology to Targeted Therapies. Int J Inflamm. 2012;2012:1-10.

[11] Efthimiou P, Georgy S. Pathogenesis and management of adult-onset Still’s disease. Semin Arthritis Rheu. 2006;36(3):144-52.

[12] Mehrpoor G, Owlia MB, Soleimani H, Ayatollahi J. Adult-onset Still’s disease: a report of 28 cases and review of the literature. Mod Rheumatol. 2008;18(5):480-5.

[13] Kasper D, Braunwald E, Fauci A, Hauser S, Longo D, Jameson J, Loscalzo J. Harrison’s principles of internal medicine (17th ed.). New York: McGraw-Hill Medical Publishing Division; 2008.

[14] Kramer M, Joosten L, Figdor C, van den Berg W, Radstake T, Adema GJ. Closing in on Toll-like receptors and NOD-LRR proteins in inflammatory disorders. Future Rheumatol. 2006;1(4):465-79.

[15] Lichtman M, Kipps T, Seligsohn U, Kaushansky K, Prchal J. Williams Hematology. 8th ed. USA: McGraw-Hill Companies; 2010.

[16] Chen D, Chen Y, Lan J, Lin C, Chen H, Hsieh C. Potential role of Th17 cells in the pathogenesis of adult-onset Still’s disease. Rheumatology. 2010;49(12):2305-12.

[17] Nordstrom D, Knight A, Luukkainen R, van Vollenhoven R, Rantalaiho V, Kajalainen A, et al. Beneficial Effect of Interleukin 1 Inhibition with Anakinra in Adult-onset Still’s Disease. An Open, Randomized, Multicenter Study. J Rheumatol. 2012 October 1, 2012;39(10):2008-11.

[18] Efthimiou P, Kontzias A, Ward C, Ogden N. Adult-onset Still’s disease: can recent advances in our understanding of its pathogenesis lead to targeted therapy? Nat Clin Prac Rheumatol. 2007;3(6):328-35.

[19] Allantaz F, Stichweh D, Pascual V. Interleukin-1 as a therapeutic target in systemic-onset juvenile idiopathic arthritis. Future Rheumatol. 2007;2(3):305-12.

[20] Sampalis J, Esdaile J, Medsger Jr T, Partridge A, Yeadon C, Senécal J, et al. A controlled study of the long-term prognosis of adult still’s disease. Am J Med. 1995;98(4):384-8.

 

Categories
Original Research Articles Articles

Social phobia in children – risk and resilience factors

Introduction: Anxiety disorders account for one third of psychiatric complaints that young people present to their general practitioners with. Social phobia (SP) is one of the most prevalent of these disorders, in children and adolescents. Methods: Sixty nine patients with carefully defined SP and a control group of 129 typically developing (TD) children were recruited through the Academic Child Psychiatry Unit, Royal Children’s Hospital. All completed the McMasters Family Assessment Device, Hopkins Symptom Checklist, and the Spanier Dyadic Adjustment Scale. Results: There were no clinically meaningful differences in family functioning between the SP group and TD group. Parents of children with social phobia reported higher rates of anxious (η2 = 0.10), obsessive compulsive (η2 = 0.12) and depressive (η2 = 0.13) symptoms, compared to parents of the control group. Furthermore, the relationships of parents with children who have SP appeared to be unhappier (η2 = 0.15) and they reported working together less (η2 = 0.14) than their counterparts. Discussion: Although family functioning per se is not associated with an increased risk of SP in children, the presence of dysfunction tends to lead to protracted SP. Moreover, the stress of having a family member with a mental illness can impact on the parental relationship, causing problems. This may or may not be related to parents of young people with SP displaying symptoms of anxiety, obsessive-compulsiveness and depression. This supports the need to consider both the parents and children when constructing a management plan, which can be initiated and executed by general practitioners.

Introduction

Mental health problems are prevalent amongst young people, with almost one in four experiencing some impairing difficulties in their adolescence. [1] While the first port of call for Australian children tends to be their general practitioners (GP), it is estimated that out of the 25% who seek help, there are at least twice as many who actually have mental health issues, since most young people present with somatic complaints. [1,2] Anxiety disorders are one of the commonest psychiatric problems and over a third of young people who present to their GPs have symptoms of anxiety and depression. [1-3] Social phobia (SP) is a social situation-dependent condition, characterised by persistent and exaggerated fear of embarrassment or humiliation in front of others. [2] It is among the most prevalent of anxiety disorders in children and adolescents, and can be significantly distressing and debilitating to its sufferers, causing social and academic impairment resulting in isolation, school avoidance and refusal. [2,3] Some studies have even shown a more serious side to this condition: anxiety disorders, including SP, are associated with increased risk of suicide attempts and deliberate self-harm. [4,5]

Existing literature found that family functioning is not associated with SP, but the persistence of SP in young people is greater when family functioning is dysfunctional. [6-8] Furthermore, parents of children with SP are likely to suffer from SP themselves, [6-8] there is an emerging association between SP and Bipolar Disorder (BPAD), while comorbid alcohol abuse in patients with BPAD and co-morbid SP seems to be recognised. [9-11] Interestingly, to date, there are no replicated findings about the impact of parental relationship factors on SP specifically.

Aim

The aim of this study is to investigate the potential risk and resilience factors in children with SP in the domains of family functioning, parental psychopathology and parental relationship.  The McMasters Family Assessment Device (FAD), Hopkins Symptom Checklist (HSCL) and Spanier Dyadic Adjustment Scale (DAS) were used to explore these three respective domains.

Hypotheses

The hypotheses that the research addresses are 1) that family functioning between the SP and TD groups would not differ; 2) that parents of children with SP would show features of SP and other anxiety disorders and 3) that parental relationship factors would not have a clear association with SP compared to TD young people.

Methods

This research represents a cross-sectional study and was conducted at the Academic Child Psychiatry Unit (ACPU), Royal Children’s Hospital (RCH) in Melbourne. The ACPU is a clinical research unit that provides comprehensive, standardised assessments and treatment for children and adolescents with internalising and externalising disorders. Prior to the assessments, informed consent was obtained from the parents and children, and a consent form was signed. The data used in the analysis were obtained from standardised questionnaires and structured clinical interviews completed by the parents and young people.

An ethics approval was not required for this paper as both the data analysis and the questionnaires used in this research did not involve the use of identifying information. In addition, the questionnaires utilised for the data are part of the full standard assessment that all patients referred to the ACPU are required to undertake as part of their management. Furthermore, this research project is not part of a Doctoral or Master’s degree.

Family Functioning

In order to assess family functioning, the McMasters Family Assessment Device (FAD) was used. Devised in the 1980s by Epstein and colleagues, the FAD described seven aspects of family functioning through a 52-item questionnaire: problem solving, communication, roles, affective responsiveness, affective involvement, behaviour control and general functioning. [12] The selection of responses for each item ranged from 1 to 4, where 1 = strongly agree, 2 = agree, 3 = disagree, 4 = strongly disagree. [13] The positively oriented items were then recoded and the total score could range from 12 to 48, where higher scores represent better functioning. [13]

Parental Psychopathology

To measure parental psychopathology, the 58-item Hopkins Symptom Checklist (HSCL), a self-report symptom inventory, was utilised. It was scored on parental distress from 1 to 4, where 1 = not at all and 4 = extremely, and it was reported from the five symptom dimensions of somatization, obsessive-compulsive, interpersonal sensitivity, depression and anxiety. [14] The outcome of the survey was in the form of raw data, i.e. mean factor scores and standard deviations, calculated using average-unit weight methods, which made it better geared towards use in clinical research. [14]

Parental Relationship

The Spanier Dyadic Adjustment Scale (DAS) is a 32-item, widely used measure of relationship quality between couples. [15] For the purpose of this study, the abbreviated seven-item version of this instrument, which has shown good internal consistency and is deemed psychometrically sound, was used. [16] The DAS-7 consists of six-point Likert-type scales with end-points of “always agree” to “always disagree” or “all the time” to “never”. [16] The last item on the questionnaire rates relationship satisfaction on a seven-point scale, with end-points of “extremely unhappy” to “perfectly happy”. [16]

Statistical Analysis

Age, social adversity status (SAS) and full-scale IQ (FSIQ) were analysed using univariate analysis of variance, while gender was controlled using the chi square test.  The HSCL, FAD and DAS variables were analysed using univariate analysis of covariance, controlling for SAS and FSIQ.  Partial eta squared was used to ascertain effect sizes for variables that differed between the groups.  The value at which a sample is considered to be clinically significant or large, was set at η2 ≥ 0.10.

Results

The 69 children diagnosed with SP and 129 TD children were identified using the Anxiety Disorders Interview Schedule for Children (A-DISC), which is a semi-structured interview conducted by clinically-trained interviewers. [17] The A-DISC comes in a parent (A-DISC-P) and child (A-DISC-C) form and is designed specifically to diagnose anxiety and other related disorders in individuals from 6-16 years of age. [17] Based on the parent and child account of the most distressing or interfering symptoms, the children are given a principle diagnosis and any other diagnoses fitting the criteria, as determined by the A-DISC. [17]   Patients with Full Scale IQ less than 70, and children living away from their parents were excluded. Patients with any coexisting DSM-IV-TR Axis I diagnosis were also excluded.

The SP and TD groups did not differ in their age or gender:  mean age of the children with SP was 11.01 while the mean age of the TDP was 10.50 years. Out of the experiment group, 40 of them were males and 29 females. Similarly there were more boys in the control group at 70, compared to girls, of which there were 53.

Family functioning

There were no clinically meaningful family functioning differences between the families of children with SP and the TD young people. This was based on scores of η2 = 0.06 for general functioning, η2 = 0.02 for problem solving, η2 = 0.03 for communication, η2 = 0.03 for roles and η2 = 0.03 for behaviour control, all of which are not clinically significant.

This suggests that families from both groups were able to effectively solve problems together and communicate, from a clinical standpoint. Furthermore, the results implied that the established roles and execution of those roles within families of either group were not dissimilar. Also, the way in which the expression and maintenance of behavioural regulation is achieved in the two groups was not different from a clinical perspective.

Parental psychopathology

The data revealed some interesting results in this regard, supporting previous literature that traits of anxiety are significantly present in parents of socially phobic children (F = 16.62, p < 0.0005, η2 = 0.10). Furthermore, it was found that parents of the control group displayed symptoms of an obsessive-compulsive (F = 20.08, p < 0.0005, η2 = 0.12) and depressive (F = 22.01, p < 0.0005, η2 = 0.13) nature. In addition, the effect size of the total HSCL score between the groups was η2 = 0.14.

This demonstrated that parents with children with SP also tended to have manifestations of anxiety, e.g. restlessness, nervousness, tension or even somatic signs like trembling. Moreover, these individuals tended to experience the presence of unwanted thoughts, impulses or actions more often than their counterparts. Interestingly, parents of SP children also appeared to suffer from more dysphoria, anhedonia, avolition and hopelessness than parents of the control group. Overall, the data showed that parents of socially phobic children seemed to have more symptomology of mental health problems than parents with TD children.

Parental relationship

Contrary to the hypothesis on parental relationship, the effect size of the total DAS scores of the two groups proved to be η2 = 0.11. Additionally, there were clinically significant problems with the happiness in the relationship (F = 20.41, p < 0.0005, η2 = 0.14) and ability of spouses with SP children to work together (F = 22.62, p < 0.0005, η2 = 0.15) compared to the control group.

This non-hypothesised result suggested that in families with children with SP, the relationships between the parents tended to be more strained, and they did not often collaborate on projects together.

Discussion

In general, the results of the data analysis were largely similar to the hypotheses put forth at the beginning of this paper. As supported by Knappe and colleagues in both their 2009 publications, family functioning was not associated with a risk of having offspring with SP. Earlier studies by Lieb et al. nearly a decade before also agreed that there was no connection between a child with SP and family functioning.

They did, however, discover that other parental factors, which were outside the scope of the measures used in this project, were associated with greater persistence of SP in children with the diagnosis. For instance, in cases where parents also had SP, negative parental rearing styles like parental overprotection coexisted (DSM-IV threshold SP: Beta = 0.23, T = 2.06, p = 0.043; at least sub threshold SP: Beta = 0.22, T = 2.07, p = 0.042). [8] In situations where there was an absence of disorders in parents, parental rejection (Beta = -0.42, T = -2.18, p = 0.032) also caused the persistence of SP in their offspring. [8] Furthermore, it was noted that, when families were dysfunctional in their functioning, SP tended to be more persistent in the children. [8]

According to the data produced in this study, parents of children with SP tend to have traits of anxiety and obsessive compulsive disorders themselves. Interestingly, the results also showed that a clinically significant portion of these also suffered from depressive symptoms.

It is well known in the literature that parents with SP themselves are at greater risk of having offspring with SP. [6-8] One study supported the findings of this paper, showing the risk of children developing SP is greater when their parents have SP (OR =3.3, 95% CI: 1.4-8.0), other anxiety disorders (OR =2.9, 95% CI: 1.4-6.1), depression (OR =2.6, 95% CI: 1.2-5.4), and even alcohol use disorders (OR =2.8, 95% CI: 1.3-6.1). [7]

This was not the first time alcohol abuse has been associated with SP. Studies by Perugi et al. found that patients with SP and co-morbid Bipolar Affective Disorder Type II (BPAD II) tended to develop alcohol abuse problems. [10] In that situation, however, they argued that the co-existence of BPAD and SP led to protracted anxiety in social situations, which may have explained their increased susceptibility to using alcohol as a social lubricant. [10-11]

Future research should seek to uncover whether the symptoms experienced by the parents are a direct result of raising children with SP, or whether their own psychopathology has contributed to their children’s condition.  Longitudinal study designs are needed.

It was hypothesised that the parental dyad would not be affected as a result of having a child with SP, due to the fact that SP, like many anxiety disorders, are internalising conditions. However, in this study these individuals ranked lower in relationship satisfaction and working on joint projects together. One explanatory theory could be that behavioural difficulties in children with SP, such as school refusal and poor academic performance, indirectly cause discord in the relationship of their parents. Conversely, a troubled parental relationship could potentially exacerbate or even contribute to symptoms of SP that their child.

Although no prior studies have been conducted exploring the use of the DAS as a parental relationship measure, the findings are not unreasonable. A study in 1997 by Friedman et al., which examined adaptive functioning in the families of patients with psychiatric disorders, agreed with this. Their research found that, regardless of diagnosis, having a family member in an acute phase of a psychiatric illness was a significant stressor and put them at risk of poor family functioning. [18]

It may be reasonable to conclude then, that having an offspring with SP puts stress on the family as a whole, and can therefore lead to difficulties within the parental relationship. For instance, the demands of caring for a child with SP in addition to other responsibilities may result in less time spent together as a couple, and hence less time spent working together on projects. Given enough time, this may lead to relationship dissatisfaction. Ideally, future research will recreate or produce more modern data looking into this area, allowing for better interpretation.

Relevance to general practice

As alluded to earlier in this paper, there is a darker side to suffering from SP: namely the risk of suicide and self-harm. Even if one disregards this aspect of the condition, it is undeniable that an individual’s development will be impaired if they are unable to fully participate with their peers socially and academically when growing up. In addition, this research supports the fact that the parents and family unit should not be forgotten when it comes to managing SP in young people. [1]

One of the more effective treatments for SP is Cognitive Behaviour Therapy (CBT). [3,19] In addition to treating SP in children, CBT is also useful in managing adult depression and anxiety disorders. [19] Also within the scope of CBT is dealing with issues related to marital distress. [19] CBT is a type of talking-therapy where a person’s emotions, thoughts and behaviours as linked to particular circumstances e.g. social situations, and negative thought patterns are challenged. [19]

While traditionally seen as a time consuming form of psychotherapy in the GP context, a recent article by Harden encourages GPs to reconsider. [19] She argues that CBT is among the least consuming of psychological therapies, due to its highly structured nature. [19] Furthermore, where CBT was once the domain of psychologists and psychiatrists, Harden outlines several resources for GPs to undergo training in basic CBT techniques, which will enable them to utilise this skill. [19]

Post-training, GPs should be well-equipped to handle the milder forms of SP and family dynamics, and still retain their ability to refer complex cases to specialists. [19] Moreover, they can serve as a bridge for more complex patients who are waiting for specialist appointments. [19] These GPs can gain satisfaction from enabling their patients to develop problem solving techniques, take more responsibility and make better choices. [19] As an added bonus, GPs trained in psychotherapy now receive greater rebates from the government as an incentive to participate in mental health care. [19]

Exposure therapy is another form psychotherapy which effectively manages SP, which GPs are able to execute. [2,20] This behavioural intervention, which incorporates activity scheduling, graded task assignment, distraction and relaxation, can be easily learned by both GPs and patients to a level of competence comparable to treatments conducted by mental health specialists. [2]

Another way to manage SP is using drug therapy, e.g. sertraline with or without psychotherapy. [20] Blomhoff and colleagues found that sertraline was one of more ‘GP-friendly’ psychiatric drugs, owing to its effectiveness and tolerability. They recommended a blend of sertraline and exposure therapy to manage SP in general practice, the latter more so in patients unsuitable for drug treatment or who do not respond to sertraline alone. [20]

Conclusion

In summary, although family functioning per se is not associated with an increased risk of SP in children, the presence of dysfunction can lead to protracted SP. Moreover, the stress of having a family member with a mental illness can impact on the parental relationship, causing problems. This may or may not be related to the parents of young people with SP displaying greater symptoms of anxiety, obsessive-compulsiveness and depression. These interplaying factors make it necessary to consider both the parents and child, when constructing a management plan.

The field of primary care is well-equipped to aid with the management of patients with SP and their families through the use of psychotherapies e.g. CBT and exposure therapy, as well as medications e.g. sertraline. This will be extremely beneficial due to the debilitating and sometimes serious nature of this problem.

Future research should be geared towards producing more modern data and exploring the areas of parental relationship and parental psychopathology in the context of SP, in more detail.

Acknowledgements

To Associate Professor Alasdair Vance – Head of the Academic Child Psychiatry Unit at The Royal Children’s Hospital. Your guidance, encouragement and support were integral to the completion of this project.

Conflict of interest

None declared.

Correspondence

S de Menezes: saramenezessldm@gmail.com

References

[1] Sanci L, Vance A, Haller D, Patton G, Chanen A. Common mental health problems in adolescents. In: Blashki G, Judd F, Piterman L, editors. General Practice Psychiatry

[2] Haug TT, Hellstrøm K, Blomhoff S, Humble M, Madsbu H-P, Wold JE. The treatment of social phobia in general practice. Is exposure therapy feasible? Family Practice. 2000 Apr;17(2):114-8

[3] Baer S, Garland EJ. Pilot study of Community-Based Cognitive Behavioral Group Therapy for adolescents with social phobia. Journal of the American Academy of Child and Adolescent Psychiatry. 2005 Mar;44(3):258-64.

[4] Nepon J, Belik S-L, Bolton J, Sareen J. The Relationship between anxiety disorders and suicide attempts: Findings from the epidemiological survey on alcohol and related conditions. Depression and Anxiety. 2010 Sep;27(9):791-8

[5] Chartrand H, Sareen J, Toews M, Bolton JM. Suicide attempts versus nonsuicidal self-injury among individuals with anxiety disorders in a nationally representative sample. Depression and Anxiety. 2012 Mar;29(3):172-9

[6] Lieb R, Wittchen H-U, Hofler M, Fuetsch M, Stein MB, Merikangas KR. Parental psychopathology, parenting styles and the risk of social phobia in offspring: A Prospective Longitudinal Community Study. Archives of General Psychiatry. 2000 Sep;57(9):859-66.

[7] Knappe S, Lieb R, Beesdo K, Fehm L, Low NCP, Gloster AT, et al. The role of parental psychopathology and family environment for social phobia in the first three decades of life. Depression and Anxiety.2009;26(4):363-70 .

[8] Knappe S, Beesdo K, Fehm L, Ho¨fler M, Lieb R, Wittchen H-U. Do parental psychopathology and unfavorable family environment predict the persistence of social phobia? Journal of Anxiety Disorders.2009 Oct;23(7):986-94.

[9] Pini S, Dell’Osso L, Amador XF, Mastrocinque C, Saettoni M, Cassano GB. Awareness of illness in patients with bipolar I disorder with or without comorbid anxiety disorders. Australian and New Zealand Journal of Psychiatry. 2003 Jun;37(3):355-61.

[10] Perugi G, Frare F, Madaro D, Maremmani I, Akiskal HS. Alcohol abuse in social phobic patients: is there a bipolar connection? Journal of Affective Disorders. 2002 Feb;68(1):33-9.

[11] Perugi G, Frare F, Toni C, Mata Bn, Akiskal HS. Bipolar II and unipolar comorbidity in 153 outpatients with social phobia. Comprehensive Psychiatry. 2001 Sep-Oct;42(5):375-81

[12] Epstein NB, Baldwin LM, Bishop DS. The McMaster Family Assessment Device. Journal of Marital and Family Therapy. 1983 Apr; 9(2) 171–180

[13] Georgiades K, Boyle MH, Jenkins JM, Sanford M. A multilevel analysis of whole family functioning using the McMaster Family Assessment Device. Journal of Family Psychology. 2008 Jun;22(3):344-54

[14] Derogatis LR, Lipman RS, Rickels K, Uhlenhuth EH, Covi L. The Hopkins Symptom Checklist (HSCL): A self-report symptom inventory. Behavioural Science. 1974 Jan;19(1):1-15

[15] Graham JM, Liu YJ, Jeziorski JL. The Dyadic Adjustment Scale: A reliability generalization meta-analysis. Journal of Marriage and Family. 2006; 68:701-717.

[16] Hunsley J, Best M, Lefebvre M, Vito D. The seven-tem short form of the Dyadic Adjustment Scale: Further evidence for construct validity. American Journal of Family Therapy. 2001;29(4):325-335.

[17] Lyneham H, Abbott MJ, Rapee RM. Interrater reliability of the Anxiety Disorders Interview Schedule for DSM-IV: Child and Parent version. Journal of the American Academy of Child and Adolescent Psychiatry. 2007; Jun;46(6):731-6.

[18] Friedman MS, McDermut WH, Solomon DA, Ryan CE, Keitner GI, Miller IW. Family functioning and mental illness: A comparison of psychiatric and nonclinical families. Family Process Journal. 1997Dec;36(4):357-67.

[19] Harden M. Cognitive Behaviour Therapy: Incorporating therapy into general practice. Australian Family Physician 2012 Sept;41(9):668-671

[20] Blomhoff S, Haug TT, Hellstrøm K, Holme I, Humble M, Madsbu HP, et al. Randomised controlled general practice trial of sertraline, exposure therapy and combined treatment in generalised social phobia. The British Journal of Psychiatry. 2001 Jul;179:23-30

Categories
Articles Letters

International medical electives: time for a rethink?

International medical electives (IMEs) are rapidly growing in popularity. A recent study by Law and colleagues [1] conducted across Australia reported that 53% of graduate entry program students and 35% of high school entry students undertook IMEs, of which just over half were in developing countries. In some medical schools the majority of students head overseas for their electives. [2] This phenomenon is not restricted to Australia; in the United Kingdon (UK) and United States (US) roughly 40% of students reported having spent some time in developing countries. [3,4] Many universities across Australia now have global health interest groups, and an increasing proportion of graduating medical students report having some experience in overseas health. [4] Traditionally, these electives are unstructured and arranged on an ad hoc basis between local partners and medical students.

There are undeniable benefits to practicing medicine in an unfamiliar, foreign setting. Students often describe IMEs as one of the highlights of their time at medical school, and it can be an opportunity for unparalleled personal and professional development. On a personal level, students report increased confidence, broadened perspectives, increased cultural competence, and improved communication skills. [5] Professionally, students benefit from being exposed to uncommon conditions and the opportunity for more hands-on experience. [6,7] IMEs also have the potential to influence future practice, with students more likely to enter public service, serve underprivileged populations, and participate in volunteering. [8,9]

However, the results of the aforementioned studies have to be interpreted with caution. Unlike other aspects of the medical course, IMEs tend to be student driven and lack a structured curriculum. Therefore many of the outcome measures are highly subjective and were assessed with unvalidated questionnaires. Given the observational nature of these studies, it is difficult to establish a causative relationship between IMEs and outcome measures. There is also the potential for selection bias (for example, where IME participants were chosen based on their commitment to global health) and publication bias in this area. [8,10] Given the subjectivity of the current literature, it remains unclear if there are indeed any long-term benefits for medical students.

Of note, much of the research on IMEs has revolved around medical students from OECD (Organisation for Economic Cooperation and Development) nations instead of host institutions or patients. Given the short term, transient nature of many IMEs, it seems unlikely that there will be any long term benefits to the local institution. There is potential for limited, temporary benefits such as increased supply of resources, incorporation of new teaching ideas, and positive support from local communities. [5] However, even this can turn out to be a double edged sword, as local institutions develop reliance on visiting medical students. Furthermore, there are cases where the donated equipment end up draining more of the hospitals resources in the long run, or are unable to be maintained. This is not to say that there are not examples of IMEs that have had a positive impact. [11] However, these programs tend to be structured, continuous partnerships between hosts and visiting students with a clear long-term goal.  Unfortunately, the vast majority of IMEs lack such a structure.

A significant proportion of IMEs involve students from developed countries heading to less developed countries. These include pre-clinical students with little to no practical medical training. As students they require proper supervision and this puts added strain on already scarce resources in developing countries. In addition to this problem, many students perceive electives as a holiday, tending to be ill-prepared both culturally and medically for the experience. [3] In worst case scenarios, the student may be placed in a position where he expected to take on the role of a qualified physician and is given responsibility for their own patients. [12] There are several reports in the literature of junior medical students being asked to participate in potentially risky procedures such as lumbar puncture and tubal ligation. [13] Students often try to rationalise this by adopting a utilitarian viewpoint, arguing that no one would look after these patients if they did not step up to the plate. The moral boundaries in these situations are vague and to date there are few established guidelines. However medical students must bear in mind that practicing beyond one’s competency is a serious breach of medical ethics. Students risk doing more harm than good, particularly when they may not be fully aware of the complexities associated with unfamiliar medical conditions and treatments.

To further aggravate this problem, patients in developing countries tend to be vulnerable and greatly disadvantaged. The risk of students developing their skills at the expense of vulnerable patients is a very real one that is probably under-reported in the literature. [14] Anecdotally, we often hear of medical students speak proudly about having been able to perform surgeries or risky procedures on their own with little supervision. There is often a lack of critical reflection surrounding this phenomenon, and clear ethical guidelines should be developed for students.

The motivation behind IMEs is slowly evolving. Traditionally, altruistic reasons were often quoted as the driving factor in medical students pursuing IMEs where students had a genuine interest in serving resource poor areas. [15,16] However, gaining a competitive advantage with the increasing demand for experiences in developing countries has become an important motivating factor. Global health programs look good on a CV and with training programs becoming more competitive, the proportion of students participating in IMEs for this reason will increase.

The threat to medical students’ well-being during electives is often an aspect that is overlooked. Medical students are often drawn by the sense of adventure, opportunity for travel, and the chance for a unique experience different to that back home. At the turn of the century, there was strong concern due to the lack of preparation by visiting British medical students to areas with a high prevalence of HIV. [17] There are often reports of a range of infectious diseases, ranging from schistosomiasis, thyphoid fever, malaria, and dysentery. [2] Literature now demonstrates that adverse events go beyond the risk of HIV and other infectious diseases. Deaths and serious injury have occurred due to risks associated with overseas travel (such as road traffic accidents), suicide, crime and political issues. [18] Aside from physical harm, psychological trauma has also been reported. [18]

Numerous studies encourage pre-departure training as a way to increase awareness of  ethical issues, encourage critical self-reflection, and practical preparation. [13,19] In spite of the physical dangers and ethical dilemmas that are sometimes posed by IMEs, studies have shown that basic practical and ethical preparations for students travelling abroad was low. [20] Only three quarters of Australian medical schools offer pre-departure training, however only half of these are mandatory. [1] The average duration of pre-departure training was 4.7 hours. Only half of Australian medical schools offered post-elective debriefing, out of which roughly half was mandatory. [1] The average duration of post-elective debriefing was 1.2 hours. [1] Medical schools have a duty of care towards medical students and it seems surprising that there is a significant lack of preparation for what is often a unique and unusual experience.

With the increasing ease and affordability of international travel, IMEs will continue to have a growing appeal to medical students. However there is a dangerous lack of critical thought and reflection in terms of the ethical aspect of IMEs, as well as the possible threat to student well-being. Given the strong consensus in the literature for more structured global health education, medical schools should consider developing training programs aimed at enabling students to conduct considered, structured and sustainable IMEs.

Acknowledgement

Saion Chatterjee for his assistance and feedback in editing the draft manuscript.

Conflict of interest

None declared.

Correspondence

Y Foong: Yichao.Foong@utas.edu.au

References

[1] Law IR, Worley PS, Langham FJ. International medical electives undertaken by Australian medical students: current trends and future directions. The Medical journal of Australia. 2013;198(6):324-6. Epub 2013/04/03.

[2] Goldsmid JM, Sharples N, Bettiol SS. A Preliminary Study on Travel Health Issues of Medical Students Undertaking Electives. Journal of Travel Medicine. 2003;10(3):160-2.

[3] Dowell J, Merrylees N. Electives: isn’t it time for a change? Medical Education. 2009;43(2):121-6.

[4] Association of American Medical Colleges. Medical School Graduation Questionnaire Report: 2000. Washington, DC: October 2000.

[5] Mutchnick IS, Moyer CA, Stern DT. Expanding the Boundaries of Medical Education: Evidence for Cross-Cultural Exchanges. Academic Medicine. 2003;78(10):S1-S5.

[6] Thompson MJ, Huntington MK, Hunt DD, Pinsky LE, Brodie JJ. Educational effects of international health electives on US and Canadian medical students and residents: a literature review. Academic Medicine. 2003;78(3):342-7.

[7] Drain PK, Holmes KK, Skeff KM, Hall TL, Gardner P. Global Health Training and International Clinical Rotations During Residency: Current Status, Needs, and Opportunities. Academic Medicine. 2009;84(3):320-5 10.1097/ACM.0b013e3181970a37.

[8] Ramsey AH, Haq C, Gjerde CL, Rothenberg D. Career influence of an international health experience during medical school. Fam Med. 2004;36(6):412-6. Epub 2004/06/08.

[9] Matar WY, Trottier DC, Balaa F, Fairful-Smith R, Moroz P. Surgical residency training and international volunteerism: a national survey of residents from 2 surgical specialties. Canadian journal of surgery Journal canadien de chirurgie. 2012;55(4):S191-9. Epub 2012/08/03.

[10] Imperato PJ. A third world international health elective for U.S. medical students: the 25-year experience of the State University of New York, Downstate Medical Center. Journal of community health. 2004;29(5):337-73. Epub 2004/10/09.

[11] Vora N, Chang M, Pandya H, Hasham A, Lazarus C. A student-initiated and student-facilitated international health elective for preclinical medical students. Medical education online. 2010;15. Epub 2010/02/27.

[12] Banatvala N, Doyal L. Knowing when to say “no” on the student elective: Students going on electives abroad need clinical guidelines. BMJ: British Medical Journal. 1998;316(7142):1404.

[13] Petrosoniak A, McCarthy A, Varpio L. International health electives: thematic results of student and professional interviews. Medical Education. 2010;44(7):683-9.

[14] Radstone SJ. Practising on the poor? Healthcare workers’ beliefs about the role of medical students during their elective. Journal of medical ethics. 2005;31(2):109-10. Epub 2005/02/01.

[15] Powell AC, Casey K, Liewehr DJ, Hayanga A, James TA, Cherr GS. Results of a national survey of surgical resident interest in international experience, electives, and volunteerism. Journal of the American College of Surgeons. 2009;208(2):304-12.

[16] Huish R. The Ethical Conundrum of International Health Electives in Medical Education. Journal of Global Citizenship & Equity Education. 2012;2(1).

[17] Wilkinson D, Symon B. Medical students, their electives, and HIV. BMJ (Clinical research ed). 1999;318(7177):139-40. Epub 1999/01/15.

[18] Tyagi S, Corbett S, Welfare M. Safety on elective: a survey on safety advice and adverse events during electives. Clinical medicine (London, England). 2006;6(2):154-6. Epub 2006/05/13.

[19] Dharamsi S, Osei-Twum J-A, Whiteman M. Socially responsible approaches to international electives and global health outreach. Medical Education. 2011;45(5):530-1.

[20] Bozorgmehr K, Schubert K, Menzel-Severing J, Tinnemann P. Global Health Education: a cross-sectional study among German medical students to identify needs, deficits and potential benefits (Part 1 of 2: Mobility patterns & educational needs and demands). BMC Medical Education. 2010;10(1):66.

 

Categories
Case Reports Articles

Mobile segment of the hamulus causing dynamic compression of the motor ulnar nerve branch in the hand

This paper is the first to document the mechanism of how a mobile segment of the hook of hamate can dynamically compress the motor branch of the ulnar nerve. Presented is the case of a professional golfer who experienced pain on the ulnar aspect of his right hand that he attributed to weakness and inability to control his hand. Imaging revealed the rare condition of os hamulus proprius causing a dynamic compression of the ulnar nerve when in power grip.  Provided is a review of wrist anatomy with particular focus on the peculiar case of the bipartite hamulus.

Introduction

Anatomy of the wrist

The wrist comprises a proximal and distal carpal row. The distal carpal row consists of the trapezium, trapezoid, capitate and hamate and acts as a base for the metacarpals. The proximal carpal row consists of the scaphoid, lunate, triquetrum and pisiform bone. These function as an intercalated segment, balancing the hand on the radius and ulna. [13]

Hamate anatomy and function

The hamate articulates with the triquetrum proximally and the bases of the 4th and 5th finger metacarpals distally. The hook of the hamate is an important structure in the hand.  Protruding from the volar surface of the hamate, it anchors the distal transverse carpal ligament, acting as a pulley for the ulnar flexor tendons and protecting the motor branch of the ulnar nerve. This branch of the ulnar nerve courses dorsally and distally around the hook of the hamate to supply nearly all the intrinsic muscles of the hand. [14]


Guyons Canal and the Ulnar Nerve

Felix Guyon described a potential space [15], which is a fibro-osseous tunnel, protecting the ulnar nerve and artery and veins as they enter the hand. The boundaries of Guyon’s canal are the pisiform bone, the tip of the hook of the hamate, the piso-hamate ligament and the transverse carpal ligament.

Os Hamulus proprium

The os hamulus ossifies from a primary ossification center in the body of the hamate; however, occasionally a secondary ossification center in the hook of the hamate is also present. [1] Rarely, the secondary ossification center in the hook of the hamate does not unite with the primary ossification center in the body of the hamate. [2] When the tip of the hook of the hamate does not fuse with the body of the hamate the result is a separate ossicle known as the os hamulus proprium or a bipartite hamulus. Whilst an os hamulus proprium or bipartite hamulus is often congenital a similar appearance can sometimes be the result of a non-union of a fracture of the hook of the hamate. [3]

Ossification of the hamate is not complete until the early teenage years. [4] Bone growth and maturation usually takes place via a single ossification center. However, a secondary ossification center independent from associated underling bone occasionally develops giving rise to an accessory ossicle. [9] This lack of fusion has been observed involving the hamulus and the hamate and is known as either os hamulus proprium or bipartite hamulus. Such cases are often congenital in nature; however, depending on the patient’s history, trauma or degenerative etiology should be considered. [10]

A study [5] conducted in 2005 on 3,218 hand radiographs revealed that variations are more prevalent than previously thought. 96 participants were found to have variations of the hook of hamate of which 42 patients had a bipartite hook, 50 had a hypoplastic hook and 4 had an aplastic hook. Furthermore, 93 of these cases presented with carpal tunnel syndrome symptoms.

In 1981, Greene et al. [6] identified a single case of bipartite hamulus with ulnar tunnel syndrome. However, since then there have been no other accounts of the os hamulus proprius, associated with dynamic ulnar neuropathy.

Case Study

History

The patient was a 37 year old professional right handed golfer with an unremarkable medical record.

He presented with an eight-week history of pain in the ulnar side of the right hand with loss of fine motor control requiring the use of his contralateral left hand to perform activities of daily living. The patient reported no other neurological symptoms at the time.

Physical examination revealed wasting of the intrinsic muscles of the right hand, most pronounced in the first dorsal interosseous muscles with weak intrinsic movements when comparison to the left side. Following initial examination a series of investigation and imaging was conducted:

It is not uncommon for golfers to fracture the hook of hamate based on the type of grip and dynamics of the golf swing. Furthermore, they can develop stress fractures of the hook of the hamate, which subsequently do not unite. [11,12]

Whilst this may have been the mechanism for the development of injury, an alternative explanation implicates a congenital anomaly where the primary ossification center the hamate fails to unite with the hook of the hamate giving rise to a bipartite bone (os hamulus proprius). [3]

Findings

This patient had a well-established long-standing asymptomatic non-union of the hamate or an os hamuli proprius, which subsequently became symptomatic following a motor vehicle accident in January 2005 resulting in an acute eight-week history of fine motor control deficit in the right hand.

Surgical intervention

A mobile segment of the hook of the hamate was identified.  Pressure over the mobile segment of the hook of the hamate compressed the motor branch of the median nerve as it traversed around the ulnar and distal hook of the hook of the hamate. The motor branch of the median nerve was swollen proximal to the point where the mobile segment of the hook of the hamate dynamically impacted on the nerve. This had the appearance of a ‘neuroma in continuity’ commonly seen from failure of regenerating nerve growth cone to reach peripheral targets.

The ulnar nerve was released in Guyon’s Canal. The motor branch of the ulnar nerve was identified and dissected as it coursed around the hook of the hamate. The hook of the hamate was very mobile and unstable. Manipulation of the mobile hook of the hamate demonstrated how it impacted and compressed the motor branch of the median nerve distal to the swollen segment of the motor branch of the median nerve. This was surgically excised.

The patient noticed a marked improvement of symptoms within two days post-operatively commenting on a return of ‘power and movement’. Following rehabilitation through daily grip strength exercises; this was further demonstrated on clinical examination at eighteen days confirming a return of intrinsic muscle power in the right hand.

The following five images describe the surgical repair of Os Hamulus Proprius as performed in this case.

Discussion

The hook of the hamate is an important structure providing mechanical stability on the ulnar aspect and protecting the motor branch of the ulnar nerve as it traverses deep into the hand from Guyon’s canal. It is also an important structure for insertion of the flexor retinaculum and as a result the muscles on the ulnar side of the hand. [16]

It is very likely that this abnormality of the hook of the hamate was present prior to his injury. The most likely explanation is that it is a secondary ossification center of the hook of the hamate (os hamulus proprius) which went on to unite. However, it is not possible to completely rule out that this represents a long standing non-union of the hook of the hamate and at some stage in the past he may have sustained a stress fracture which resulted in a non-union. [1,3,6,8,11,17]

Clinical examination plays a crucial role in isolating cases of os hamulus proprius. Patients will often present with clinical signs suggesting ulnar neuropathy such as intrinsic muscle weakness and altered sensation of the hand. In differentiating a case of bipartite hamulus, there will also be marked local tenderness over the hook of hamate with symptomatic pain due to dynamic compression such as when performing a power grip. Further hand and upper limb evaluation can compliment the diagnosis by quantifying and comparing loss of strength in the hand. [17]

The patient had marked motor (intrinsic hand muscles) weakness and some minor impairment of sensation in the ulnar distribution, which is consistent with the electrophysiological abnormalities in the hand. Surgery to remove the mobile segment of hamulus resulted in major improvement – particularly in terms of the level of his symptoms and restoration of normal power to the intrinsic muscles of the hand. Excision of the mobile os hamulus proprius has restored control and sensation of his left hand and enabled him to resume his career as a professional golfer.

Ulnar nerve compression in the hand could be due to a multitude of factors, including a tumour, a ganglion cyst, a fracture of either the pisiform or the hamate, compression in Guyon’s Canal, and an aneurysm of the ulnar artery. [18] To discriminate between a congenital bipartite hamulus or a non union of the hook of the hamate five criteria [17] have been described:

  • Bilaterally similar bipartite hamulus
  • Absence of history or signs of previous trauma
  • Equal size and uniform signal intensity of each part on imaging
  • Absence of progressive degenerative changes between the two components of the hamate or elsewhere in the wrist
  • Smooth well corticated and rounded margins of the hamate and mobile separate hook

Treatment

There are a limited number of options to treat a mobile hamulus segment causing ulnar nerve compression. [8] Initial splinting of the hand can be trialed to prevent dynamic compression of the nerve in the hope that pain and weakness resolve. [5] Furthermore, avoidance of sports relying on grip strength may provide symptomatic relief. If these interventions do not result in the resolution of symptoms, then there is the option of surgically excising the accessory ossification center on the tip of the hook of the hamate with subsequent decompression and release of the ulnar nerve such as presented in this case.

Consent declaration

Informed consent was obtained from the patient for publication of this case report and accompanying figures. IMAGE ONE is taken from http://upload.wikimedia.org/wikipedia/commons/3/31/Gray422.png. This image is in the public domain because its copyright has expired. This applies worldwide.

Acknowledgments

This paper was written under the supervision of Jeff Ecker from the Western Orthopaedic Clinic in Perth, WA.

Conflict of interest

None declared.

Correspondence

S Moniz: monizsheldon@gmail.com

References

[1] Andress M, Peckar V. Fracture of the hook of the hamate. British Journal of Radiology. 1970;43(506):141-143.

[2] Blum AG, Zabel J-P, Kohlmann R, Batch T, Barbara K, Zhu X, et al. Pathologic Conditions of the Hypothenar Eminence: Evaluation with Multidetector CT and MR Imaging1. Radiographics. 2006;26(4):1021-1044.

[3] Bianchi S, Abdelwahab I, Federici E. Unilateral os hamuli proprium simulating a fracture of the hook of the hamate: a case report. Bulletin of the Hospital for Joint Diseases Orthopaedic Institute. 1990;50(2):205.

[4] Grave K, Brown T. Skeletal ossification and the adolescent growth spurt. American journal of orthodontics. 1976;69(6):611-619.

[5] Chow JC, Weiss MA, Gu Y. Anatomic variations of the hook of hamate and the relationship to carpal tunnel syndrome. The Journal of hand surgery. 2005;30(6):1242-1247.

[6] Greene M, Hadied A. Bipartite hamulus with ulnar tunnel syndrome–case report and literature review. The Journal of hand surgery. 1981;6(6):605.

[7] O’Driscoll SW, Horii E, Carmichael SW, Morrey BF. The cubital tunnel and ulnar neuropathy. Journal of Bone & Joint Surgery, British Volume. 1991;73(4):613-617.

[8] Pierre-Jerome C, Roug I. MRI of bilateral bipartite hamulus: a case report. Surgical and Radiologic Anatomy. 1998;20(4):299-302.

[9] Garzón-Alvarado D, García-Aznar J, Doblaré M. Appearance and location of secondary ossification centres may be explained by a reaction–diffusion mechanism. Computers in biology and medicine. 2009;39(6):554-561.

[10] Freyschmidt J, Brossmann J. Koehler/Zimmer’s Borderlands of Normal and Early Pathological Findings in Skeletal Radiography. TIS; 2003.

[11] Koskinen SK, Mattila KT, Alanen AM, Aro HT. Stress fracture of the ulnar diaphysis in a recreational golfer. Clinical Journal of Sport Medicine. 1997;7(1):63.

[12] Torisu T. Fracture of the hook of the hamate by a golfswing. Clinical orthopaedics and related research. 1972;83:91-94.

[13] Viegas SF, Patterson RM, Hokanson JA, Davis J. Wrist anatomy: incidence, distribution, and correlation of anatomic variations, tears, and arthrosis. The Journal of hand surgery. 1993;18(3):463-475.

[14] Berger R, Garcia-Elias M. General anatomy of the wrist. In: Biomechanics of the wrist joint: Springer; 1991.

[15] SHEA JD, McCLAIN EJ. Ulnar-nerve compression syndromes at and below the wrist. The Journal of Bone & Joint Surgery. 1969;51(6):1095-1103.

[16] LaStayo P, Michlovitz S, Lee M. Wrist and hand. Physical Therapies in Sport and Exercise. 2007:338.

[17] Evans MW, Gilbert ML, Norton S. Case report of right hamate hook fracture in a patient with previous fracture history of left hamate hook: is it hamate bipartite? Chiropractic & osteopathy. 2006;14(1):1-7.

[18] Zeiss J, Jakab E, Khimji T, Imbriglia J. The ulnar tunnel at the wrist (Guyon’s canal): normal MR anatomy and variants. AJR. American journal of roentgenology. 1992;158(5):1081-1085.

Categories
Letters Articles

What comes next after #interncrisis?

Will last year be a turning point or just another chapter in the saga of medical workforce planning in Australia? The story, so ably publicised by the high profile grassroots #interncrisis campaign, [1] AMSA and the AMA, covered the lapse of planning by jurisdictional health authorities to provide graduates from Australian medical schools with internships. [2]

The reality was significantly more complex and will pose broader questions about the entire medical education continuum. As with many arguments in health, the complexity arises from the divide in funding, and therefore in control, of different levels of medical training.

Medical graduate numbers have increased by more than 200% in the last decade. [3] This increase was fuelled by both increases in Commonwealth supported places, and the unregulated expansion in international student numbers by many universities to cross-subsidise programs. Between 2003 and 2011, vocational medical training places have more than doubled in order to accommodate this growth. [3] However, capacity at all levels has supposedly been reached, with state and territory governments, who fund post-graduate training, now unwilling to match ongoing graduate increases. This has resulted in not only the well-publicised intern crisis but also a lack of post-internship prevocational positions in Queensland and the ever-increasing competition for specialty training places nationally. The disconnect between national and state medical workforce goals has never been clearer.

The crisis has far deeper themes than the simplistic media message typified by statements such as ‘unemployed doctors driving taxis’. As shown with the recent deployment of 60 regional internships, funded by the Commonwealth Department of Health and Ageing, bonding mechanisms are being used to attain better workforce geographic distribution. The ultimate prize is solving the chronic maldistribution of the medical workforce, both in terms of location and specialty. The goal is to create a sustainable medical workforce capable of delivering quality health care to the broader Australian community.

The events occurring at the end of 2012 contrast the conflicting goals of the Commonwealth with those of the states and territories. The Commonwealth Government considered the bigger picture of medical workforce reform and sought to retain the graduates of Australian medical schools, and yet was thwarted by the brinksmanship of the states and territories. The creation of additional medical internships was welcomed but ultimately, given the late hour, saw little uptake, as graduating students activated other plans. This was an opportunity missed but not one that should be forgotten as a new cohort nears graduation. The potential to partner with the private sector to open new pathways for training at both the prevocational and vocational levels is obvious, but, as always, will require funding.

It is clear that states and territories are planning and investing only for their own immediate need, and forgoing the potential of meeting Australia’s medical workforce requirements for short-term budgetary reasons. These decisions will have real world consequences including limiting accessibility to core services, leading to poorer health outcomes and unnecessary hospital admissions, particularly for those already disadvantaged, especially by distance. [4]

There is abundant evidence to support the employment of all Australian medical graduates. Health Workforce 2025 modelled the future health workforce required for Australia and proposed a number of possible futures. [5] In the baseline scenario, based upon current graduate trends, Australia will be short of more than 2000 doctors by 2025. The geographic maldistribution of the medical workforce and poor alignment of training pathways to the health requirements of the community is anticipated to further amplify perceived shortages.

We agree that Australia’s medical training system must change. Coordinating internship allocation at a national level, addressing the bottlenecks of entry into specialty training, and ensuring that community need shapes the medical workforce is just the beginning. Students and doctors-in-training also have an integral responsibility for creating a balanced, responsive medical workforce in their choices of specialty and location of training and practice.

Some action is already underway, with jurisdictions agreeing to the creation of the National Medical Training Advisory Network, but this will not be a panacea. Standing Council on Health meetings need to move beyond perpetuating the blame game, and create a timetable for reform while opening a discourse to consult meaningfully with the profession.

Innovative solutions to training issues must be influenced and informed by the experiences of students and doctors-in-training if they are to have the intended impact on the future medical workforce. Moreover, as the issue descends on states, ensuring that members of parliament are aware of the issues and their implications for the future of health service delivery will be a key opportunity for students to support long-term reform.

Conflict of interest

None declared.

Correspondence

M Bonning: michaelbonning@gmail.com

References

[1] Medical Student Action on Training. Interncrisis. 2013 [accessed 30 March, 2013]. Available from: http://interncrisis.org

[2] Australian Health Ministers’ Advisory Council. Communique 18 June 2012. Perth: AHMAC, 2012.

[3] Australian Government Department of Health and Ageing.  Medical Training Review Panel Fifteenth Report.  Canberra: DoHA, 2012.

[4] Australian Institute of Health and Welfare. Australia’s Health: 2010. Canberra. p.248

[5] Health Workforce Australia. Health Workforce 2025. Volume 3. 2012.

Categories
Letters Articles

National Leadership Development Seminar: developing the health care leaders of the future

The vast field of medicine transcends the mere finding of cures for ailments, seeking approaches to prolonging life, and undertaking research in the pursuit of wellbeing, important as these duties are. However, medicine’s empathetic pledge to the ill requires us to exercise leadership, amongst other qualities, as an important tool to advance the interests of both the individual and the population at large. Practical leadership and advocacy is the cornerstone of the increasingly complex environment in which 21st century healthcare is provided. Patients, institutions and communities often perceive doctors as agents of change i.e. leaders. However, some physicians may have been marginalised by the healthcare system because they either do not receive good leadership and management training, or they occupy positions that require leadership and managerial skills, which they initially do not possess. This apparent lack of appropriate leadership and management development may preclude doctors from participating in essential roles to shape the delivery of health services. [1] The problem can be traced back to medical school, where relatively little, or perhaps non-existent emphasis is given to nourish medical students’ attitudes towards leadership. Current medical curricula offer students little leadership education of the kind considered necessary to develop competences essential in becoming actively involved in the planning, implementation and provision of patient care. [2]

The Australian Medical Students’ Association (AMSA), being the peak advocating body for key affairs that concern medical students across the country, has identified this issue. It has responded by establishing the National Leadership Development Seminar (NLDS), an initiative aimed to assist motivated students interested in leading the medical profession. Each year, since its inception in 2005, NLDS attracts hundreds of applications from bright students who are keen to enhance their leadership skills. The seminar allows for approximately 90 applicants to participate annually. The NLDS program is carefully constructed to equip attendees with knowledge, skills and attitudes regarding leadership, advocacy and management, with a focus on current national health issues. The three-day seminar, which is held in Canberra, integrates guest speaker presentations, small group activities and interactive workshops to teach students how to link necessary leadership competencies with actual service opportunities.

The NLDS focus on leadership is closely aligned with Health LEADS Australia, a health professional leadership framework draft that has recently been published by Health Workforce Australia. The framework describes some of the most important leadership attributes that health workers who are involved in building a flourishing and sustainable health system should embrace and promote. This leadership framework is divided into five arms, including emotional intelligence and self-performance reflection, acknowledging the abilities of others whilst helping them to develop, and concentrating on achieving goals and pursuing innovative change. [3]

Although long term evaluation data is required to assess the effectiveness of NLDS (especially in meeting the key objective competences as outlined by Health LEADS Australia and Domain 4 of the Australian Medical Council’s Graduate Outcome Statements [4]), this program offers an innovative model of a leadership-based course. This can have a positive impact on leadership skills development among medical school students and can be incorporated into the medical school curriculum. We understand that NLDS has some limitations in terms of its primordial structure compared to other more established leadership programs in the realm of business and economics. These limitations include the program’s exclusivity to only a minor number of students per year, non-exhaustive coverage of all aspects of what it takes to become a successful leader in the clinical arena, and lack of networking past the event’s conclusion. Despite these drawbacks, NLDS is a unique national attempt to illustrate the importance of leadership in medical education. We invite medical schools to look at NLDS as a template whilst designing an innovative, socially accountable curriculum to engage students in the practices of advocacy, management and leadership.

Conflict of interest

None declared.

Correspondence

L Ngu: lngu45@student.monash.edu

References

[1] Abbas MR, Quince TA, Wood DF, Benson JA. Attitudes of medical students to medical leadership and management: a systematic review to inform curriculum development. BMC Med Educ. 2011;11:93.

[2] O’Connell MT, Pascoe JM. Undergraduate medical education for the 21st century: leadership and teamwork. Fam Med. 2004 Jan; 36 Suppl:S51-S56.

[3] Health LEADS Australia: Consultation for an Australian Health Leadership Framework: Health Workforce Australia; 2012. [cited July 2013]. Available from: http://hwaleadershipframework.net.au/files/HWA-HealthLeadershipFramework-forConsultation.pdf

[4] Accreditation Standards for Primary Medical Education Providers and their Program of Study and Graduate Outcome Statements: Australian Medical Council; 2012. [cited July 2013]. Available from: http://www.amc.org.au/images/Accreditation/FINAL-Standards-and-Graduate-Outcome-Statements-20-December-2012.pdf

Categories
Case Reports Articles

Acute viral bronchiolitis in the setting of extensive family history of asthma

This case report describes a previously healthy eleven-month old ex-preterm female with a severe presentation of acute viral bronchiolitis with an extensive family history of asthma. The link between viral bronchiolitis and asthma has always been controversial despite extensive research. Several studies have linked respiratory syncytial virus (RSV) bronchiolitis to the development of persistent wheezing or asthma later in childhood, even suggesting that a dose-response relationship may exist between the two entities. Some studies have also demonstrated that severe lower respiratory infections in the first year of life are important contributors to asthma, particular in those sensitized during infancy. On the other hand, it has also been studied as to whether an individual at risk of asthma has any impact on the severity of bronchiolitis. Despite numerous studies, results have largely been inconclusive, and the question of whether it is RSV that directly results in asthma, or if the susceptibility to RSV is conferred due to predisposing pulmonary pathology, still remains unknown.

Case

Sally was an eleven-month old ex-preterm (35 weeks) female who presented to the Emergency Department (ED) with symptoms of fever, coryzal symptoms and a wheeze, subsequently diagnosed as viral bronchiolitis.

History

Sally had become acutely febrile two nights prior to presentation, developing coryzal symptoms the following morning. Sally was initially treated for an upper respiratory tract infection (URTI) and acute otitis media with amoxicillin by her general practitioner, but began to worsen over the subsequent 24 hours, with laborious and wheezy breathing, coupled with a persistent fever of 38.4oC. Despite two doses of salbutamol, her breathing continued to deteriorate, leading to her presentation at the ED. There were no apnoeic or cyanotic episodes, rigors or any associated inspiratory stridor. During this period, Sally was anorexic, with subsequently fewer nappy changes, and was reported by her parents to be far less active than usual. She was previously well, with no known sick contacts, and her vaccinations were up-to-date.

Sally had a similar episode of bronchiolitis in at eight months of age but was treated then as an outpatient. There was an extensive family history of asthma (Figure 1), and both the patient’s siblings had bronchiolitis as infants. At eleven months, Sally was meeting all the developmental parameters for her age. There were no known drug allergies and other than salbutamol PRN, the patient was not on any other medication. There was no remarkable social history.

Examination

Sally appeared lethargic and was in respiratory distress, with tachypnoea, an audible wheeze and classical signs of increased breathing effort (nasal flaring, tracheal tug and subcostal recession). She had a respiratory rate of 78 and was saturating at 100% on 8L supplementary oxygen, which decreased to 90% on room air, and was febrile at 38.5oC. She was tachycardic at a heart rate of 170. There were no clinical signs of dehydration. On auscultation, air entry was reduced bilaterally, with an expiratory wheeze and diffuse crackles present. There was no evidence of stridor, increased vocal resonance, or dullness to percussion. An ear, nose and throat examination revealed an erythematous pharynx but was otherwise unremarkable. All other examination findings were unremarkable.

Workup and Progress

The presenting symptoms suggested a diagnosis of acute viral bronchiolitis. However, with the extensive family history of asthma, Sally’s presentation could be her first virus- triggered asthma attack. While the clinical presentation suggested otherwise, there were concerns over the possibility of pneumonia, which had to be ruled out in the workup. In consideration of the severity of her initial presentation and the likely further deterioration until its peak at day 2-3, basic investigations were ordered. These included a full blood count, urea, electrolytes and creatinine parameters, a chest X-ray and a nasopharyngeal aspirate. There were no abnormal findings.

Sally was commenced on immediate supportive therapy. An IV line was inserted and she was commenced on 75% maintenance fluids as per guidelines to avoid Syndrome of Inappropriate Antidiuretic Syndrome. [1] She was also commenced initially on 2L oxygen as per guidelines for respiratory distress, [2] but failed to saturate appropriately until given 8L of humidified oxygen via nasal prongs, where she maintained 100% O2 saturation. While the efficacy of short-acting beta agonists (SABAs) in the acute management of bronchiolitis has been inconclusive despite extensive research, current guidelines recommended a trial of bronchodilators in infants >6 months. [3] Sally was administered six puffs of Salbutamol MDI (100mcg/ puff) via spacer [1] but with no results, hence the regime was discontinued.

The Royal Children’s Hospital (RCH) provides further guidance regarding management based on clinical signs and symptoms. Admission to the intensive care unit (ICU) was indicated for Sally to allow continuous cardiorespiratory monitoring and supportive management. Observations were performed hourly and with only supportive management, her oxygen requirements were weaned down to 2L over 24 hours.

Discussion

Bronchiolitis during infancy and asthma in childhood – is there a causal link? Should infants at high risk of asthma receive Palivizumab immunization?

Several studies have linked respiratory syncytial virus (RSV) bronchiolitis to the development of persistent wheezing or asthma later in childhood. In a long-term prospective cohort study, there was a relative risk of 2.8 of developing wheezing at 5.5 years in children who had had bronchiolitis. [4] Sigurs et al. (2005) also reported a ten-fold excess of asthma in a similar study. [5]

It has also been suggested that a dose-response relationship exists between bronchiolitis and asthma. In a study involving 90,341 children, Carroll et al. (2009) demonstrated that the odds ratios (OR) for asthma as a child were 1.86 (95% CI, 1.7-2.0), 2.41 (95% CI, 2.2-2.6) and 2.8 (95% CI, 2.6-3.0) in the outpatient, ED, and hospitalization groups, respectively, compared to children without bronchiolitis. [6] Henderson et al. (2005) also noted an OR of 2.5 (95% CI 1.4-4.3) of developing asthma with hospitalization for RSV bronchiolitis, [7] as did two prospective studies, which showed a 30-40% likelihood of subsequent asthma. [8]

RSV bronchiolitis as a direct cause of asthma

In an extensive seven-year REBEL prospective cohort study, Bacharier et al. (2012) reported that increased Chemokine (C-C motif) Ligand 5 (CCL5) expression in nasal epithelial cells during RSV infection carried an OR of 3.8 (95% CI, 1.2-2.4) for developing asthma. [9] This is in concordance with studies demonstrating increased CCL5 levels in subjects with asthma, [10] as well as in vitro studies demonstrating increased expression and transcription by RSV. [11] Unfortunately, the study failed to measure CCL5 levels prior to infection and thus the causal relationship has not been established. Hence, whether it was RSV that directly resulted in asthma, or if the susceptibility to RSV was conferred due to predisposing pulmonary pathology, [12] still remains unknown.

A five-year cohort study on children at high risk of atopy by Kusel et al. (2007) demonstrated that severe lower respiratory infections in the first year of life are important contributors to asthma, particular in those sensitized during infancy. [13] These findings suggest that protecting high-risk individuals from infection during infancy may be considered for long-term asthma prevention.

Effect of family history of asthma or atopy on severity of bronchiolitis

It has also been studied whether an individual at risk of asthma has any impact on the severity of bronchiolitis. This was particularly relevant in the Sally’s case, with her significant family history of asthma. Results in this field have been conflicting, with most studies not eliciting any significant association. However, Gurwitz et al. (1981) demonstrated that hospitalized cases were associated with a higher incidence of first-degree relatives with bronchial hyper-responsiveness. [14] A study by Trefny et al. (2000) also demonstrated similar results. [15]

Should high-risk atopic individuals receive Palivizumab immunization during RSV season for prevention of asthma?

Passive immunization with Palivizumab is currently recommended only for high-risk infants to prevent serious complications arising from RSV infections. [16] However, a recent double-blinded RCT in the Netherlands has begun examining its preventive effect on recurrent wheeze in healthy preterm children 33-35 weeks gestational age (MAKI trial), based on a non-randomized trial suggesting a prevention of wheeze in 50% of preterm children. [17] Such a study would complement this case study’s patient profile, and would be especially relevant in the context of her rich family history of asthma which puts her at high risk, and the abovementioned association but inconclusive causation between bronchiolitis and asthma.

From an economic standpoint, studies assessed the cost-effectiveness of Palivizumab, albeit in the context of high-risk premature infants (32-35 weeks). Unfortunately, the predisposition of these infants to a higher disease burden and costlier hospitalizations constitutes a higher cost per QALY [18]      compared to this case study’s patient, but even then there is still considerable controversy over its cost-effectiveness, especially across various healthcare systems.

Conclusion

In summary, this was a case of severe viral bronchiolitis warranting ICU admission for supportive management, on a background of an extensive family history of asthma. While studies have shown a clear association of bronchiolitis with asthma, causation has not been conclusively established, with family history of atopy possibly interacting in the development of asthma. Current research is lacking in the area of Palivizumab prophylaxis in the interest of asthma prevention in healthy children, but the evidence would suggest that it is likely to be cost-ineffective.

Conflict of interest

None declared.

Correspondence

G Yong: glenn.yong.kw@gmail.com

References

[1] Royal Children’s Hospital. Bronchiolitis- Ongoing Management. http://www.rch.org.au/rchcpg/hospital_clinical_guideline_index/Bronchiolitis_Ongoing_Management/#Fluid_requirements (accessed 11 March 2013)

[2] Royal Children’s Hospital. Oxygen Delivery. http://www.rch.org.au/rchcpg/hospital_clinical_guideline_index/Oxygen_delivery/ (accessed 11 March 2013)

[3] [Guideline] Diagnosis and management of bronchiolitis. Pediatrics. 2006 Oct;118(4):1774-93.

[4] Murray M, Webb MS, O’Callaghan C, Swarbrick AS, Milner AD. Respiratory status and allergy after bronchiolitis. Arch Dis Child. 1992 April;67(4): 482-7.

[5] Sigurs N, Bjarnason R, Sigurbergsson F, Kjellman B. Respiratory syncytial virus bronchiolitis in infancy is an important risk factor for asthma and allergy at age 7. Am J Respir Crit Care Med. 2000;161:1501–7.

[6] Carrol KN, Wu P, Gebretsadik T, Griffin MR, Dupont WD, Mitchel EF, Hartert TV. The severity- dependent relationship of infant bronchiolitis on the risk and morbidity of early childhood asthma. Journal of Allergy & Clinical Immunology 2009;123:1055-61.

[7] Henderson J, Hilliard TN, Sherriff A, Stalker D, Al Shammari N, Thomas HM. Hospitalization for RSV bronchiolitis before 12 months of age and subsequent asthma, atopy and wheeze: a longitudinal birth cohort study. Pediatric Allergy Immunology. 2005;16:386–92.

[8] Sign AM, Moore PE, Gern JE, Lemanske RF, Hartert TV. Bronchiolitis to asthma A review and call for studies of gene-virus interactions in asthma causation. Am J. Respir. Crit Care Med. January15 2007;175(2):108-19.

[9] Bacharier LB, Cohen R, Schweiger T, Yin-Declue H, Christie C, Zheng J et al. Determinants of asthma after severe respiratory syncytial virus bronchiolitis. Journal of Allergy & Clinical Immunology 2012;130:91-100.

[10] Humbert M, Ying S, Corrigan C, Menz G, Barkans J, Pfister R et al. Bronchial mucosal expression of the genes encoding chemokines RANTES and MCP-3 in symptomatic atopic and nonatopic asthmatics: relationship to the eosinophilactive cytokines interleukin (IL)-5, granulocyte macrophage-colony-stimulating factor, and IL-3. Am J Respir Cell Mol Biol 1997;16:1-8.

[11] Koga S, Novick AC, Toma H, Fairchild RL. CD81T cells produce RANTES during acute rejection of murine allogeneic skin grafts. Transplantation 1999;67:854-64.

[12] Adamko DJ, Friesen M. Why does respiratory syncytial virus appear to cause asthma? Journal of Allergy & Clinical Immunology 2012 Jul;130(1):101-2.

[13] Kusel MM,  de Klerk NH, Kebadze T Vohma V, Holt PG, Johnston SL et al. Early-life respiratory viral infections, atopic sensitization, and risk of subsequent development of persistent asthma. Journal of Allergy & Clinical Immunology 2007;119:1105-10.

[14] Gurwitz D, Mindorff C, Levison H. Increased incidence of bronchial reactivity in children with a history of bronchiolitis. J Pediatr 1981;98:551–5.

[15] Trefny P, Stricker T, Baerlocher C, Sennhauser FH. Family history of atopy and clinical course of RSV infection in ambulatory and hospitalized infants. Pediatric Pulmonology 2000;30:302–6.

[16] Wang D, Byliss S, Meads C. Palivizumab for immunoprophylaxis of respiratory syncytial virus (RSV) bronchiolitis in high-risk infants and young children: a systematic review and additional economic modelling of subgroup analyses. Health Technol Assess 2011 Jan;15(5):1-124.

[17] Broughton S, Bhat R, Roberts A, Zuckerman M, Rafferty G, Greenough A. Diminished lung function, RSV infection, and respiratory morbidity in prematurely born infants. Arch Dis Child Jan 2006;91(1):26-30.

[18] Smart KA, Paes BA, Lanctot KL. Changing costs and the impact of RSV prophylaxis. Journal of Medical Economics. 2010;13(4):705-8.