Child health

Jennifer A O'Dea Senior Lecturer, Faculty of Education, University of Sydney, Building A35, Sydney, NSW 2006. j.o'deaATedfac.usyd.edu.au To the Editor: As part of a large, national nutrition study, height and weight were measured among 4441 students from 38 schools randomly selected from lists of all state and territory schools in Australia in 2000. Public, private and Catholic schools, in both rural and urban areas, were represented. Schools were categorised as being of low or middle/high socioeconomic status (SES),1 based on direct measurement of parental income. Parental consent was obtained, and the study was approved by the University of Sydney Ethics Committee and all state departments of education. Overweight and obesity, as defined by an international standard definition,2 were identified in 17.3% and 6.4% of participants, respectively. These characteristics showed a trend towards greater prevalence among students from low-SES backgrounds compared with those from middle/high-SES backgrounds for the total group (19% v 16.8% overweight [P = 0.09]; 8.9% v 5.8% obese [P = 0.02]), females (19.7% v 17.2% overweight [P = 0.2]; 6.9% v 6.2% obese [P = 0.56]), and males (18.5% v 16.3% overweight [P = 0.23]; 9% v 5.5% obese [P = 0.003]), although not all differences were statistically significant. After controlling for SES differences in age and height, mean body mass index (BMI) was significantly higher among low-SES than middle/high-SES participants for the total group (20.3 kg/m2 [95% CI, 20.1–20.5 kg/m2] v 19.7 kg/m2 [95% CI, 19.6–19.9 kg/m2]; P < 0.001), females (20.4 kg/m2 [95% CI, 20.1–20.7 kg/m2] v 19.8 kg/m2 [95% CI, 19.6–19.9 kg/m2]; P < 0.001), and males (20.2 kg/m2 [95% CI, 20.0–20.5 kg/m2] v 19.6 kg/m2 [95% CI, 19.5–19.8 kg/m2]; P < 0.001). A breakdown of results by SES, sex and school level is shown in the Box. Low-SES primary school children were also 1–2 cm shorter, on average, than middle/high-SES primary school children (boys: mean 141.5 cm [95% CI, 140.6–142.5 cm] v 143.5 cm [95% CI, 143.0–144.0], P < 0.001; girls: mean 141.0 cm [95% CI, 140.8–142.6 cm] v 143.3 cm [95% CI, 142.5–143.6 cm], P = 0.01). The average proportions of overweight and obese children and adolescents in the study were similar to those found in other Australian studies.3-5 The results suggest that SES is a factor in the development of overweight and obesity among Australian school children. This may be a relatively recent trend, as these data were obtained in late 2000. Low SES in children may also be associated with nutritional deprivation and height retardation. Further research should clarify these relationships among children from low, middle and high SES backgrounds, as well as examining the combined impact of both SES and ethnicity. School students classified as overweight or obese* according to socioeconomic status (SES), school level and sex Males (n = 2232) Females (n = 2209) Low SES (n = 574) Middle/high SES (n = 1658) Low SES (n = 508) Middle/high SES (n = 1701) Primary school students (grades 1–6; ages 6–13 years) Overweight students 19.4% (42/216) 16.2% (110/680) 23.2% (51/220) 17.8% (136/766) Obese students 6.9% (15/216) 5.3% (36/680) 6.4% (14/220) 5.7% (44/766) High school students (grades 7–12; ages 13–18 years) Overweight students 17.6% (63/358) 16.4% (160/978) 17.0% (49/288) 16.8% (157/935) Obese students 10.1% (36/358) 5.6% (55/978) 7.3% (21/288) 6.5% (61/935) * Overweight and obesity are classified according to the international standard definition.2

Jennifer A O'Dea

Tony D Merritt,* Carolyn Herlihy† * Epidemiologist/Biostatistitian, † Environmental Health Officer, Hunter Public Health Unit, Hunter Area Health Service, LMB 119, Wallsend, NSW 2287. tmerrittATdoh.health.nsw.gov.au To the Editor: Travelling animal shows, with animals such as young poultry, rabbits and reptiles, commonly visit childcare centres in Australia. Transmission of Salmonella infection to children from ducklings and chickens is well documented in the United States1,2 and United Kingdom,3 but not in Australia. We investigated a cluster of Salmonella agona cases in children, identified through routine laboratory surveillance. Initial investigations identified a potential association with visits to childcare centres by a single local hatchery. At each show, children saw an egg hatching, watched day-old ducklings swim and had the opportunity to hold a day-old chick or duckling, and to touch an adult chicken. Details of gastrointestinal illness were sought from childcare centres that had hosted visits from the hatchery. This identified laboratory-confirmed cases with onsets between 14 April and 6 May 2002 in seven people. All attended one of four childcare centres; six were children and one was a staff member. Onset of illness occurred 2–12 days after the hatchery visit. A total of 316 children attended shows by the hatchery between 9 April and 8 May. Environmental sampling at the hatchery recovered Salmonella agona from multiple sites over repeated visits. These included the egg incubator, faeces from day-old hatchlings returning from a show with children, faeces from young chicks and ducklings in the brooder cage, duck faeces from their yard, corn meal used in the preparation of feed for all poultry, and rat droppings in the feed preparation and storage areas. No Salmonella were detected in samples collected from the corn meal supplier. New procedures based on guidelines from South Australia4 and the Centers for Disease Control and Prevention5 were introduced for all shows from 8 May. These included supervised handwashing after handling animals, avoiding carpeted areas for the show, adequate cleaning of macroscopic faecal contamination and disposal of the water used by swimming ducklings into the sewerage system. No further cases were identified among the 251 children who attended shows over the subsequent two weeks until the hatchery cancelled all further visits. We conclude that infection was acquired through contact with chicks and ducklings from a single hatchery, and that the outbreak was halted by the introduction of measures emphasising handwashing after animal contact. Contact between young children and young animals, both of whom have naïve immune systems, represents a special ecological niche. Young poultry are particularly vulnerable to salmonella infection and subsequent high level excretion, and young children are similarly vulnerable. As animal visits to childcare centres are highly valued by children and carers, appropriate guidelines should be widely promoted to the childcare sector and the petting zoo industry to reduce the risks these visits pose. The petting zoo guidelines developed in South Australia4 are an excellent resource and NSW Health is currently developing a fact sheet on the topic.

Tony D Merritt · Carolyn Herlihy

The epidemic is subsiding as we improve the delivery of assisted reproductive technology The multiple birth rate in Victoria rose from 12 to 17 per 1000 pregnancies between 1986 and 1997.1 There were comparable increases in the United Kingdom and United States. During this period, the rate of all multiple births increased, but the more significant increases were in high-order multiple pregnancies. In Victoria, the triplet pregnancy rate increased more than three-fold from 0.19 to 0.61 per 1000 pregnancies between 1986 and 1998.1 There was a similar three-fold rise in the rate of triplet pregnancies in the United Kingdom,2 while in the United States, the rise was six-fold for triplets and 12-fold for quadruplets.3 These rates of multiple pregnancies peaked at the end of the century and are now slowly falling. There are several reasons why there has been an increase in twin pregnancies and the "epidemic" of high-order multiple pregnancies with low birth rates in the developed world. Monozygotic twinningMonozygotic twinning occurs independently of ethnicity, maternal age, parity, nutritional status and environmental factors. It is a random genetic event occurring in 1 in 250 pregnancies. Although the incidence of monozygotic twinning may be doubled after induction of ovulation, and is increased with in-vitro fertilisation for reasons that are yet to be explained, monozygotic twinning has contributed minimally to the global epidemic of multiple pregnancy. 4,5 Dizygotic twinningIn developed countries, there was a global decline in dizygotic twinning rates from 1960 until the mid-1970s. Although the exact cause remains unknown, this decline has been attributed to either environmental pollutants or a reduction in sperm quality.6 Chronologically, the fall in dizygotic twinning can be attributed to reduced fertility after oral contraceptive pill use became widespread. From the 1980s, the steady increase in dizygotic twinning related to agents that induce ovulation may have masked a true, progressive decline in the spontaneous dizygotic twinning rate.6 The rate of dizygotic twinning is increased in women aged 35–39 years, with higher parity and tall stature. Rates fall in severely malnourished women. Dizygotic twinning runs in families. If a mother or a sister has dizygotic twins, a woman has double the risk of having dizygotic twins herself. If she has dizygotic twins herself, her risk of having future dizygotic twins is quadrupled.7 Folic acid supplementation at the time of conception is widely promoted for reducing the risk of neural tube defects. A systematic review of periconceptual supplementation with folic acid or multivitamins, or both, identified a tendency to an increased risk of twinning (pooled relative risk 1.40; 95% CI, 0.93–2.11).8 It is unclear why such an association exists; possibly folic acid improves early fetal survival rather than promoting multiple ovulation. Assisted reproductionThe most significant cause for the increase in the multiple birth rate in developed countries has been the use of assisted reproductive technology. Inducing ovulation with clomiphene citrate carries an 8% risk of a multiple pregnancy, and with gonadotrophins, a 20% risk. These agents (which act by hyperstimulation of ovarian follicles resulting in one or more oocytes being released per cycle) are responsible for most high-order multiple pregnancies. Ultrasound monitoring can detect the potential for multiple ovulation in a woman's cycle, and she can be advised accordingly. However, even with ultrasound monitoring, and in the most careful and experienced hands, multiple ovulations can occur. With in-vitro fertilisation (IVF) and gamete intrafallopian transfer (GIFT), multiple pregnancy rates vary with maternal age and the number of embryos transferred; a 20% multiple pregnancy rate with double embryo transfer and 25% with triple embryo transfer is typical. The increased use of microinjection techniques has seen an increase in monozygotic multiple pregnancies. Improvements in stimulation and laboratory culture techniques have improved embryo quality and, subsequently, the success of IVF and GIFT, particularly in recent years. Risks of multiple pregnancyObstetric complications occur more frequently in multiple pregnancies (Box 1), and perinatal mortality escalates with increasing fetal number. The most recent Victorian figures indicate a perinatal mortality of 9.2 per 1000 pregnancies for singletons, 42.9 per 1000 for twins (first twin 37.8; second twin 47.9), and 145.5 per 1000 for triplets (first triplet 145.5; second triplet 127.3; third triplet 163.6).1 Preterm delivery is the major cause of adverse outcomes (both short-term and long-term), and is directly related to fetal number (Box 2). The consequences of prematurity (including cerebral palsy, hearing and visual disturbance, behavioural disorders and respiratory disease, among many others) can result in significant social, emotional and financial burdens for families. Costs to the community also increase with the number of infants. Monochorionic twins have specific risks because they share a placenta. These risks are twin–twin transfusion syndrome, twin reversed arterial perfusion sequence, monoamnionicity and conjoined twins. All of these conditions have very significant risks of fetal and neonatal mortality and morbidity. The risks associated with being born in a multiple pregnancy do not end with delivery. Apart from the consequences of prematurity, or the morbidity associated with monochorionicity, children born as twins or in higher order multiple pregnancies have increased rates of neonatal death, speech and reading difficulties, and behavioural disorders including attention deficit hyperactivity disorder. Twins have a four-fold, and triplets a 20-fold, increase in cerebral palsy compared with singletons. If a twin develops cerebral palsy, the risk of its co-twin developing cerebral palsy is 12%. Death of a co-twin increases the risk of cerebral palsy for the survivor to around 5% with the risk rising to 40% with monochorionicity.10,11 Preventing multiple pregnancyAll women undergoing ovulation induction should be offered monitoring of follicular development. They should be fully advised of the potential for a multiple pregnancy, and should be made aware of the consequences of pursuing a conception when multiple follicles are present. It is axiomatic that the fewer the number of embryos transferred after IVF, the lower the risk of a multiple pregnancy. It has been recent practice to offer women the transfer of only two embryos to optimise the chance of pregnancy without significantly increasing the risks of a multiple pregnancy. This practice also helps reduce the costs to the patient associated with repeated embryo transfers. However, with the continuing improvement in pregnancy rates with IVF, single embryo transfer should now be considered by all women, particularly those who are younger and those who already have children. Multifetal pregnancy reduction (MFPR) by intracardiac potassium chloride injection to reduce the number of "excess" fetuses is often seen as the answer to reducing the risks and avoiding the complications of high-order multiple pregnancy. Notwithstanding the social, moral and ethical issues associated with this technique, there are significant risks to the remaining fetuses associated with the procedure. For example, MFPR from a triplet to a twin pregnancy is associated with an 8% risk of miscarriage of the remaining twins. However, the procedure offers a marginal reduction in perinatal mortality and reduces the handicap rate from 1.5% to 0.6% per fetus.12 ConclusionsMultiple pregnancy usually has a satisfactory outcome, culminating in the birth and development of healthy children, often long awaited, and much loved by their parents. However, the consequences of some multiple pregnancies for the parents, the children and the community remain significant. Multiple pregnancies reached epidemic proportions in the late 1990s as a consequence of assisted reproductive technology. The rate is now falling and this trend should continue as practitioners respond with careful monitoring of ovulation induction and with reduced numbers of embryos transferred after IVF. 1: Significant risks associated with twin pregnancies1,2,9 Obstetric complication Risk* Anaemia x2 Pre-eclampsia x3 Eclampsia x4 Antepartum haemorrhage x2 Postpartum haemorrhage x2 Fetal growth restriction x3 Preterm delivery x6 Caesarean section x2 * Compared with singleton pregnancies 2: Rates of preterm delivery by fetal number1 Delivery Number of fetuses < 28 weeks < 37 weeks Singleton 0.7% 6.2% Twins 4.4% 52.1% Triplets 21.8% 98.2%

Mark P Umstad MB BS, MD, MRCOG, FRANZCOG · Michael J Gronow MB BS, MD, MRCOG, FRANZCOG

To the Editor: Spilsbury et al argue that "improved education for physicians, and perhaps parents, with regard to foreskin development and management is required."1 However, updating of textbooks and medical curricula is required to accomplish this objective. Articles by Caldamone et al2 and Cendron et al3 are two examples of incorrect data in text books. Gairdner was the first to provide data on the normal development ...

George Hill

To the Editor: The study by Spilsbury and colleagues provides new data on rates of phimosis, balanoposthitis, and lichen sclerosus.1 However, it does cite sources — Øster2 and Shankar and Rickwood3 — that indicate very low rates of phimosis. Øster was a school medical officer who followed up his subjects for several years, making frequent penile inspections and giving them continual instruction on prepuce care. ...

Stefan A Bailis

To the Editor: In the recent article by Spilsbury and her colleagues on circumcision for phimosis, a key part of their argument hinged on probable rates of phimosis among boys.1 I take no stance for or against circumcision, but I have published on evolutionary aspects of the human foreskin and the origins of circumcision,2 for which I surveyed the literature on the occurrence of phimosis. Spilsbury ...

Guy Cox

To the Editor: The article by Spilsbury et al starts well by acknowledging at least some of the serious health consequences of not circumcising,1 but then digresses into a study of whether a particular medical reason for circumcision — namely phimosis early in life — has been overstated in medical records. So what! Circumcision is a simple procedure that conveys significant lifetime health benefits. Like immunisation, ...

Brian J Morris

In reply: We support Hill in his call for improving the dissemination of information and data about foreskin development and management. Circumcision is a highly emotive issue. Our study on phimosis1 was carried out under the Western Australian Safety and Quality of Surgical Care Project, established in 1996, to assess the safety, quality, appropriateness and outcomes of surgical care in the state. The purpose of our ...

James B Semmens

To the Editor: While I generally applaud the comments on phimosis in Dewan's recent editorial,1 there are two points on which I would take issue with him — one historical, one ethical. Firstly, the incidence of circumcision in Britain never reached anything like 95%, and most of the decline in the practice occurred in the 1940s. According to surveys in the 1940s and 1950s,2 even at ...

Robert JL Darby

In reply: I thank Darby for his correction of the historical facts, and for raising for further discussion the ethics of circumcision. While I concur that removal of the prepuce for cosmetic reasons may not be in the interests of the child, we do need to allow for the parents' perceptions, and for their willingness and ability to care for the prepuce appropriately, if ...

Paddy A Dewan

Objectives: To describe the short-term outcomes in Aboriginal children admitted to hospital with radiological alveolar lobar changes; and determine whether predischarge chest radiography can predict respiratory morbidity found at follow-up.Design, participants, setting: Prospective cohort study of Aboriginal children admitted to Alice Springs Hospital between October 2000 and April 2001 with alveolar lobar abnormalities (area of consolidation, ≥ 1 cm) on chest radiographs. Participants were to have a predischarge radiograph and be followed up for 12 months.Main outcome measures: Comorbidities, follow-up rate, and new respiratory disease found at follow-up.Results: Of 113 children hospitalised with radiological alveolar lobar changes, 109 were Aboriginal. Their median age was 1.8 years (range, 0.2 months–13.3 years), and 124 episodes were recorded. Comorbidities were common in these children (anaemia, 51.5%; suppurative otitis media, 37.3%). The follow-up rate one year after admission was 83.1% of episodes. New treatable chronic respiratory morbidity was found in 20 (25.6%) of the 78 children with completed follow-up. Predischarge chest radiographs were predictive of all chronic respiratory morbidity when they showed no or minimal resolution (0–20% resolution) (relative risk, 7.43; 95% CI, 2.07–26.60).Conclusions: Central Australian Aboriginal children admitted to hospital with alveolar changes on chest radiographs have a substantial burden of chronic respiratory illness, and should be clinically followed up for early detection and management of chronic respiratory morbidity. A predischarge radiograph is useful, and patients whose radiograph shows no or minimal resolution should have a follow-up x-ray film.

Anne B Chang MPHTM, PhD, FRACP · John P Masel MB BS, FRACR · Naomi C Boyce BNursing · Paul J Torzillo MB BS, FRACP, FFICM

Cries unheard. A new look at attention deficit hyperactivity disorder. George Halasz, Gil Anaf, Peter Ellingsen, Anne Manne, Frances T Salo. Alton, VIC: Common Ground, 2002 (x+91pp). ISBN 1 86335 497 2. There have been controversies about the existence and nature of attention deficit hyperactivity disorder (ADHD) since George Still first described it in 1902. The “new look” presented here is the notion that the diagnosis is used as a quick fix to deal with complex situations, allowing one to write a prescription rather than attempt to understand a complex psychodynamic problem. There can be no objection to this approach as our profession has some expertise in producing epidemics which disappear as rapidly as they have appeared — consider the rise and fall of repetitive strain injury (RSI). This book argues that there are many pressures in modern society urging us to find a quick fix, ranging from the activities of the pharmaceutical companies, to the DSM-IV-driven push to confine human distress within categorical boundaries. This is true, however one must examine the total situation, and this is where the authors’ bias shows. Being psychodynamically oriented, they see it as likely that wisdom is to be found in that direction. So it may be, but it is not many years since their antecedents proclaimed that schizophrenia was due to the activities of “schizophrenic” mothers, autism to cold-hearted parents, and the “psychosomatic” disorders to psychodynamic mechanisms. For example, Alexander regarded ulcerative colitis as a regression to the anal stage of psychosexual development, while Szasz and Cushing emphasised the orality of the disease. There were descriptions of the “typical conflict situation” associated with peptic ulcer and asthma. No theoretical position has a mortgage on wisdom. There are some facts about ADHD which will not go away. They emerge most clearly when one deals with adults who are able to give a good account of themselves. It has a firm correlation with dyslexia and anomalies of motor dominance, such as being left-handed and right-footed. Recent work suggests that chromosome 6p may be involved. There are well-established neuroimaging anomalies and there is the paradox that the hyperactivity calms with stimulants instead of being exacerbated. By all means let us look carefully, but let us look in all possible directions. John H T EllardPsychiatrist Balmoral Beach, NSW

John H T Ellard

The overarching cause is energy imbalance The health and wellbeing of Australia's children and adolescents, now and in the future, is under threat. In 2002–2003, the most prevalent child health issues affecting children are preventable: obesity, dental disease, emotional and behavioural problems, bullying and learning delays. These problems often present as comorbidities. Overweight and obesity affect about 23% of Australian children and adolescents, with 6% being obese.1 These are conservative estimates, as there has been no systematic monitoring of the prevalence of overweight and obesity in Australian children and adolescents since 1995. However, over the previous decade, the prevalence of overweight children almost doubled, and the prevalence of obese children more than tripled.1,2 There is no reason to believe that the rapid rise in prevalence rates has not continued. Studies of historical datasets have also revealed that the prevalence of overweight and obesity in children and adolescents doubled over the period 1985–1997, a far greater rate of increase than in the preceding 16 years.3 Health inequalities related to overweight and obesity are evident. There is a higher incidence of overweight and obesity in children of parents of particular backgrounds,3 and maternal education is the strongest social determinant of overweight and obesity in childhood.4 Although there are limited national data, and combined New South Wales, Victorian and National Nutrition datasets1 failed to find a rural/urban difference, Victorian epidemiological data show a statistically significant, higher proportion of overweight and obese boys in metropolitan areas, but this difference was not found for girls (Ms K Hesketh, NHMRC PhD Scholar, Centre for Community Child Health, Melbourne, VIC, personal communication). The health consequences of overweight and obesity are substantial, although Australian data remain unclear in certain areas.5 At least in the United States, obesity carries more stigma in children than any physical disability, and this is evident across all socioeconomic and ethnic groups.6 Issues of social acceptance, athletic competence and physical appearance are well known to obese children and affect their sense of social and psychological wellbeing. Obese children with decreasing self-esteem are more likely to smoke and drink alcohol compared with those whose self-esteem increases or remains the same.7 Obese children and adolescents may also have a range of medical conditions including hypertension, dyslipidaemia, and even type 2 diabetes. Other problems, such as musculoskeletal discomfort, obstructive sleep apnoea, heat intolerance, asthma and shortness of breath, greatly affect their lifestyle.8 Implications for the future can be gathered from longitudinal studies. Combined cohort studies indicate that relative body weight is sustained from childhood to adulthood, and, once children or adolescents are overweight or obese, their weight is unlikely to track backwards.5 If this is not sufficient reason for concern, reflect that these studies (of the long-term consequences of child and adolescent obesity) were all performed before the worldwide obesity epidemic developed. What, then, will be the outcome, in 10 or 20 years' time, of large numbers of children and adolescents entering adulthood, already with abdominal obesity and well established risk factors for cardiovascular disease and type 2 diabetes? Focusing on children highlights their contribution to contemporary society and future populations. Addressing the determinants of health and wellbeing for children and adolescents will improve population health and wellbeing overall. The overarching cause of the obesity epidemic is energy imbalance — a relative increase in energy intake (food intake) together with a decrease in energy expenditure (decreased physical activity and increased sedentary behaviour). Identifying the most important predictive determin-ants of each of these behaviours, as well as the most effective and sustainable remedial strategies, is complex and involves parental education and employment; housing environments; play, recreation and physical activity; food and nutrition; accessible active transport; and child-friendly physical and social environments.9 Some simple trends suggest relatively amenable remedies. Children's fruit and vegetable consumption has decreased over the past 20 years. Their physically active time has also decreased, while time spent in sedentary activities such as television watching and computer games has increased. Finally, consumption of energy-dense foods (including sweet soft-drinks and snack bars with a high sugar content) has increased. Possible remedies include: parental education strategies regarding healthy food choices, activity options, obesity trends, as well as supportive behavioural change strategies; supportive policies and environments in the places children and families spend their time (child care, school, workplaces, home, local neighbourhoods); and prioritisation of free time for physical activities. Evidence from controlled trials (although these trials are heterogeneous as regards the age groups and settings studied) highlights the potential for school-based programs that promote physical activity, modify dietary intake and reduce sedentary behaviours. However, recent qualitative research indicates that differences in outcomes will only be achieved if sustainable changes involve all generations, tackle the widely held beliefs regarding eating and activity,10 involve population-wide health promotion messages, and dispel myths such as children's overweight being just "puppy fat". Further, there are environmental aspects that are well beyond an individual family's ability to modify, including: regulation of marketing of unhealthy food choices for children; provision of safe, cheap and accessible public transport; and urban planning initiatives that give priority to child-friendly and pedestrian-friendly environments. The latter options are more controversial, and vested interests may seek to cloud the community's perceptions of factors driving the overweight epidemic. We need to actively involve industry in partnerships for environmental change. Health practitioners working in the community, child and family nurses and general practitioners are crucial in any comprehensive strategies, as they provide a widely available service to families and can tailor specific strategies for individual families.11,12

Elizabeth B Waters MPH, DPhil · Louise A Baur PhD, FRACP

The evidence for behavioural therapy Autism is a developmental disorder characterised by impairment of communication and social interaction, and stereotyped, restricted patterns of behaviour. The young child with autism fails to develop normal language and imaginative play. Autism (or autistic disorder) affects one in 1000 children and is the core disorder of a wider spectrum of pervasive developmental disorders. Australian paediatricians identify it as one of the more difficult areas of practice1 — there is still no cohesive explanation for the child's developmental arrest, and a plethora of therapies exist. Diagnosis needs to be made by a multidisciplinary team. Parents then face a long list of possible interventions, and will usually be directed first to speech pathologists. Sensorimotor integration therapy (which stimulates or desensitises visual, auditory and tactile senses), and dietary interventions (eg, casein and gluten exclusion) are widely practised in Australia, but data for their efficacy are inadequate.2,3 A controlled trial of auditory integration (where the patient listens to music that has been computer modified to remove frequencies to which he or she is hypersensitive) showed no effect, yet it continues to be offered as a therapy.2 While ineffective therapies may be harmless, they waste parents' money and the child's valuable therapy time. Furthermore, the delay in implementing effective treatment may compromise the child's outcome. Augmented communication, using visual modes such as pictures, symbols and signs, promotes communication and language in children with severe communication deficits and poor verbal imitation skills.4 However, the early intervention that has been subjected to the most rigorous assessment is behavioural intervention. There is now definite evidence that behavioural intervention improves cognitive, communication, adaptive and social skills in young children with autism. In 1987, Lovaas showed apparent recovery, persisting into adolescence, in nine of 19 young children who received an intensive home-based intervention based on applied behavioural analysis, a scientific method of reinforcing adaptive and reducing maladaptive behaviours.5,6 Subsequent studies also showed that behavioural intervention caused significant, albeit somewhat lesser, gains.7-11 This has modified the orthodox view that autism is always a severe, lifelong disability. Criticisms of the adequacy of the design and power of these studies are being addressed by the multisite Lovaas replication Early Autism Project. The first US site has released data (Wisconsin Early Autism Project).12 Again, after three to four years of intensive applied behavioural analysis intervention, about half the preschool children with autism acquired near-normal functioning in language, performance IQ and adaptability. Ninety-two per cent of intervention children acquired some language. Control children who received special education showed no gains in IQ or adaptability.12 Why is intensive applied behavioural analysis intervention more effective than special education for children with autism? This can not be simply explained by the intensity of these programs (30–40 hours per week). Children in a school-based Scandinavian study who received behavioural intervention gained an average of 25 language IQ points in the first year of the intervention, with improvements in performance IQ, communication and adaptability. On all scores, they surpassed control children who received special education according to best practice for autism, and the same intensity, duration and supervision of therapy.13 The superior outcome from behavioural intervention is thought to result from the targeting of specific deficits in autism that prevent learning: imitation, attention, motivation, compliance, and initiation of interaction. Skills are taught in small steps, mastered, and then generalised. Intensive, individualised one-to-one therapy is usually provided by students, behavioural therapists, or parents, under the supervision of behavioural experts. More natural settings of play and learning, augmented communication support, and other powerful visual learning tools, such as video modelling, may be used. Parents play a major coordinating role, and are trained to generalise the skills learnt by the child and to provide incidental teaching. Only positive reinforcement is used to teach the children. Several preschool programs in the United States and the United Kingdom report comparable success to home-based behavioural programs. These programs have low child-to-staff ratios, collect detailed behavioural data, generally integrate the children with typically developing peers, and train parents intensively in behavioural methods.14 However, most young children with autism in Australia do not receive intensive behavioural intervention programs — partly because such programs are not recommended by many health professionals and partly because of their prohibitive cost for families. Only Western Australia has achieved partial government funding for preschool behavioural programs, as justified by a review by the Disability Services Commission of Western Australia.15 This State is also the first to have a prospective autism register, placing it in a unique position to provide Australian outcome data. We are unaware of comprehensive Australian outcome data (from specialised preschools and schools for autism) with which to compare outcomes of applied behavioural analysis programs. For those of us who are parents of children with autism, this seems to be a pressing need. In the United States, parents have effectively advocated for evidence-based interventions using expert statements.2 If intensive behavioural programs in young children with autism allow about half of the children to no longer require special education and other costly interventions, government funding of such programs would provide economic returns in the long term. The returns to the children who respond and their families would, of course, be priceless.

Jennifer J Couper MD, FRACP · Amanda J Sampson FRACOG, DDU, COGUS

To the Editor: An 8-month-old girl, born in Perth, Western Australia, who received only breast milk feeds for her first six months of life, was referred urgently to Princess Margaret Hospital for Children (the tertiary paediatric hospital in Western Australia) for investigations of seizures. Her parents described the seizures as episodic, involving all limbs, lasting less than five minutes and occurring over the 10 days before presentation. At presentation, the infant had carpopedal spasm. She was afebrile, and a septic screen gave negative results. Venous blood gas analysis showed a low ionised serum calcium level of 0.71 mmol/L (reference range [RR], 1.13–1.32 mmol/L). An x-ray film of her wrists showed signs of rickets, with cupping and fraying of the distal metaphyses of both radius and ulna. Other blood tests revealed elevated parathyroid hormone levels of 8.6 pmol/L (RR, 0.80–8.00 pmol/L), an elevated alkaline phosphatase level of 523 U/L (RR, 100–350 U/L), and an extremely low level of serum 25-hydroxyvitamin D3 (25OHD3) of 5 nmol/L (RR, 30–150 nmol/L). The infant responded to treatment with an infusion of 0.5 mg/kg of calcium gluconate and a 4-month oral course of 0.2 μg calcitriol daily. After treatment commenced, she did not have any further seizures. Radiogaphy performed 4 months later showed increasing calcium deposition at the distal metaphyses of the radius and ulna. Seizures are described as a presenting feature of hypocalcaemia in vitamin D deficiency rickets.1,2 Ultraviolet radiation and/or dietary vitamin D are required to prevent rickets in children. Perth, Western Australia, located at latitude 32° South has an average daily sunshine duration of at least five hours per day. However, even with this amount of sunshine, vitamin D deficiency can still occur in people who, for various reasons, receive little or no sun exposure, especially if their skin is darkly pigmented, and who have an inadequate dietary intake of vitamin D. The girl's mother, who wore a veil and clothing which protected her body from exposure for religious reasons, had a serum 25OHD3 level of 7 nmol/L. Breast milk is a poor source of dietary vitamin D, especially when the lactating woman is vitamin D deficient.3 In Australia, vitamin D deficiency rickets in infants of parents who have migrated from Mediterranean, African, Middle Eastern and southern Asian regions has been reported since the 1960s.4,5 Education of healthcare providers and their patients about the requirement for sunlight exposure or dietary supplementation to prevent vitamin D deficiency rickets needs to continue.

Graeme H Johnson · Francis Willis

Clinical question"Is sunlight an effective treatment for jaundice in term infants?" A women's health educator at Southern Health wanted to know if there was any evidence that sunlight helps to reduce physiological jaundice in healthy term infants. Search question Definition of physiological jaundice1 Physiological jaundice is a diagnosis of exclusion. It should not fill any of the following criteria: Clinical jaundice in the first 24 hours of life; Total serum bilirubin level > 300 μmol/L in a term infant or > 255 μmol/L in a preterm infant; Direct reacting serum bilirubin level > 30 μmol/L, persisting more than 10 days in a term infant or 14 days in a preterm infant. The formulated search question followed a standard patients/interventions/comparisons/outcomes (PICO) format. Patients were term newborn infants with physiological jaundice (see Box), and the intervention was exposure to sunlight. Clinical outcomes of interest were primarily a reduction of jaundice. A randomised controlled trial comparing sunlight exposure to no treatment or another treatment would be the most appropriate study design to answer this clinical question. Search The search terms "neonatal jaundice", "hyperbilirubin(a)emia" or "icterus" were combined with the treatment search terms "sunlight", "heliotherapy" or "phototherapy". We searched the following electronic databases: the Cochrane Library, Best Evidence, MEDLINE, CINAHL (Cumulative Index to Nursing and Allied Health Literature), Current Contents and Biological Abstracts. MEDLINE indexes articles published since 1966, but a widely cited and historically important article that provided the first English-language report of an association between light and a reduction in neonatal jaundice was published in 1958.2 In light of this, we hand-searched the print versions of Index Medicus and Science Citation Index from 1958 to 1966. We also searched the websites of a number of organisations: Bandolier, University of Michigan Department of Pediatrics (Evidence-Based Pediatrics), US National Guidelines Clearinghouse, National Health and Medical Research Council of Australia (Publications Catalogue), Scottish Intercollegiate Guidelines Network, and UK National Health Service (Institute of Health Sciences Guideline Project). Summary of findingsOur extensive search identified only the one, original study that examined sunlight exposure as a treatment for neonatal jaundice.2 This was a case series reporting the effect of sunlight in jaundiced preterm, rather than term, infants. The same authors then reported a case series of artificial light therapy for jaundiced preterm infants, which stimulated the subsequent considerable volume of research articles on the effectiveness of phototherapy for neonatal jaundice in both term and preterm infants. Current recommendations for artificial phototherapy are summarised elsewhere.3 We found no controlled trials comparing sunlight against either no treatment or artificial light treatment for jaundice. The use of sunlight appears to have resulted from anecdotal reports of its effectiveness4 rather than from rigorous medical evidence. And if the effectiveness of sunlight exposure for jaundice is unknown, so too is the incidence of potential risks to the neonate — for example, sunburn or photosensitivity. OutcomeThere is insufficient evidence to support exposure to sunlight for the treatment of jaundice. The persistence of this practice 40 years after publication of a report on a single case series raises questions about the influence of evidence on the beliefs of professional healthcare workers. Based on our search results, a recommendation against using sunlight exposure to treat jaundice was distributed to Southern Health staff and used in an education program for midwives.

Renea V Johnston PhD · Jeremy N Anderson MSc MD FRANZCP · Cheryl Prentice

The Longitudinal Study of Australian Children (LSAC) is a major research endeavour to assess emerging health and developmental concerns and their determinants in children. Previous longitudinal studies of children in Australia and New Zealand have contributed significantly, but have had limitations, which LSAC will attempt to address. A new generation of longitudinal studies is needed to enable transnational and historical comparisons. Members of the LSAC (Longitudinal Study of Australian Children) Research Consortium John Ainley, Australian Centre for Educational Research; Donna Berthelsen, Queensland University of Technology; Michael Bittman, University of New South Wales; Dorothy Broom, Australian National University; Linda Harrison, Charles Sturt University; Bryan Rogers, Australian National University; Michael Sawyer, University of Adelaide; Sven Silburn, Curtin University; Lyndall Strazdins, Australian National University; Judy Ungerer, Macquarie University; Graham Vimpani, Newcastle University; Melissa Wake, Murdoch Children's Research Institute; Stephen Zubrick, TVW Telethon Institute for Child Health Research. In March 2002, the Commonwealth Department of Family and Community Services announced the commencement of the Longitudinal Study of Australian Children (LSAC).1 This study is being implemented by a large multidisciplinary research consortium led by the Australian Institute of Family Studies. It will track the health and development of two national, population-representative cohorts of children recruited in their first and fourth years of life. The study will assess a broad range of individual, family and environmental determinants of health and wellbeing, and will focus on identifying factors that influence good and poor life-course outcomes. The study aims to provide data that will inform the development of health, family and social policy and services within Australia (Box 1). LSAC represents a significant government investment ($20.2 million over nine years) in longitudinal research on children, and it is timely to consider the extent to which it will address the shortcomings of past studies and add to our knowledge of children's health and development. Longitudinal studies are essential to understand the causes of health problems and identify possible solutions.2 Using this type of study, Australian and New Zealand researchers have contributed significantly to our knowledge of health and development3-7 (Box 2). However, there are several reasons why Australia needs a new longitudinal study of children. Past studies typically recruited cohorts during the 1970s and 1980s8 and are therefore limited. First, the health profile of Australian children has changed significantly in recent decades. Emerging health concerns include increasing rates of atopic and chronic diseases and mental health problems, and unacceptably high levels of suicide, preventable injuries and harmful health behaviours during childhood and adolescence.9 Second, the environments in which children are being raised have altered profoundly in the last 30 years.9,10 There have been changes to children's immediate environments (family, childcare, schools and neighbourhoods) and the broader sociopolitical climate (including widening social disparities).10 Past study findings may not be relevant to modern childhood environments. In addition, past cohorts were typically drawn from confined geographical locations,8 and data on health and social services and policies may not be more widely applicable. Third, there have been considerable advances over the last 30 years in theory, measurement tools and analytic techniques.11 Current epidemiological models highlight the central role of individual genetic and pathobiological factors in the expression of poor health and the need to examine multiple levels of influence (Box 3).12 Past studies have been unable to explore these factors adequately, often lacking sufficient sample sizes or the appropriate measures to disentangle multilevel influences. LSAC addresses a number of these limitations. Specifically, it will: measure a wide range of outcomes and determinants; recruit cohorts nationally from rural–regional and urban settings; and have a sufficient sample size to explore multiple determinants and the occurrence of relatively rare events.1 However, it is unrealistic to expect this study to address all research needs. For example, LSAC will not capture sufficient children from minority groups to enable exploration of life-course pathways that may be unique to these populations. In addition, cost and methodological constraints are likely to result in the exclusion or under-representation of children from remote areas, and some forms of data collection will be too time-intensive or costly to use (eg, certain observational, biological or environmental measures). Also, LSAC will not involve the systematic provision of interventions. Given the growing evidence of the benefits to health that arise from prevention and early intervention,13 a strong case can be made for a coordinated program of longitudinal intervention trials to assess the impact of interventions delivered around key life-course transition times.14 Thus, studies of specific populations, studies addressing research questions that require costly data collection, and studies that involve the assessment of interventions could all add value. These could be designed in parallel with LSAC, or as studies nested within LSAC for creating efficiencies in research costs. Australia is unique in geographical distribution of the population, family structures, ethnic diversity, social structures, policies and service provision. International research may have limited applicability in the Australian context. Nonetheless, it is important to consider Australian longitudinal research in an international context. Other Western nations are establishing new longitudinal studies,8 with European countries notable for coordinating studies that enable cross-country comparisons.15 LSAC may provide a foundation upon which other studies could be built to facilitate transnational comparisons and comparisons of changes to health pathways over time. Use of common design and measurement tools will facilitate these objectives. Longitudinal studies are expensive and demanding, not to be embarked upon lightly.2 Coordinated research efforts that strategically build upon the substantial national investment in LSAC may further enrich the evidence base for policy development and service provision to facilitate our nation's future health and wellbeing. 1: Questions to be addressed by the Longitudinal Study of Australian Children1 How well are Australian children doing on key developmental outcomes? What are the pathway markers, early indicators, or constellation of behaviours that are related to different child outcomes? How are child outcomes interlinked with children's wider circumstances and environment? In what ways do features of children's environment (such as families, communities and institutions) affect their outcomes? What helps maintain an effective pathway, or change one that is not promising? How is a child's potential maximised to achieve positive outcomes for children, their families and society? What role can government play in achieving these outcomes? 2: Achievements of past Australian and New Zealand longitudinal studies of children's health and development Study (starting date) Achievements Australian Temperament Study (1983)3 Clarified the contribution of temperament, family and environmental factors to later life adjustment. Christchurch Health and Development Study (1977)4 Contributed to child health, family and mental health policy, safety regulations for swimming pools and bicycle riding, and the development of early intervention programs for high risk mothers of infants. Dunedin Multidisciplinary Health and Development Study (1972)5 Findings across a range of physical and psychosocial health areas contributed to the development of health interventions for substance use, safe driving and cycling practices. Port Pirie Cohort Study (1979)6 Assessed the effects of environmental lead exposure and provided the impetus for changes in regulations relating to lead in petrol. Tasmanian Infant Health Study (1988)7 Assessed possible causes of sudden infant death syndrome, resulting in changed recommendations for infant sleeping positions, with documented reductions in infant deaths. 3: Ecological model of health across the life-course (modified from Lynch12)

on behalf of the LSAC Research Consortium

Three children presented with adrenal crises, manifested by vomiting and hypoglycaemia, after protracted courses of high-dose inhaled corticosteroids for asthma. Significant dose reduction was possible in all three without loss of asthma control, emphasising the importance of back-titration to minimise dose. Parents of children taking high doses of inhaled corticosteroids should be alerted to the clinical features of adrenal insufficiency. If suspected, prompt medical assessment should be arranged, including serum glucose and cortisol measurement. The effectiveness of prophylactic inhaled corticosteroids (ICS) in childhood asthma is well established1 and these drugs are recommended as a safe, first-line preventive therapy.2-4 Dose-dependent biochemical adrenal suppression with ICS has been well documented,3-5 although, until recently, reports of frank adrenal insufficiency in children have been rare.6-9 We present the first documented Australian report of three children who presented with adrenal crises while being treated with ICS for asthma. Each had a history of an intercurrent illness during which they were unable to mount a stress-response rise in cortisol level. Clinical recordsPatient 1Presentation: A seven-year-old boy presented with hypoglycaemia associated with vomiting, abdominal pain and drowsiness preceded by two days of fever, rhinorrhoea and fatigue. He had had a previous episode of hyponatraemia and vomiting, but his blood glucose level was not documented at the time. Although he had been noted to have a cushingoid appearance in the past, he was normal on physical examination, his height and weight were on the 3rd percentile, and his growth velocity was normal. He was found to be hypoglycaemic and hyponatraemic, with a low serum cortisol level (Box 1). A short Synacthen test confirmed adrenal insufficiency (Box 2). Asthma history: The patient had a history of "poorly controlled" asthma, but his wheeze was minimal and not associated with increased work of breathing. He undertook normal physical activity and was rarely absent from school. Spirometry findings in the past had been normal. Medications: He was taking fluticasone propionate (1500 μg daily), nebulised budesonide (1000 μg daily till three weeks before presentation), salmeterol (50 μg twice daily), nebulised salbutamol (5 mg four times daily) and ipratropium (250 μg four times daily) and montelukast (5 mg daily). From the age of two years his ICS doses had been progressively increased and had been at these levels for 10 months before this presentation. He had received frequent doses of oral prednisolone from the age of four years, but had had none for the past eight months. Treatment and clinical course: Immediate treatment included a glucose bolus, fluid replacement and hydrocortisone. Ongoing treatment involved giving regular hydrocortisone while reducing the dose of ICS, with no deterioration of asthma control. Four months after his presentation he was taking 500 μg fluticasone daily and being weaned off hydrocortisone. Patient 2Presentation: A four-year-old boy was referred for investigation of two episodes of hypoglycaemia associated with vomiting and lethargy. There was a third episode of vomiting and lethargy; his blood glucose level was normal on this occasion (his mother had treated him with glucose before presentation at hospital). The patient was normal on physical examination, with height and weight between the 10th and 25th percentiles and with normal growth velocity. He was not cushingoid in appearance and had no abnormal pigmentation. Results of available baseline investigations during the hypoglycaemic episodes are shown in Box 1. A short Synacthen test confirmed secondary adrenal insufficiency (Box 2). Asthma history: The patient had had a history of episodic cough and wheeze since the age of four months, but had good exercise tolerance and minimal nocturnal symptoms between episodes. Past spirometry findings were normal. He had started taking ICS at 18 months of age, with progressively increasing doses in an attempt to control acute episodes. Medications: His medications were 1–2 puffs of 250 μg fluticasone propionate with 25 μg salmeterol (Seretide 250/25; Allen & Hanburys) twice daily (giving a daily fluticasone dose of 500–1000 μg), and salbutamol and ipratropium as required. He had never previously required oral steroids. Treatment and clinical course: With each hypoglycaemic episode he was treated with intravenous fluids, with good clinical response, but on one occasion he also received a short course of prednisolone for a "mild exacerbation of asthma". With the normoglycaemic episode he was treated with intravenous fluids and hydrocortisone for two days. After adrenal insufficiency was confirmed (Box 2), therapy with replacement hydrocortisone was commenced and he was weaned from his ICS dose. He currently takes 100 μg fluticasone and 4 mg montelukast daily, with no significant symptoms. He takes hydrocortisone as needed in times of stress, such as during infections. The patient was also subsequently found to have normal spirometry results, even during acute episodes of asthma. Patient 3Presentation: A 10-year-old boy presented after a hypoglycaemic seizure preceded by 24 hours of vomiting. He was normal on physical examination. His height was on the 90th percentile, weight between the 25th and 50th percentiles, and his growth velocity was normal. He was not of cushingoid appearance and had no abnormal pigmentation. He was found to have hypoglycaemia (Box 1), and hyponatraemia was also detected, but was possibly dilutional, as it was collected from the same intravenous cannula through which the dextrose bolus was given. A short Synacthen test confirmed secondary adrenal insufficiency (Box 2). Asthma history: The patient had a history of frequent episodic wheeze and breathlessness during early childhood, and had been admitted to a rural intensive care unit for asthma exacerbation at the age of five years. Past spirometry findings had been normal. He had been taking his current ICS dose for the previous two years, despite having no acute episodes of asthma or interval symptoms. Medications: He was taking Seretide 500/50 twice daily (giving 1000 μg fluticasone propionate daily) and salbutamol as needed. Treatment and clinical course: Therapy with replacement hydrocortisone was begun while the ICS dose was gradually decreased to two puffs of Seretide 50/25 twice daily (giving 200 μg fluticasone daily); hydrocortisone therapy was continued for four months and is now taken as stress cover for intercurrent illness. DiscussionOur case series further highlights the potential for the systemic activity of ICS to manifest as an acute adrenal crisis. These children, as well as patients in previously reported cases,6-9 all had biochemical evidence of adrenal insufficiency in the absence of other causes (normal long-chain fatty acids, excluding adrenoleukodystrophy, and normal adrenal antibodies, excluding autoimmune adrenalitis [Box 2]). The vomiting associated with hypoglycaemia seen in the three children has been previously described,6-9 as have seizures7-9 seen in our Patient 3. The hyponatraemia found in two of our patients is an unexpected feature, but mild hyponatraemia with normokalaemia has been documented in secondary adrenal insufficiency and postulated to be the result of inappropriate vasopressin secretion10 or subnormal aldosterone secretion in response to severe sodium restriction.11 Hydrocortisone has some mineralocorticoid action, and its use as sole replacement therapy was sufficient to restore electrolyte balance in these instances. While growth suppression was noted in one case series,6 and has been reported in association with asymptomatic adrenal suppression,12,13 it was not a feature in our patients, or in other reports.7,8 This suggests that adrenal suppression may manifest differently, perhaps related to differing patient susceptibility, or dose or duration of ICS use. Our patients and most children in previous reports6-9,12,13 were taking high doses of fluticasone. This may reflect current prescribing habits. While fluticasone may be more likely to cause severe adrenal suppression owing to its higher potency compared with other ICSs,7-9,12,13 all ICS medications have been shown to produce dose-dependent adrenal suppression in children.3-5 The low doses used in some reported cases6 again suggest varying patient susceptibility. Screening for asymptomatic adrenal insufficiency in children receiving high doses of ICS is problematic, particularly as abnormal results do not accurately predict clinically meaningful adrenal-axis suppression.4 Clinical indicators of systemic effects, such as poor growth or cushingoid features, were not seen in our patients or in previously reported cases.6-9 Results of tests such as 24-hour urinary free cortisol excretion and random serum or salivary cortisol levels are often indeterminate.14 Early-morning levels of serum or salivary cortisol which are at the high end of the normal range reassure that there is no serious adrenal suppression, but lower levels can be indeterminate.15 "Gold standard" tests, such as insulin-induced hypoglycaemia or metyrapone suppression, carry significant risks and are difficult to justify in this situation. The standard dose (250 μg) short Synacthen test is generally reliable, but may give false normal results in some instances where central hypothalamic–pituitary–adrenal-axis suppression predominates. The low-dose (0.5 μg/1.73 m2) short synacthen test has been proposed as being less prone to such errors,14 but abnormal test results do not always have clinical significance. A more pragmatic approach would be to warn the parents of children taking high dose ICS of the potential for adrenal suppression so that they seek medical advice during an intercurrent illness associated with unexpected lethargy, vomiting, abdominal pains or seizures. Such "non-respiratory" presentations warrant urgent assessment and tests for baseline blood glucose level (for hypoglycaemia) and serum cortisol level (which may be inappropriately low). Prompt recognition and treatment with hydrocortisone and intravenous fluids containing glucose may be life saving in the event of an adrenal crisis. In less acute presentations, suspected adrenal suppression warrants referral for endocrine assessment and adrenal testing, although there is considerable debate as to the best method for doing this.16,17 If significant adrenal suppression is evident by either a low cortisol level at the time of hypoglycaemia or an extremely low response to cortisol stimulation, then maintenance hydrocortisone should be used in the short term to facilitate safe weaning of the child from ICS. However, it is important to remember that some degree of adrenal suppression and risk of adrenal crisis may persist in such children if any steroid therapy continues, or for up to 12 months after steroid therapy is ceased. Two other important messages arise out of these case reports. Firstly, it is important to ensure that ICS therapy is appropriate for the child. The United Kingdom national survey indicated that around 20% of patients presenting with adrenal crisis were later shown not to have asthma.9 Other areas where ICS have no proven benefit are children presenting with recurrent cough18 or episodic viral wheeze.19 Secondly, in children with asthma receiving ICS therapy, it is important to minimise the dose by "back-titration" or by adding long-acting β-agonists (or both), as highlighted in recent guidelines.2,3 All our patients were taking more than 500 μg per day of fluticasone, which is currently the upper limit of the recommended dose for children.2 Further, significant dose reduction was possible without loss of asthma control, suggesting that these children were being overtreated. This report also serves to reinforce the recent guideline recommendation for specialist referral for children requiring high doses of ICS.2,3 1: Baseline investigations Daily dose of fluticasone propionate Blood glucose level (normal, > 3.5 mmol/L) Sodium level (normal, 135–145 mmol/L) Urinary sodium concentration (normal, < 20 mmol/L) Patient 1 1500 μg 1.3 mmol/L 130 mmol/L* 88 mmol/L Patient 2 (two episodes) 500–1000 μg 2.2 mmol/L < 1.2 mmol/L Not available 135 mmol/L Not available Not available Patient 3 1000 μg < 1.0 mmol/L 126 mmol/L† Not available * Specimen collected before glucose bolus. † Specimen collected after glucose bolus. 2: Adrenal investigations Stimulated cortisol level (normal, > 600 nmol/L) Adrenocorticotropic hormone level (normal, 2–10 pmol/L) Adrenal antibodies (normal, negative) Very long chain fatty acids (normal, negative) Patient 1 108 nmol/L* Not available Negative Negative Patient 2 44 nmol/L† < 1 pmol/L Negative Negative Patient 3 129 nmol/L† < 1 pmol/L Negative Negative * At time of presentation with hypoglycaemia (blood glucose level, 1.3 mmol/L). † During short Synacthen test (60 minutes after an injection of 250 μg Synacthen).

Joseph S Macdessi MB BS, DCH · Peter P van Asperen MD, FRACP · Tabitha L Randell MB ChB, MRCP, MRCPCH · Kim C Donaghue MB BS, PhD, FRACP · Geoffrey R Ambler MD, FRACP · Craig M Mellis MD, MPH, FRACP

To the Editor: Halasz and Vance1 are correct to point out that there is a diversity of causes that can contribute to a child exhibiting symptoms of attention-deficit hyperactivity disorder (ADHD), as defined in the Diagnostic and statistical manual of mental disorders (DSM-IV).2 In their article, they describe a child who meets the DSM-IV criteria for diagnosis of ADHD and in addition has been affected by environmental factors including poor bonding (due to maternal depression), domestic violence and parental separation. The child also exhibits developmental disability, as exemplified by delayed language development. The message is that, by explaining his symptoms in terms of his early experiences and his developmental disability, a diagnosis of ADHD can be excluded. Children with ADHD frequently come from families with disharmonious parental relationships. This may be associated with ADHD in one of the parents, perhaps the violent father in the case described. As clinicians our aim is to ameliorate symptoms as promptly and effectively as possible, and I am frequently impressed by the dramatic improvement that stimulant medication can make to a child's functioning both at school and within the family, with follow-on improvements in mood and self-esteem. Behavioural interventions and family therapy are important adjuncts to medication, but families such as the one described can be difficult to work with and this can limit the effectiveness of such interventions. A carefully monitored one-month trial of stimulant medication, with behavioural rating scales completed by the class teacher, may be appropriate in cases such as the one described. On the other hand, to deny a child a trial of stimulant medication on the basis of adverse early experiences and developmental disability may be to keep from the child the treatment that would help most.

Alison Poulton

In reply: We believe the core symptoms of attention-deficit hyperactivity disorder (ADHD) in children reflect a behavioural "final common pathway" of developmental risk factors,1 which can include transgenerational associations of core symptoms, as Poulton notes. Current scientific evidence suggests both genetic and environmental contributions, such as verbal and visuospatial executive dysfunction2 and/or early patterns of attachment deficits.3 Increased levels of parental psychopathology, associated with (in the child) deficiencies in problem solving, affect regulation, emotional communication and secure attachment, may contribute to the child's symptoms. For this reason, we advocate that medical management be based on a thorough assessment, to ensure that appropriate psychological interventions (eg, parent and teacher management training) are offered alongside psychostimulant medication. In a recent speech at a scientific meeting of the Faculty of Child and Adolescent Psychiatry, Dr A Mawdsley, a distinguished child psychiatrist, expressed his belief that "prescribing medication in the absence of a careful emotional state assessment is inferior medical practice". He went even further to state that "prescribing medication in the absence of a behavioural modification program should be considered medical negligence".

George Halasz · Alasdair LA Vance

First, let's decide what we really mean by phimosis Circumcision remains a topic of significant debate in Australia, even though there has been a marked reduction in the rate of circumcision in this country, which has reflected that of England, where 95% of boys were circumcised in the 1930s, declining to 6.5% in the early 1980s.1 In this issue of the Journal (page 155), Spilsbury and colleagues report that many boys are circumcised for phimosis before the age of five years, despite phimosis being rare in boys of this age.2 They reviewed all circumcisions in Western Australian hospitals between 1981 and 1999, recording that the rate of medically indicated circumcisions increased during that period, and that, if the 1999 rate remains stable, it would be seven times the expected incidence of phimosis in the group of boys aged less than 15 years. These findings imply a high rate of unnecessary surgery, similar to the findings from studies conducted in England.1,3 Spilsbury and colleagues define phimosis as "narrowing of the preputial orifice leading to non-retractability of the prepuce". Such a definition would result in many boys under the age of five years being diagnosed with a condition for which surgery is considered to be justified. Their use of the term phimosis seems to mean pathological phimosis. To clarify, the prepuce is regarded as normal in boys if non-retractable because of preputial adhesions, or if the skin is physiologically non-retractable because of narrowing (ie, physiological phimosis). Figure 1 shows a normal foreskin that is non-retractable. The terms phimosis and non-retractable are not sufficiently clear in isolation, and need to be qualified. Rickwood and colleagues have recently given a succinct definition, stating that the ". . . normality, with an unscarred and pliant preputial orifice, is clearly distinguishable from pathological phimosis [shown in Figure 2], a condition unambiguously characterised by secondary cicatrisation of the orifice . . .".1 The addition of the word "pathological" or "physiological" is necessary to differentiate the different prognoses for phimosis, and, if the foreskin is not retractable because of adhesions to the glans, that information needs to have been included in the definition and documentation. Thus, rewriting the extract from the article by Spilsbury and colleagues, "many boys are circumcised for (pathological or physiological) phimosis before the age of five years, despite (pathological) phimosis being rare in this group". Why does the rate of circumcision for phimosis exceed the expected rate of phimosis?Clearly, the word "phimosis" in isolation does not have sufficient power to separate disease from a normal condition. Further, if parents feel there will not be support from the general practitioner, they may complain of symptoms in their child for the purpose of avoiding the debate about the appropriateness of circumcision for cosmetic reasons. Alternatively, the GP may support the parents' desire to have their boy circumcised, but expect resistance from the surgeon, and thus tend to present the child as having a pathological diagnosis. Such manipulation is not surprising when dealing with such an emotive topic. Nor is it necessarily improper given the differing cultural and medical views on the value of circumcision. A further explanation for the high circumcision rate for (pathological) phimosis might be a reluctance to record non-medical circumcision as such, using the appropriate International classification of diseases codes.4,5 What is the optimal treatment for phimosis?A wide body of evidence shows that most boys can be treated successfully with steroid cream, and that circumcision is required only infrequently.6-9 Unfortunately, almost all of these studies lack the distinction between pathological phimosis, as defined by Rickwood et al,1 and other foreskins that are non-retractable either as a result of preputial adhesions or because they are physiologically non-retractable. However, clinical experience suggests that most cases of pathological phimosis can be successfully treated with steroid cream, provided the steroid cream is applied to the partly retracted prepuce three times daily. After 4–6 weeks the prepuce should be retracted at the time of bathing and after voiding.10 It also appears that even balanitis xerotica obliterans can be successfully treated without circumcision,11 particularly if steroid treatment is supplemented with the minor operation of preputioplasty, in which the distal end of the prepuce is widened.12 Unfortunately, because the use of the term phimosis does not recognise the variations of the normal foreskin, the roles of observation, steroid cream and circumcision have not yet been compared in a study that has used a rigorous definition of pathological phimosis. There remains debate about the care of the normal prepuce in infant males. Parents are usually advised not to touch it, whereas the normal hygiene approach to body parts is one of not hurting, but keeping clean. The latter policy may help prevent skin irritation at the end of the prepuce, which may be part of the cause of pathological phimosis and balanitis (although this needs to be supported by appropriate studies). Evidence-based discussion about circumcision with parents will only be able to occur once we have undertaken prospective studies of the care of the prepuce and the use of steroids for treating phimosis. Parents will then be confident that their uncircumcised boy will not develop disease attributable to the nature of the foreskin. However, we should first focus on integrating a standard definition of phimosis into the study protocols. In the meantime, we should respect the view of parents who regard circumcision as good treatment for their child, given certain provisos. One is that they have been made aware of other options. The other is that they are making an appropriately informed decision about the management of their boy's prepuce because they are aware that "phimosis" does not equate to "pathology", and "pathology" does not always need surgery. Finally, Van Howe et al warn that physicians who perform "involuntary" circumcision are required to provide full disclosure. However, they also warn that, "with current legal precedent, this may not be enough" to protect the doctor from legal action,13 further emphasising the need to develop sound definitions on which to base our treatment of the prepuce. 1: A normal foreskin that is non-retractable, with pouting of the most distal portion when gentle retraction is attempted. 2: This foreskin shows the dome configuration of a boy with "true" phimosis. The fibrosis and pinhole meatus are also seen.

Paddy A Dewan PhD, MD, FRACS

Objective: To investigate the incidence rate of circumcision for phimosis and other medically indicated reasons in Western Australian boys from 1 January 1981 to 31 December 1999.Design and setting: A population-based incidence study using hospital discharge data of all circumcisions performed in all WA hospitals during the study period.Main outcome measures: Changes in the incidence rate of circumcision for medically indicated reasons.Results: The rate of medically indicated circumcisions increased in boys aged less than 15 years during the study period. Phimosis was the most common medical indication for circumcision in all age groups. The rate of circumcision associated with phimosis was eight times that associated with balanoposthitis and 21 times that of balanitis xerotica obliterans. Boys aged less than five years had the highest rate of circumcision to treat phimosis, at 4.6 per 1000 person-years, representing about 300 circumcisions per year. Boys aged less than five years living in country areas were 1.5 times more likely to be circumcised for phimosis than boys living in metropolitan Perth.Conclusion: The rate of circumcision to treat phimosis in boys aged less than 15 years is seven times the expected incidence rate for phimosis. Many boys are circumcised before reaching five years of age, despite phimosis being rare in this age group.

Katrina Spilsbury BSc, PhD · James B Semmens MSc, PhD · C D'Arcy J Holman MB BS, MPH, PhD · Z Stan Wisniewski MB BS, FRACS

Certain social expectations of medicine combine with characteristics of subspecialised technological paediatrics to facilitate the form of child abuse labelled "Munchausen by proxy syndrome". Examining this form of child abuse highlights possible shortcomings of medical practice. The primary medical tasks of diagnosing and curing illness and of preventing suffering are sometimes overridden by other motivations of which doctors may not be fully aware. More open discussion of what motivates health professionals in their work may improve medical practice and lead to a reduced incidence of Munchausen by proxy syndrome.

Jon N Jureidini PhD, FRANZCP · Allan T Shafer MA (Clin Psych), D Litt et Phil · Terence G Donald MBBS FRACP

Much has been written about the motivation of perpetrators in Munchausen by proxy syndrome (MBPS), but little, as far as I know, about the motivation of doctors who "collude" with parents in these behaviours. In general, I have no quibble with the analysis by Jureidini and colleagues.1 It is interesting, as they have done, to put forward a theory as to why doctors who are motivated to make "the diagnosis" might fail to recognise invented illness; however, the motivation of the "relentless investigator" is not the only one that should be examined. Indeed, doctors do not practise in a vacuum and are subject to pressures, societal among others, which may change with the clinical setting, affecting their practice. As a result, we may make mistakes in aspects of the clinical encounter — whether in our assessment of the process, the history, the examination or our interpretation of tests. Thus, when dealing with children, persistent parents may force the hand of even-thinking doctors because of current medicolegal and societal pressures. I do agree that children are more at risk from doctors who are enthusiastic about making a medical diagnosis and who ignore the social aspects of the consultation. However, concentrating on the motivation of the parent in MBPS cases has not proven useful in practice because of the complex psychopathology underlying such behaviour,2-4 and I doubt that focusing on the motivation of doctors will improve practice for similar reasons. While the relentless investigator is a particular problem in these cases, factors such as being busy, stressed or forgetful may also account for failure to identify the problem sooner. My concern is that, for an understanding of motivation to be useful in preventing the phenomenon of MBPS, it would be necessary to both reliably identify doctors' motivations and intervene to change these, and I do not believe that either of these steps is feasible. As a practising paediatrician, I suggest that a more useful approach to this problem of paediatric diagnosis would be to establish guidelines for the problem of the unresolved clinical problem or "difficult diagnosis" (see Box). The unresolved clinical problem: a practical approach4 Always consider MBPS in the differential diagnosis when the pieces do not seem to fit. Pay attention to the process of the consultation (how does the patient present?), especially where there is a history of maternal psychological problems. If worried about missed disease, draw up a list of hypotheses that should be used to direct further testing. Include the process of consultation in these; consult a colleague. Do not order invasive tests to "reassure" parents. Consider the cost and harm of tests versus the benefits. Do not overinterpret test results in the clinically well child. Get corroboration of previous illness from independent sources. Do not accept at face value a history of severe illness in a well child. Consult with a colleague before doing invasive tests where there is a major and unexplained incongruity between the story and the assessment. This is particularly important in gastroenterology and neurology. If all appropriate treatments inexplicably fail, be very wary of invasive testing. Consult with a colleague.

Kieran T Moran FRACP

In reply: The support for my study1 is pleasing, especially the letter from Martin. I agree with Hockey and Pitt that including Queensland data would have enhanced my study, as Queensland is the only other Australian State with a substantial injury database like South Australia's and Victoria's. However, when I analysed the Queensland data only around 1% of baby-walker injuries could be classified as "proximity", compared with 20%–25% in Adelaide and Melbourne. Surveillance collection can very easily miss critical details. The SA questionnaire asks "What was the victim doing at the time of the injury?", then "What went wrong?" and, finally, "How exactly was the injury caused?". As an example, "a child in a baby walker accesses the fireplace and burns her hand". If one or two of the above questions are left out, or the coder does not capture all the detail, this narrative easily becomes "child burns hand on fireplace" and the detail that the baby walker facilitated the child's access to the fireplace is lost. In 1995, after the Victorian Injury Surveillance System moved to a "minimum" dataset, their "proximity" component dropped to just 1%, the same as for Queensland. Analysts at the Victorian Injury Surveillance System recommended that these post-1995 data not be used for my study. In my opinion the Queensland system has the same systematic problem, and discussions with Hockey suggest that this is a possibility (Richard Hockey, Senior Data Analyst, Queensland Injury Surveillance Unit, personal communication). My assertion — that the data I used are largely representative of baby walker injury events in metropolitan Australia — is justified, as any variations in "proximity" rates (even if they do exist) caused by different architectural styles in Queensland would represent only a very small proportion of the events Australia-wide. The presence of additional steps and higher steps would only explain a higher proportion of these types of injuries, and possibly increased severity, but not the almost total absence of "proximity" injuries, as there is no association between them. The recent finding by the US Consumer Product Safety Commission2 that steps and stairs injuries still occur with new-style walkers confirms the need to discontinue the proposed mandatory regulations and instigate an immediate ban. The letter from Beard is disturbing in its attitude. Inadequate supervision is another name for victim blaming, and an unsafe environment is a perfect description of a baby walker. Moreover, to suggest that the ban would include any form of purpose-designed apparatus, including a commercial baby walker, to assist disabled children is nonsense.

Peter G Thompson