Dear Editor:

Partsch and colleagues speculate that a rise in testicular temperature in infants and young children consequent to the wearing of modern plastic lined disposable nappies [diapers] may have contributed to both the fall in sperm counts and the increase in testicular cancer which have been reported in some countries.[1] In support of this hypothesis Partsche et al refer to reports of rising incidence rates in testicular cancer.[2-6] These studies report increased cancer risk in men born from before 1900 to 1988 diagnosed up to 1988, from 1916 to 1978, before 1970, from 1907 to 1981, and from 1865 to 1975, respectively. None reports incidence of testicular cancer in men who have had the opportunity to wear modern disposable nappies which have only been widely available since the late 1970s. There is some suggestion that the rise in testicular cancer may not be continuing.[7]

There are no data on testicular cancer incidence in men who are likely to have worn modern disposable nappies as the peak age for this disease is the late 20s and the men are simply too young. Similarly, the reports of a declining sperm count do not include any men born after 1975,[8-11] and even the most recent study from Denmark includes men born only before 1978.[12]

There is no way that modern disposable nappies could have contributed to the changes in male gonadal health reported in men observed to date since all studies are of men who were born before they were available.

Partsch et al[1] claim that the scrotal cooling mechanism was often completely abolished in boys wearing modern disposable nappies. This would appear to be over-interpretation of the data they had available, for two reasons. Firstly, they compared 24-hour mean scrotal temperature with spot rectal temperature and were unable to take into account the considerable circadian variation in core temperature in infants.[13] Secondly, they used measurement techniques validated in adults but not in nappy-wearing infants. They have not demonstrated that their methods, which included covering a considerable portion of the scrotal surface with adhesive tape, did not interfere with two aspects of scrotal cooling, ie, evaporation from the skin surface and contraction and relaxation of the scrotal sac. There is the very real possibility that the measurement process itself increased scrotal temperatures in all children.

There are several inconsistencies in the results table (table 1 in the paper by Partsch et al)[1]; for example, the mean temperatures and the calculated difference between temperatures measured with modern plastic lined disposable nappies and cotton nappies can not be reconciled, which the authors may wish to clarify.[1]

Further scientific research is clearly needed to establish the normal range of temperatures in infant testes and the extent to which they may be affected by different types of nappies and also the extent to which any changes may affect testicular health. However, it is clear that modern plastic lined disposable nappies can have played no role in the changes in male gonadal health reported worldwide to date.

References
1. Partsch C-J, Aukamp M and Sippell WG. Scrotal temperature is increased in disposable plastic lined nappies. Arch Dis Child 2000;83:364-8.

2. Adami H, Bergström R, Möhner M, et al. Testicular cancer in nine northern European countries. Int J Cancer 1994;59:33-8.

3. Forman D, Möller H. Testicular cancer. Cancer Surv 1994;19-20:323-41.

4. Hoff Wanderas E, Tretli S, Fossa SD. Trends in incidence of testicular cancer in Norway 1955-1992. Eur J Cancer 1995;31A:2044-8.

5. Weir HK, Marrett LD, Moravan V. Trends in the incidence of testicular germ cell cancer in Ontario by histologic subgroup, 1964-1996. CMAJ 1999;160:201-5.

6. Zheng T, Holford TR, Ma Z, Ward BA, Flannery J, Boyle P. Continuing increase in incidence of germ-cell testis cancer in young adults: experience from Connecticut, USA, 1035-1992. Int J Cancer 1996;65:723-9.

7. Andersen AG, Jensen TK, Carlsen E, et al. High frequency of sub-optimal semen quality in an unselected population of young men Hum Reprod 2000;15:366-72.

8. Auger J, Kunstmann JM, Czyglik F, Jouannet P. Decline in semen quality among fertile men in Paris during the past 20 years. N Engl J Med 1995;332:281-5.

9. Benshushan A, Shoshani O, Paltiel O, Schenker JG, Lewin A. Is there really a decrease in sperm parameters among healthy young men? A survey of sperm donations during 15 years. J Assist Reprod Genet 1997;14:347-53.

10. Carlsen E, Giwercman A, Keiding N, Skakkebaek NE. Evidence for decreasing quality of semen during past 50 years. BMJ 1992;305:609-13.

11. Irvine S, Cawood E, Richardson D, MacDonald E, Aitken J. Evidence of deteriorating semen quality in the United Kingdom: birth cohort study in 577 men in Scotland over 11 years. BMJ 1996;312:467-71.

12. Pharris-Ciurej ND. Cook LS. Weiss NS. Incidence of testicular cancer in the United States: has the epidemic begun to abate?. Am J Epidemiol 1999;150:45-6.

13. Wailoo MP. Petersen SA. Whittaker H. Goodenough P. Sleeping body temperatures in 3-4 month old infants. Arch Dis Child 1989;64:596-9.

Conflict of Interest
I have provided advice on scientific and epidemiological aspects of this study to 2 trade associations which act on behalf of modern plastic lined disposable nappy manufacturers EDANA and AHPMA.

Dear Editor

We read with interest this report by Grech et al.[1] It appears from their population-based study that as a result of the eradication of stray dogs, the annual incidence of visceral leishmaniasis (VL) declined considerably in Malta. VL is still endemic around the Mediterranean sea and sporadic cases are reported in children living in Northern Europe. It seems likely that with increasing tourism the incidence of VL will also increase in areas where until recently this condition would not even be thought of. During the last 18 months, we diagnosed three children with VL. As the presentation features can be fairly dramatic and physicians in Northern Europe are not always alert to the possibility of this condition, we would like to call attention again to the possibility of VL in non-endemic countries.

The main clinical features of the patients are shown in the table.

All three children presented with spiking high fevers, anorexia, hepato- splenomegaly and pancytopenia. The onset of the symptoms was insidious and it took 3-12 weeks to establish the diagnosis. In all three patients this was achieved through bone marrow aspiration and the demonstration of the typical amastigotes in macrophages. The diagnosis was further confirmed through the demonstration of antibodies to the leishmania parasite. All three patients needed erythrocyte transfusions and patient 3 also platelet transfusions. A 5-10 day course of liposomal amphotericin-B was given to all three children. The treatment was well tolerated, and they all became afebrile within a week. Pancytopenia subsided over the ensuing 2-3 weeks and the children gradually returned to normal activity in a few months.

Naturally, we cannot draw epidemiological conclusions from such a small number of patients. Nevertheless, it is rather intriguing to find three unrelated cases within a relatively short time. While the eradication of stray dogs may go a long way to reduce the incidence of VL, vaccination would be more desirable.[2] Although resistance and immunity against the leishmania parasites is not well understood, the seemingly increasing incidence of VL in children travelling from Northern Europe might be due to the fact, that they have no transplacental immunity against the parasite and are therefore more prone to develop VL than local children. There is much in common between the presentation features of the haemophagocytic syndromes and VL. It is noteworthy that all three of our patients showed signs of macrophage activation and haemophagocytosis was observed in their bone marrow smears. With increased awareness over this condition by physicians in non-endemic countries, the time required to reach the correct diagnosis and institute therapy should be reduced.

TOM RÉVÉSZ
TOM F W WOLFS
GABRIELLA KARDOS
MARCELLINE A VAN FURTH
University Medical Centre
Utrecht, The Netherlands
Free University Amsterdam
Amsterdam, The Netherlands

References

(1) Grech V, Mizzi J, Mangion M, Vella C. Visceral leishmaniasis in Malta - an 18 year paediatric, population based study. Arch Dis Child 2000;82:381-5.

(2) Herwaldt B. Leishmaniasis. Lancet 1999;354:1191-9.

Dear Editor:

We noted with interest the recent study reported by Murray et al[1] in which the characteristics of a cohort of 46 children with reflex sympathetic dystrophy (RSD) were reported. In a similar fashion in our paediatric rheumatology unit, chronic idiopathic musculoskeletal pain syndromes including RSD form a significant proportion of referrals. A retrospective review of 23 children referred to us in whom a diagnosis of RSD was made between April 1993 and November, revealed many similarities with the population described by Murray et al. This is illustrated in the table below:

Clinical characterisitics of 23 children with RSD compared with 46 described by Murray et al
 

In a similar manner many of our patients had seen multiple medical and health practitioners prior to referral and diagnosis. Five patients had pre-existing medical conditions (2 inactive juvenile idiopathic arthritis, 2 recurrent transient synovitis of the hip, one mild cerebral palsy,and one patient with asthma). A high level of pre-exisiting conditions with psychological components was seen in our cohort with 4 patients reporting a history of chronic recurrent headaches, 1 of chronic abdominal pain, 5 of sleep disorders, 2 of anxiety and 2 of depression. Three patients had a history of conversion disorders: and one recurrent hyperventilation. In addition 11 (48%) had one or more close relatives with a chronic or debilitating illness, and in seven of these it was of musculoskleletal origin. Five patients had a total of 7 first degree relatives with a diagnosed psychiatric condition.

Presenting symptoms were similar to the reported study with all patients reporting severe pain in the affected limb(s) in addition to restriction of joint range of motion (91%); sensory symptoms (55%); swelling (50%); colour change (55%), and abnormal sweating (14%). Seventeen patients (77%) had used crutches at some point during their illness and 7 (32%) had used a wheelchair.

Investigation prior to diagnosis was similar to the reported study. Eighteen patients (82%) had had plain radiographs of which 4 showed osteopenia. Eight patients (36%) underwent a bone scan of which 3 demonstrated reduced uptake in the affected limb. No other investigations, which included MRI, EMG, thermography and nerve conduction studies were felt to be contributory to diagnosis or management.

Six of 19 patients were admitted to another hospital prior to referral to our unit of which only 2 of these were reported to have received in-patient physiotherapy. Ten patients (53%) had outpatient physiotherapy in some form. In contrast 10 patients (53%), were treated with a period of limb immobilisation.

Examination findings on presentation to our unit included tenderness in 100%, hyperalgesia 76%, reduced temperature 71%; discolouration 67%; muscle wasting 48%; swelling 38%; abnormal sweating 24%, hair loss14%, and skin atrophy in 9%. Fifteen patients required admission to hospital, and all received physiotherapy, in addition to simple analgesics and non steroidal anti-inflammatory drugs as required. All patients had a pscyhological assessment by the paediatric team, received specific education and were taught self-management strategies. All were given a tailored home-exercise programme on discharge. 5 patients in whom adverse psychological factors were felt to be severe were referred for formal clinical psychology assessment and intervention. Intervention included patient and family counselling, behavioural and pain management interventions. Of the patients not admitted, information about RSD was given in the outpatient department, referral made to local physiotherapy services and specific medical follow-up arranged.

Average follow-up time from diagnosis was 26 months in our group (range 5 months to 7 years). Current status was determined from the time of last clinic visit or by telephone interview (if this was more than 6 months previously). Full outcome information is currently available for 19 patients, 11 of whom were completely well (58%), 5 were clinically improved but still symptomatic (26%), and 3 patients had not improved since diagnosis (15%). Of those who had recovered fully the median time to definite improvement was 4 weeks (range 2-10 weeks) and the mean time to full recovery 25 weeks (range 4 weeks to 15 months). As with the reported study by Murray et al, a high rate of reccurence or relapse in our patient group was seen with 11/19 (58%) having further episodes, of which 4 patients relapsed more than once, and in 2 patients the recurrence developed in different limb.

A full recovery from the original episode was achieved in 80% of patients. We found that although there was no reduction in the time reported to definite improvement in symptoms, patients admitted had a significantly reduced time interval between diagnosis and full recovery being 3.25 months, (range 1-7 months) compared with 6 months, (range 2-12) months in those not admitted. In our cohort a higher proportion of patients in whom the diagnosis was made within 4 months of onset of RSD made a full recovery (88% compared to 67%).

We agree with Murray et al[1] that RSD in children is an important cause of morbidity and suggest that earlier recognition and treatment is likely to result in improved outcome. It is clear that psychological issues are common and perhaps under recognised, and we believe it is mandatory that these be actively sought and addressed in all patients, as has been suggested by previous authors.[2,3] Although formal psychological intervention or psychiatric referral may only be required in some patients, we believe psychological assessment and teaching of coping skills and pain management strategies is essential in the management of all patients with chronic pain syndromes including RSD. If intensive outpatient physiotherapy support is not available, admission for a period of intensive physiotherapy should always be considered. We also strongly agree that there is a very limited role for sympathetic nerve blocks, or narcotic analgesia in the management of RSD in children, and that limb immobilisation in this condition is inappropriate and contraindicated.

Karen Davies
Specialist Registrar in Paediatric Rheumatology

R Khubchandani
Consultant Paediatrician

Kevin J Murray
Consultant Paediatric Rheumatologist

Institute of child Health
Great Ormond Street Hospital
London WC1N 3JH, UK
Kevin.Murray@gos-tr.nthames.nhs.uk

References

(1) Murray CS, Cohen A, Perkins T, Davidson JE, Sills JA. Morbidity in reflex sympathetic dystrophy. Arch Dis child 2000;82:231-3.

(2) Sherry DD, McGuire T, Mellins E, Salmonson K, Wallace CA, Nepom B. Psychomatic musculoskeletal pain in childhood: clinical and psychological analysis of 100 children. Pediatrics 1991;88:1093-9.

(3) Sherry DD, Wallace CA, Kelley C, Kidder M, Sapp L. Short- and long-term outcomes of children with complex regional pain syndrome type I treated with exercise therapy. Clin J Pain 1999;15:218-23.

Dear Editor:

I was interested by the report of Smyth et al regarding the finding of markers of intestinal inflammation in whole gut lavage in patients with cystic fibrosis.[1] As the alpha 1 antitrypsin levels were not increased compared to controls, perhaps another hypothesis needs to be considered.

Conceivably the inflammatory markers are not actually increased within the bowel, but rather, they are not degraded due to the lack of intestinal enzymes. Alpha 1 antitrypsin, which is resistent to proteolytic enzyme activity, would not be affected by such a phenomenon and therefore, would be the same in patients with cystic fibrosis and controls. Perhaps the authors would need to resort to the somewhat dated technique of radiolabeled albumin to definitively answer this question.

Larry Eisenberg

Reference

(1) Smyth RL, Croft NM, O'Hea U, Marshall TG, Ferguson A. Intestinal inflammation in cystic fibrosis. Arch Dis Child 2000;82:394-9.