Dental enamel defects in coeliac disease

Letters to the Editor

Residence in the London

area

froml05(9-7)to 76(7-5) x 106 perml (p < 0’05),andfrom6l’7 (6-5) to 40-8 (4-9) x 106 per mL (p<0-05), respectively, whereas in those living outside the TWA (group B) sperm density and motile sperm density remained unchanged. Similarly, comparison of the changes in mean sperm density and in motile sperm density with time between the two groups (B and C) showed significant (p < 0-05) differences. These data relate solely to the partners of women treated for anovulatory infertility and so are not necessarily representative of the general population. Nevertheless, the evidence

and sperm

density SiR-The disturbing trend of decreasing sperm count and seminal volume over the past 50 years, and the concomitant increase in cryptorchidism and testicular cancer, 2,3 have been attributed to oestrogenic environmental pollution.4 We found adverse changes in sperm function in partners of 260 infertile women who received gonadotropin therapy between 1984 and 1989 compared with that recorded between 1978 and 1983. Mean sperm density fell from 101to 96 million per mL, the proportion of men with abnormal sperm motility increased from 20-7 to 34-4%, and those with (< 50%) abnormal sperm morphology (>50%) rose from 1 to 12-3%. There had been no changes in the pattern of referral to our clinic, in the mean age, clinical or socio-economic characteristics of the couples in the two cohorts, or in the laboratory techniques over this period. To investigate possible environmental influences, we have reviewed our data with reference to the water supply of the area where the couples lived. The Royal Free Hospital is situated within the Thames Water supply area (TWA)6 and, since more of our patients live within this area than in any other water authority area, we compared sperm data from partners of women who received gonadotropin therapy and lived within the TWA with data from partners of women living outside this area. We excluded those from outside the UK, those with temporary addresses, and those living on the border of the TWA. This left 183 couples (group A), of whom 104 were resident outside the TWA (subgroup B) whereas 79 resided within the area (subgroup C). Since the variables analysedsperm density, sperm motility, percentage of abnormal spermatozoa, and motile sperm density (density x motility per

1 sperm counts suggests that the phenomena we have observed are only part of a general adverse change in semen quality. We have no knowledge of the precise effect changes in semen quality may exert on male fertility. It is, however, of interest that polychlorinated biphenyl congeners were found in semen and that their concentrations correlated inversely with sperm motility.7 In addition, environmental factors affecting male fertility could also influence ovarian activity and hence female fertility. We emphasise that we do not assume that the only environmental factor peculiar to those living within the TWA was their water supply. However, despite exhaustive inquiries, we were unable to ascertain whether there had been any change in TWA testing of water supplies at source and delivery after 1983. Furthermore, some general environmental factor appears to be involved in that the increase in abnormal sperm morphology was not related to geographical area. Analysis of further years will show whether this adverse trend continues, but if aspects of altered semen function can be linked to a specific environmental influence, it should be susceptible to correction.

worldwide of

falling

J Ginsburg, S Okolo, G Prelevic, P Hardiman Division of Clinical Pharmacology, Royal Free Hospital School of Medicine, London NW3 2QG, UK

100)-were normally distributed, parametric tests (one-way analysis of variance andt test) were applied. As shown in the table, the mean (SE) percentage of abnormal forms increased in the second 6-year period (1984-1989) in all subjects irrespective of where they lived (group A, from 19 [1’1] ] to 31 [1-8]%, p<00005; group B, from 18 [1’3] to 32 [2’5]0, p < 00005; and group C, from 19 [ 17] to 30 [2-4]%, p < 0°005). In contrast, sperm density and motile sperm density fell significantly only in those living within the TWA (group C),

1

2 3 4

5

6 7

Carlsen E, Giwercman A, Keiding N, Skakkebaek NE. Evidence for decreasing quality of semen during past 50 years. BMJ 1992; 305: 609-13. Giwercman A, Skakkebaek NE. The human testis: an organ at risk? Int J Androl 1992; 15: 373-75. Boyle P, Kaye SN, Robertson AG. Changes in testicular cancer in Scotland. Eur J Cancer Clin Oncol 1987; 23: 827-30. Sharpe RM, Skakkebaek NE. Are oestrogens involved in falling sperm counts and disorders of the male reproductive tract? Lancet 1993; 341: 1392-95. Ginsburg J, Hardiman P. Ovulation induction with human menopausal gonadotrophins: a changing scene. Gynecol Endocrinol 1991; 5: 57-78. Map of water supply zones, Issue 2. Reading Bridge House, Reading: Thames Water Utilities Limited, Jan, 1993. Bush B, Bennett AH, Snow JT. Polychlorobiphenyl congeners, p,p’-DDE and sperm functions in humans. Arch Environ Contam Toxicol 1986; 15: 333-41.

Dental enamel defects in coeliac disease ,

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previous conort.

Comparison of mean (SE) semen quality between two 6-year cohorts In partners of Infertile women according to place of residence Table:

230

SiR-Coeliac disease is a condition in which there is an abnormal jejunal mucosa that improves morphologically when the patient is treated with a gluten-free diet. The incidence in the UK has been estimated to be 1 in 2000 but screening studies suggest that the incidence may be as high as 1 in 266 because many patients with mild or no symptoms remain undiagnosed.’ These patients should be identified and treated to correct nutritional deficiencies and reduce the increased risk of

malignancy which occurs in untreated patients.2 Specific dental enamel defects, defined as occurring symmetrically and chronologically in all four sections of dentition, have been reported in 96% of children and 83-100%of adults with coeliac disease compared with 4% of healthy controls.3-5 On this basis it has been suggested that dentists are important in screening patients with undiagnosed coeliac disease. Identification of these enamel defects would

warrant

referral for further

investigation. We have studied 45 consecutive patients attending the adult coeliac clinic and compared the results with 18 control patients with functional bowel disease. The teeth were examined by 1 member (CH) of the department of maxillofacial surgery who was unaware of the diagnosis. The teeth were cleaned and examined in an ordered manner with the aid of a mirror and an artificial light. Dental enamel defects were graded (grade 0-4= normal-severe structural defects)3. 3 patients with dentures of more than 4 teeth extracted were excluded from the analysis; the results are presented for the remaining 42 patients. The mean age at the time of the study was 53 years (range 19-80) and the mean age at diagnosis was 41 (range 1-79). 5 patients had developed severe gastrointestinal symptoms at an early age ( < 2 years). All other patients had presented after the age of 16. Only 4 patients (9-5%) had specific dental enamel defects and these were all patients who had symptoms at an early age. Enamel defects occurred significantly (p < 0 0005, Fisher’s exact test) more frequently in the early-onset group than in patients who developed symptoms after 16 years of age. 2 (111%) of the control group also had specific enamel defects; this was not significantly (p>0-05 Chi-square test) different from the coeliac group as a whole. We conclude that specific enamel defects are uncommon in adult coeliac patients and would not provide a sensitive screening test. However, enamel defects were present in 4 of 5 (80%) patients who had developed symptoms at an early age-the time of enamel formation. The finding of enamel defects only in those who present at a young age lends support to the theory that coeliac disease may develop at different times of life. This theory suggests that patients who present in adult life did not have gluten-sensitive enteropathy in childhood and, therefore, the dental enamel developed normally. Anne Ballinger, Ceri Hughes, Parveen Kumar, lain Hutchinson, Michael Clark Departments of Gastroenterology and Maxillofacial Surgery. St Bartholomew’s Hospital, London EC1A 7BE, UK

1 Hed J, Lieden G, Ottoson E, et al. IgA anti-gliadin antibodies and jejunal mucosal lesions in healthy blood donors. Lancet 1986; ii: 215. 2 Holmes GKT, Prior P, Lane MR, Pope D, Allan RN. Malignancy in coeliac disease: effect of a gluten free diet. Gut 1989; 30: 333-38. 3 Aine L. Dental enamel defects and dental maturity in children and adolescents with coeliac disease. Proc Finn Dent Soc 1986; 82 (suppl 3): 4 5

1-71. Aine L, Maki M, Collin P, Keyrilainen O. Dental enamel defects in coeliac disease. J Oral Pathol Med 1990; 19: 241-45. Maki M, Aine L, Lipsanen V, Koskimies S. Dental enamel defects in first-degree relatives of coeliac disease patients. Lancet 1991; 337: 763-64.

Kaposi’s sarcoma and volcanic soils SIR-Ziegler (Nov 27, p 1348) suggests that endemic Kaposi’s (KS) and podoconiosis (non-filarial elephantiasis), which have common regions of high prevalence in tropical Africa, also have a shared aetiological factor-namely, exposure to volcanic soils in highland areas of seasonal rainfall. sarcoma

A similar correlation, however, is not found elsewhere. Podoconiosis also occurs in Ecuador and India,l where endemic KS has not been reported, suggesting either the absence in these countries of another environmental factor

(possibly an infectious agent), which is present in central Africa, or that genetic predisposition is an essential component in the aetiology of endemic KS. Before the AIDS epidemic,

regions of tropical Africa affected by endemic KS were those where KS accounted for the highest proportion of childhood cancer.2 Outside Africa, non-AIDS-related childhood KS is most frequent in Papua New Guinea,3 another tropical region of widespread volcanic activity and heavy seasonal rainfall; published registration data for cancer among adults in that country do not show the frequency of KS.4,5The search for aetiological factors for endemic KS may also be assisted by information on the presence or absence of adult KS, podoconiosis, and concentrations of volcanic clay soil in Papua New Guinea. C A Stiller Childhood Cancer Research Oxford OX2 6HJ, UK

1 2 3

4

5

Group, Department of Paediatrics, University of Oxford,

Manson-Bahr PEC, Bell DR. Manson’s tropical diseases, 19th ed. London: Baillière Tindall, 1987: 816. Stiller CA, Parkin DM. International variations in the incidence of childhood soft-tissue sarcomas. Paediatr Perinatal Epidemiol (in press) Jamrozik K, White R, Misch K. Papua New Guinea: Cancer Registry of Papua New Guinea, 1979-1983. In: Parkin DM, Stiller CA, Draper GJ, Bieber CA, Terracini B, Young JL (eds). International incidence of childhood cancer. IARC Scientific Publications No 87, Lyon: IARC, 1988: 335-37. Misch K, Atkinson L, Reay-Young P. Papua New Guinea: Cancer Registry of Papua New Guinea, 1971-1978. In: Parkin DM (ed). Cancer occurrence in developing countries. IARC Scientific Publications No 75. Lyon: IARC, 1986: 331-34. Martin WMC, Sengupta SK, Murthy DP, Barua DL. The spectrum of cancer in Papua New Guinea: an analysis based on the Cancer Registry 1979-1988. Cancer 1992; 70: 2942-50.

SiR-Ziegler states that, before the AIDS epidemic, the putative sexually transmitted KS agent probably was geographically restricted and has spread world wide concomitantly with HIV. Does this mean that the unidentified infectious KS agent originated recently in equatorial Africa where the endemic form of KS is highly prevalent? However, the epidemiology of classic sporadic KS, which, like endemic KS, affects mainly the skin of the lower limbs, does not favour this hypothesis. Since the first description of classic European KS by Moritz Kaposi in 1872, sporadic KS cases have been recorded all over the world. Before the emergence of AID S, KS in polar Eskimos,l for example, and Japanese. If one assumes a common pathogenetic process for endemic and classic KS, it would be difficult to ascribe the world-wide occurrence of classic sporadic KS, especially in non-barefoot populations, to damage of dermal lymphatics of the feet and legs by absorbed volcanic clay soils with subsequent impaired local immunity to the KS agent. was seen

Karl-Horst Marquart Institute of Molecular Virology, GSF-Forschungszentrum, D-85758 Oberschleißheim, Germany

1 2

Neuherberg,

Mikkelsen F, Nielsen NH, Hansen JPH. Kaposi’s sarcoma in polar Eskimos. Acta Dermatol Venereol 1977; 57: 539-41. Tange T. Kaposi’s sarcoma. Case report and review of Japanese cases. Acta Pathol Jap 1979; 29: 319-32.

SIR—Ziegler presents data showing the geographical proximity of endemic KS, podoconiosis, and chronic exposure to ultrafine volcanic clay minerals. He suggests that clay soil indirectly induces local immune suppression, which increases susceptibility to the development of KS. However, a possible geographical association of malaria with endemic KS might also provide information for the understanding of the pathogenesis of KS. We looked, therefore, for an association 231