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Title: Childhood Lichen Sclerosus: Incidence, aetiological factors and outcome.
Author: Powell, Dr Jennifer
Awarding Body: University of Oxford
Current Institution: University of Oxford
Date of Award: 2006
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Abstract:
Lichen sclerosus (LS) is a well-recognised inflammatory dermatosis but the aetiology remains uncertain. It has a predilection for the anogenital area and affects both sexes at all ages. In females it presents most commonly post menopausally but also affects a considerable number of prepubertal children. In this thesis I wished to document how commonly lichen sclerosus affects children, what the possible causes may be, to find the ideal treatment to minimise both short and longterm problems and lastly to clarify whether the longterm risks for this group ofchildren after puberty are the same as those for adults. Development of the specialist dermatology paediatric vulval clinic proved the ideal setting for studying the presentation, clinical features and course of the disease in girls and the paediatric urology clinic offers the same facility for boys. Both genetic and environmental factors may play a role in the aetiology of lichen sclerosus and assessment of familial incidence, immunogenetics and possible infective 'triggers' provide further areas of study. Lastly, treatment options have been assessed, the course of the disease recorded and risks for the future including development of malignancy is addressed. I The incidence of childhood lichen sclerosus is assessed using sources below 1. paediatric dermatology clinic presentation 2. paediatric urology clinic presentation 3. numbers diagnosed histologically per year 4. numbers from the GP database II Epidemiological data - 3 Supplied by The British Library - 'The world's knowledge' collected from patients by means ofquestionnaires and facts gathered during the course of diagnosis, treatment and follow up in clinic. This documents the increasing incidence oflichen sclerosus in children and its possible relation to anxieties about sexual abuse. III Genetic Influences Familial incidence oflichen sclerosus and its association with autoimmunity is confirmed. HLA tissue typing in the department ofTransplant Immunology at The Churchill, Oxford allowed examination ofimmunogenetic influences. The association with HLA class II DQ7 is documented in prepubertal girls with LS. IV Environmental Influences Several infective triggers have been suggested for the onset oflichen sclerosus, investigated and refuted. Considering the role ofhuman papilloma virus (HPV) as a trigger for lichen sclerosus we investigated cohorts ofmale and female children with LS for the presence ofHPV compared to controls. HPV DNA was detected and typed using a nested PCR technique with the help ofthe Microbiology and PHL at Addenbrookes Hospital Cambridge. HPV carriage is common in all children but girls with LS were found to carry more dysplastic-associated types ofHPV. The incidence ofabnormal smears and anogenital warts in mothers ofaffected children is also raised. Sexual abuse has also been suggested as a possible trigger for the onset oflichen sclerosus but the figures in this study do not support this. V Clinical Data Information is gathered from paediatric vulval and urology clinics as to mode and age ofpresentation ofLS. The natural history ofthe disease and how it is influenced by treatment is studied retrospectively and prospectively, and ideal treatment regimes formulated. Studying postpubertal children with LS suggests that the disease does not always clear at puberty so there is a theoretical risk of suffering the disease in adulthood with the concomitant risk ofdeveloping associated malignancy. It is wise to give patients and parents the information given to adults with the disease in case of problems later in life even if the children appear to be 'disease-free' after treatment.
Supervisor: Not available Sponsor: Not available
Qualification Name: Thesis (Ph.D.) Qualification Level: Doctoral
EThOS ID: uk.bl.ethos.491683  DOI: Not available
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