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Bristol Cup Posters: Summaries of Posters Bristol Cup Posters: Summaries of Posters P-1 P-2 Nurse-led diphencyprone treatment clinic: A case of SAPHO syndrome: did isotretinoin development of the role of the DPC treatment trigger it? treatment nurse L. Paul and J.A. Newton Bishop E. Walsh and S. MacDonald Hull St James’s University Hospital, Leeds, U.K. Mid Yorkshire Hospitals NHS Trust, Pontefract, U.K. SAPHO syndrome is characterized by any combination of A registered nurse trained in the use and application of diph- synovitis, acne, pustulosis, hyperostosis and osteitis (Hayem encyprone (DPC) runs a weekly DPC treatment clinic. She has G, Bouchaud-Chabot A, Benali K et al. SAPHO syndrome: a in-house training assessed by a multiple choice questionnaire long term follow up study of 120 cases. Semin Arthritis Rheum and quarterly formal reviews in the form of ‘observed prac- 1999; 29: 159–71). We describe a case that may have been tice’ for 12 months after completing training. The study triggered by isotretinoin therapy. A 14-year-old boy had acne describes her training, her management of a patient starting vulgaris affecting his face, chest and back that had deteriorated DPC treatment, giving details of pretreatment investigations, recently. Scarring was noted. He was systemically well, in par- consent to treatment, treatment documentation, practical ticular had no fevers or joint pains. His previous medical his- details relating to the handling, use and storage of DPC, and tory included learning difficulties and well-controlled epilepsy. advice given to patients. She also describes the setting up and He was commenced on isotretinoin 0Æ3 mg kg–1 daily for 2 ongoing development of a monthly Self Help Group and the weeks before increasing to 0Æ6 mg kg–1 daily. His GP added results of three audits evaluating different aspects of the ser- in trimethoprim 300 mg twice daily and his skin began to vice. Audit one: The patient’s perspective on the value of the improve. However after 4 weeks at the higher dose of isotre- Self Help Group. This audit confirmed that patients derive tinoin, he developed bony pain, an abnormal gait and consti- practical and psychological benefit from attending the Self pation. He was admitted by the general surgeons. An MRI Help Group. Audit two: Sensitization of children (16 years scan revealed spondylolisthesis at L5/S1, with abnormal sig- and under) to DPC. The aim was to identify the number of nals at C7, T10/T11 and both sacral ala, suggestive of an infil- children who required more than one application of DPC to trative process. Blood tests, bone marrow culture and bone become sensitized. Nineteen children were included; their biopsy showed no infective or neoplastic features. In retro- mean age was 11Æ6 years. Nine children had alopecia areata, spect, a diagnosis of aseptic osteitis was made suggesting the one alopecia totalis, three alopecia sub-totalis, four alopecia SAPHO syndrome. In this patient the isotretinoin was stopped sub-universalis and two alopecia universalis. Of these, four and his bony symptoms resolved spontaneously. His acne vul- required two applications and one required three applications garis has since been controlled using trimethoprim and low- to become sensitized. Audit three: Comparison of the treat- dose dapsone, with no recurrence of bony symptoms. The ment outcome for children requiring more than one applica- SAPHO syndrome is characterized by a combination of skin tion to become sensitized with those who were sensitized and bony pathology. The skin disease is either acne of the ful- after one application. The same cohort as for Audit two was minans or conglobata type, or hidradenitis suppurativa, or studied. Of the 10 requiring more than one application for pustulosis including palmoplantar psoriasis or pustular psoria- sensitization, three with alopecia areata and one with alopecia sis. The characteristic bone feature is aseptic osteitis, most totalis achieved complete regrowth; two with alopecia sub- commonly affecting the anterior chest wall, sacroiliac joints universalis achieved almost complete regrowth, two with alo- and vertebrae (Hayem G et al. 1999). A previous case has been pecia areata had no regrowth, one with alopecia sub-univer- reported in which isotretinoin appeared to trigger SAPHO syn- salis discontinued treatment and one with alopecia universalis drome (Chua SL, Ravenscroft J. Acne fulminans: part of the moved to another Trust. Of those sensitized after one applica- spectrum of SAPHO. Br J Dermatol 2007; 156: 1408 Abstract). tion, four with alopecia areata completely regrew their hair, Reintroduction of the isotretinoin reproduced the same symp- one each with alopecia areata, alopecia sub-totalis and alopecia toms in that patient. In our case, the timing of the initiation universalis and two with alopecia sub-universalis did not of isotretinoin and simultaneous development of symptoms, regrow. There was no statistical difference between the two as well as resolution of the condition on withdrawal of groups. the drug, would suggest that the bony aspect of the SAPHO syndrome was triggered by the isotretinoin. Ó 2008 The Authors 24 Journal Compilation Ó 2008 British Association of Dermatologists • British Journal of Dermatology (2008) 159 (Suppl. 1): 24–69 Bristol Cup Posters 25 P-3 P-4 Do we require age-specific reference ranges for The management of primary cicatricial alopecias androgen values when assessing females with M. Harries, R. Sinclair,* S. MacDonald Hull, cutaneous disorders associated with D. Whiting,à C.E.M. Griffiths and R. Paus§ hyperandrogenism? The Dermatology Centre, The University of Manchester, Manchester, U.K.; I. Ali, T. Jones,* N. Meston,* B. Shine* and *Department of Dermatology, University of Melbourne, Melbourne, Austra- F.T. Wojnarowska lia; Department of Dermatology, Pontefract General Infirmary, Pontefract, John Radcliff Hospital, Oxford, U.K. and *Department of Dermatology, U.K.; àBaylor Hair Research and Treatment Centre, Dallas, U.S.A. and Churchill Hospital, Oxford, U.K. §Department of Dermatology, University of Lubeck, Lubeck, Germany Androgen assays are important in the investigation of viriliza- The primary cicatricial alopecias (PCA; scarring alopecias; per- tion, hirsutism and alopecia in females. The latter two of these manent alopecias) are a diverse group of inflammatory hair conditions have a greater prevalence in the postmenopausal disorders of unknown aetiology that result in irreversible hair age group. Androgen levels can change with increasing age loss. They are characterized clinically by permanent loss of vis- and menopausal status (Longcope C. Hormone dynamics at ible follicular ostia and pathologically by replacement of follic- the menopause. Ann N Y Acad Sci 1990; 592: 21–30). The exact ular structures with fibrous tissue. In PCA the hair follicle is nature of these changes is a controversial topic; there have the main target of the disease process whereas in secondary been a limited number of studies with varying methodology cicatricial alopecias the follicular damage occurs indirectly and follow-up. Adrenal androgen levels peak in the mid 20s from events occurring outside the follicular unit (e.g. trauma, and thereafter decline in a linear manner which appears to be infection, etc.). The management of PCA is challenging. Poor independent of the menopause. Testosterone levels have been correlation between clinical and pathological features has shown to decline just prior to the menopause and then rise in resulted in problems with disease classification, with incom- the late menopause. Studies of sex hormone binding globulin plete or inaccurate disease definition and inconsistent use of (SHBG) levels in the ageing female have variable results terminology in the medical literature. Monitoring disease (Longcope C 1990). Reference intervals provided by most lab- activity is also notoriously difficult, particularly in conditions oratories for androgens do not include specific data for post- with minimal visible inflammation, e.g. pseudopelade of menopausal status. This could lead to misinterpretation of Brocq. To complicate matters, no fully satisfactory, evidence- androgen values. Age-specific reference ranges may provide based regimens are available for the treatment of defined scar- greater diagnostic accuracy. The objective of this study was to ring alopecias. In 2001 the North American Hair Research determine population specific reference intervals and evaluate Society produced a working classification of PCA based on the changes related to age for dihydroepiandrosterone (DHEAS), predominant inflammatory infiltrate found on scalp biopsy. androstenedione, testosterone, SHBG and free androgen index Differentiation of recognized clinical conditions, within each (FAI). Serum was collected from 182 postmenopausal females histological subgroup, could then be made, based on the clini- aged 45 or over using a standard technique and stored at cal features expressed. The main aims of treatment are to )70ºC until analysis. The results were divided into four age reduce symptoms and, most importantly, to slow or stop pro- groups, 45–54, 55–64, 65–74 and >74 years. A Kruskal–Wal- gression of the scarring process. We present an evidence-based lis test was performed to assess the differences between andro- summary of treatment options for PCA and propose a thera- gen and SHBG values between the different age groups. peutic hierarchy based on a broad appraisal of the reported Androstenedione (P < 0Æ05) and DHEAS significantly efficacy and safety of each agent. Studies were
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