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2006 Experimental Dermatology HSF Welcome/Symposium Abstracts 2006 Experimental Dermatology HSF Welcome/Symposium Abstracts Document Transcript

  • Experimental Dermatology 2006: 15: 405 Copyright # 2006 The Authors. Journal compilation # 2006 Blackwell Munksgaard Blackwell Munksgaard . Printed in Singapore EXPERIMENTAL DERMATOLOGY ISSN 0906-6705 HSF Welcome Letter New society affiliation It is with great pleasure we can announce that The Hidradenitis Suppurativa Foundation, Inc. (HSF) and Experimental Dermatology (EXD) have agreed on a partnership making EXD the official journal of the HSF. The Editors and the Publisher look forward to working closer with the HSF and wish them welcome aboard. Thomas A. Luger Editor Angela M. Christiano Editor Thomas Trier-Mork Journal Publishing Manager Blackwell Munksgaard Experimental Dermatology is the official journal of the Hidradenitis Suppurativa Foundation, Inc. Michelle Barlow, Robert Howes, Ralf Paus, Christos C. Zouboulis Co-founders of the Hidradenitis Suppurativa Foundation, Inc., San Diego, California, United States. E-mail: info@hs- foundation.org. The Hidradenitis Suppurativa Foundation, Inc. (HSF), a non-profit organization based in San Diego, California, warmly welcomes Experimental Dermatology (EXD) and Blackwell Munksgaard to an exciting alliance that provides the HSF a home in Europe’s leading basic skin research journal. As the official journal of the HSF, EXD joins the HSF and its international medical and scientific advisory board in our global mission to improve the quality of life and quality of care for individuals and families affected by Hidradenitis Suppurativa (HS), also called Acne Inversa. Forging productive and cooperative research efforts, an expanding international network of research- ers met in Dessau, Germany for ‘Directions 2006’, the First International HS Research Symposium, from 30 March 2006 to 2 April 2006. This event was organized by the HSF, the Dessau Medical Center and the EADV Task Force on ‘Acne and Rosacea’. The HSF thanks EXD for the publication of its symposium abstracts which will help provide essential, up-to-date public health information and detail new concepts and research findings on HS pathogenesis and management. 405
  • Experimental Dermatology 2006: 15: 478–482 Copyright # 2006 The Authors. Journal compilation # 2006 Blackwell Munksgaard Blackwell Munksgaard . Printed in Singapore EXPERIMENTAL DERMATOLOGY ISSN 0906-6705 Abstracts from ‘‘Directions 2006: The First International Hidradenitis Suppurativa Research Symposium’’ Dessau, Germany – March 30–April 2, 2006 Organized by the Hidradenitis Suppurativa Foundation Inc. (HSF), San Diego, CA, USA, www.hs-foundation.org Hidradenitis suppurativa/acne inversa: the clinical challenge HS is a chronic inflammatory disorder of the pilosebaceous unit, which occurs in intertriginous, ‘apocrine’ skin after puberty, and in Christos C. Zouboulis* more women than men. The clinical picture is dominated by late Departments of Dermatology and Immunology, Dessau Medical events resulting from follicular occlusion and its tissue-destructive, Center, Dessau, Germany; inflammatory sequelae, aggravated by bacterial superinfection. Laboratory of Biogerontology, Dermato-Pharmacology and Likely, HS originates from events in the hair follicle epithelium Dermato-Endocrinology, Institute of Clinical Pharmacology and outer root sheath (ORS) rather than the apocrine gland and/or its Toxicology, Charite´ Universitaetsmedizin Berlin, Campus duct. Genetic and endocrine factors influence HS incidence and Benjamin Franklin, Berlin, Germany course, at least in defined subpopulations. Hidradenitis suppurativa (HS)/acne inversa (AI) is a chronic, If follicular hyperkeratosis, leading to the retention of follicular recurrent, debilitating skin disease that presents with painful, material, represents the earliest histological abnormality, while inflamed lesions in the apocrine gland-bearing areas of the body, inflammation and apocrine involvement are secondary ones, then the most commonly the axillary, inguinal, and anogenital areas. The cause of hyperkeratosis and hyperproliferation of ORS keratinocytes disorder is associated with several clinical disputes, such as the must be identified. In normal skin epithelium, this typically results from discussion on the exact skin appendage involved, namely the sweat inflammatory stimuli. Because the skin epithelium is a crucial element glands, the apocrine glands or the terminal hair follicles, and the in the control of innate immunity, one may speculate that the primary discussion on the pathogenetic background, namely a genetic, defect in HS is an insufficiently controlled and/or abnormal release of hormonal, bacterial, genuine inflammatory, environmental, or pro-inflammatory ‘danger’ signals from the follicle ORS (e.g. physical one. The significance of gender, body mass, and smoking chemokines, defensins, TLR ligands). This may only lead to HS, if on disease prevalence is still under investigation. Moreover, the additional predisposing factors (e.g. genetically lowered threshold for major challenge is the choice of the optimal treatment: antibiotics, activation, or defective suppression of, innate immune response loops; retinoids, corticosteroids, incision and drainage, local wound care, constitutive overexpression of TLRs or underexpression of limited or radical local excision, radiation, laser therapy, and modern immunoinhibitory signals) are present and once pro-inflammatory drugs, such as biologics, have been proposed. The psychological stimuli are further up-regulated, e.g. by microtrauma, (neuro-) impact on the patient can be great, encompassing social, personal, endocrine and/or local microbial signals. In this scenario, and occupational challenges. The sequelae of the disease, including hyperkeratosis/hyperproliferation of ORS keratinocytes only dermal contraction, keloid formation, restricted limb mobility, represents the first visible sign of a primary imbalance of the lymphedema, and fistula formation dominate the long-term burden intrafollicular controls of epithelial innate immune mechanisms. associated with patient’s quality of life. This chiefly calls for systematic exploration of the hair follicle Significant research is required to elucidate the etiology of innate immune system in as yet uninvolved intertriginous skin of HS/AI, classify the clinical picture, identify appropriate drug HS patients versus normal controls as a key to unravelling the candidates, and evaluate the social, economic, and elusive pathogenesis of HS. psychological impact of the disease. Role of bulge cells in wound healing: possible implications for hidradenitis suppurativa Open questions in hidradenitis suppurativa research: a hair M. Ito, Y. Liu, Z. Yang, J. Nguyen, R. Morris, F. Liang and biologist’s perspective G. Cotsarelis* Ralf Paus* M8 Stellar-Chance Laboratories, University of Pennsylvania Department of Dermatology, University Hospital Schleswig- School of Medicine, Philadelphia, PA, USA Holstein, University of Luebeck, Luebeck, Germany The hair follicle bulge harbors a cluster of quiescent epithelial To clarify the obscure cause(s) of hidradenitis suppurativa (HS), stem cells, which generate the new lower hair follicle during plausible, comprehensive, and testable hypothetical pathogenesis follicle cycling. The role of bulge cells for maintenance of the scenarios urgently need to be developed. hair follicle and epidermis during normal homeostasis and after wounding remains controversial. To address these questions, we targeted the suicide gene, thymidine kinase (TK), or CrePR *Presenting author. recombinase to hair follicle bulge cells using a K15 promoter. 478
  • HSF Abstracts Administration of ganciclovir to K15-TK mice caused their 6q25.1-25.2 containing six genes. We have already screened a death due to gastrointestinal injury. We then treated immuno- large number of exons in both loci for mutations without deficient mice carrying K15-TK skin grafts with ganciclovir. identifying any obvious mutations. This resulted in injury of bulge cells within several days. Loss The eventual identification of genes causing HS will shed light of hair and permanent injury to hair follicles ensued. Dermal on the pathomechanisms underlying inherited forms of the scarring was also present. The epidermis without follicles disease and, ultimately, will set the scene for rational design of survived for several months. therapies aimed at treating the root causes of this disorder. To further assess the contribution of bulge cells to epidermis, we used double transgenic K15-crePR1, R26R mice. After 5 days of treatment with RU-486 (time 0), bulge cells and a small fraction of epidermal cells expressed LacZ under control of the Genetic analysis of three large Indian pedigrees with autosomal ROSA promoter. To determine whether bulge cells moved into dominant hidradenitis suppurativa the epidermis under normal homeostatic conditions, we counted Uppala Radhakrishna1,2*, Timir Y. Mehta3, Jitendra V. Solanki4 labeled epidermal basal cells at times 0, 30 days, and 180 days. and Swapan K. Nath5 We found that the percentage of labeled epidermal basal cells 1 Department of Genetics, Boystown National Research Hospital, (approximately 1%) remained constant, indicating an absence of Omaha, NE, USA; cell movement from the bulge to the epidermis. After punch 2 Green Cross Blood Bank & Genetics Research Centre, wounding, bulge progeny were detected in a radial pattern in Ahmedabad, Gujarat, India; the re-epithelialized area. Radial streaks of bulge cell progeny 3 Samarpan Medical & Research Organization, Modasa, Gujarat, emanated from the hair follicles at the wound edge. Bulge cell India; progeny proliferated and expressed normal epidermal 4 Department of Animal Genetics & Breeding, Veterinary College, differentiation markers such as keratin 10 and loricrin. Gujarat Agriculture University, Anand, Gujarat, India; Overall, these results indicate that follicular stem cells in the 5 Arthritis and Immunology Research Program, Oklahoma bulge are necessary for hair follicle survival and that these cells Medical Research Foundation, Oklahoma City, OK, USA functionally contribute to epidermal regeneration in response to wounding; however, the epidermis self-renews autonomously of Hidradenitis suppurativa (HS: OMIM 142690) is an aggravating the bulge under normal conditions. chronic inflammatory skin condition characterized by swollen, The role of bulge cells in hidradenitis suppurativa (HS) is not painful, inflamed lesions in the axillae, groin, and other parts of known; however, pathological studies suggest aberrant the body that contain apocrine glands. Incidence ranges from proliferation of the hair follicle may accompany HS and 0.1/100 to 4/100 in adults. It affects more females than males. inappropriate stem cell activation and differentiation could be The risk of developing non-melanoma skin cancer is very high a component of this disorder. The K15-crePR1 transgenic among patients with HS (Arch Dermatol 2001: 137: 730–734). mouse will serve as a powerful tool for evaluating the role of This chronic condition is familial, which supports its genetic hair follicle stem cells in mouse models of HS. Studying hair basis. However the causative gene(s) is not yet identified. follicle stem cells with recently defined markers for these cells We have studied three large Indian hidradenitis suppurativa will be useful for evaluating stem cell behavior in HS lesions. families with an apparent autosomal dominant mode of inheritance and 100% penetrance. No skipping of generations was observed. Pedigrees consist of 149 individuals, including 48 Mapping of two genetic loci for autosomal dominant hidradenitis affecteds (20 males/28 females). The age of onset is 25–35 years. suppurativa Histopathological studies were conducted in selected affecteds. W. H. Irwin McLean1*, Pam Wood1, Alan D. Irvine2 and Jan The expression of the phenotype was quite variable within these von der Werth3 families, and the majority was severely affected. All affecteds had 1 Human Genetics Unit, Division of Pathology and Neuroscience, typical characteristics of HS. Clinical findings included folliculitis, Ninewells Hospital and Medical School, University of Dundee, gastrointesinal (GI) polyps, sinuses axillae, cutaneous scars, retinal Dundee, UK; haemorrhage, epidermoid carcinoma, and polymorph function 2 Department of Paediatric Dermatology, Our Lady’s Hospital defects. Six of the affected females had hirsutism. Affected body for Sick Children, Crumlin, Dublin, Ireland; parts included tops of inner thighs, genitals, groin, buttocks, and 3 Department of Dermatology, Conquest Hospital, Hastings, UK under the breasts in women. Skin grafting was performed in some Hidradenitis suppurativa (HS) is a severe and debilitating skin of the affecteds. A few deaths due to squamous cell carcinoma condition that is relatively common in the population. In many were reported in these families. Cytogenetic analysis of two cases, there is a strong family history showing autosomal affecteds from each family did not show any abnormality. dominant inheritance with incomplete penetrance. We Genomewide linkage analysis is in progress to map the identified several large kindreds from the United Kingdom elusive locus and provide a target for positional cloning. and Ireland showing dominant transmission and carried out two genome-wide scans by genetic linkage analysis with 400 closely spaced microsatellite markers. A DNA array project for hidradenitis suppurativa/acne inversa This resulted in the identification of two separate genetic loci Sabine Fimmel1*, Evgenia Makrantonaki1,2 and Christos that show statistically significant linkage with HS. Marker C. Zouboulis1,2 D19S414 gave a significant log-of-the-odds (LOD) score of 1 Laboratory of Biogerontology, Dermato-Pharmacology and 3.66 in a single kindred and represents robust genetic linkage. Dermato-Endocrinology, Institute of Clinical Pharmacology and Recombination with markers D19S911 and D19S1170 limited Toxicology, Charite´ Universitaetsmedizin Berlin, Campus the interval to a 16.5-Mb region on chromosome 19, containing Benjamin Franklin, Berlin; at least 35 known or strongly predicted genes. Many of these 2 Departments of Dermatology and Immunology, Dessau Medical are transcription factors thought to be involved in modulation Center, Dessau, Germany of the immune system, which may be consistent with HS. Two additional kindreds did not map to this locus but instead Hidradenitis suppurativa (HS)/acne inversa (AI) is a chronic skin showed linkage to marker D6S290, giving a maximum disorder involving the apocrine glandular zones, where combined 2-point LOD score of 4.0 with no recombination. inflammation of sebaceous glands and terminal hair follicles can Visible recombinants narrowed this disease interval to a be identified. The exact aetiology of HS/AI is unclear. The 1.48-Mb region between D6S440 and D6S441 on chromosome reported incidence is 1:300 with a female-to-male predominance 479
  • HSF Abstracts of 4:1. There is an apparent strong influence of sex hormones on Conclusions: Based on CK expression in HS and PS, sinus HS/AI and association with acne vulgaris and hirsutism. The epithelium in HS and PS may possess characteristics of fragility, disease occurs at post puberty and disease activity declines at the hyperproliferation, and undifferentiation. climacteric, whereas improvement is seen during pregnancy. Most HS/AI patients have normal androgen profiles and apocrine glands, nevertheless, there have been reports of symptomatic improvement with the use of anti-androgen therapy. This DNA The potential role of acetylcholine receptors in acne inversa (HS) array project studies differences in gene expression of axillary pathogenesis apocrine secretory cells in vivo, between HS/AI patients and Hjalmar Kurzen*, Anisa Hana, Giovanni Frongia, Sergij Goerdt controls. Additionally, the gene expression pattern in axillary and Alexei Gratchev apocrine secretory cells of age-matched men and pre- vs. post- Department of Dermatology, University Medical Center menopausal women is evaluated to identify those genes, whose Mannheim, University of Heidelberg, Heidelberg, Germany expression is linked to circulating androgen levels, and highlights Acne inversa is a chronic inflammatory disorder that has been any gender/age differences in apocrine gland function. These shown to be influenced by tobacco smoking, which may experiments will further identify genes and biochemical represent a ‘natural’ model of exogenous nicotine-mediated pathways that could act as new targets for classical drugs and activation of the nicotinic acetylcholine receptors (AChR). In also targets for siRNA molecules in the treatment of HS/AI. previous works, we have provided a concise mapping of AChR present in normal skin and have demonstrated a crucial role of AChR in terminal differentiation and barrier formation. In Finding models for skin diseases: successes and failures addition, several studies suggest a role for the non-neuronal John P. Sundberg1,2* and Lloyd E. King Jr2 cholinergic system in immunomodulation. To date, the AChR 1 The Jackson Laboratory, Bar Harbor, ME, USA; composition of the cells involved in the pathogenesis of acne 2 Vanderbilt University, Nashville, TN, USA inversa has not yet been characterized. Using immunohistochemistry and RT – PCR, we could show Hidradenitis suppurativa (HS) is a disfiguring and debilitating, that nicotinic a3, a5, a7 and a9 AChR as well as muscarinic M1– relatively rare disease in humans. Reports of similar diseases in 5 M AChR are produced in lesional epidermis in a pattern domestic animals are also very rare. A clinically similar disease comparable with normal epidermis. In vitro and in lesional skin, was reported in dogs, particularly in Collies and Shelties, but we could demonstrate the presence of a3, a5 and a7 nAChR as this was later re-evaluated and determined to be a form of well as M1–M5 mAChR in variable amounts on macrophages. vesicular cutaneous lupus erythematosus. The alternative term In lesional epidermis, the choline-acetyltransferase (ChAT)- for HS, acne inversa, suggests forms of acne in domestic reactivity was particularly pronounced in the epidermal basal animals might provide an alternative approach to finding a layer, while in the sinus tracts, ChAT reactivity was extended to model. Mice, unlike dogs and cats, do not generally develop all epithelial layers. High levels of ChAT especially in the hair acne. However, the icthyosis mutation (Lbric) does develop follicle infundibulum indicate that endogenously produced ACh bacterial infections within the infundibulum that may result in may act synergistically with tobacco-delivered nicotine in furunculosis. If combined with various immunodeficiencies, aggravating infundibular hyperkeratosis. ChAT could not be through the creation of double mutant mice, then this might detected in macrophages in vitro or in vivo indicating that provide a useful model. However, finding spontaneous or macrophages do not actively contribute to ACh signalling in genetically engineered mouse models usually result in acne inversa but may rather be targets of ACh, and hence obtaining the best tools. The success obtained by developing nicotine induced immunomodulation. or finding simple single gene mutation or complex polygenic disease models for pseudoxanthoma elasticum and alopecia areata, respectively, through support from their respective foundations, provide a road map on how such models can be Clinical characteristics and outcome measures in hidradenitis found, defined, and provide unique tools to unravel the suppurativa research complex mechanisms of HS and, more importantly, provide Jean Revuz* preclinical tools for drug efficacy testing approaches. Department of Dermatology Hoˆpital Henri-Mondor, Paris XII University, Cre´teil, France Introduction: Outcome measures for medical treatment of HS Cytokeratin expression in hidradenitis suppurativa and pilonidal are needed. sinus Patients and methods: One hundred and sixty-four Ichiro Kurokawa1*, Arata Hakamada1, Ken-ichi Isoda1, Kei-ichi consecutive patients were prospectively evaluated. A new score Yamanaka1, Hitoshi Mizutani1 and Airo Tsubura2 1 was evaluated: HSPAIN ¼ pain intensity on a visual analog Department of Dermatology, Mie University Graduate School of scale (VAS 1-10) Â number of painful days/month. Medicine, Edobashi, Tsu, Mie, Japan; 2 Results: Mean age was 34 years (+ 11), sex ratio F/M 2.8:1. Department of Pathology, Kansai Medical University, Forty-two per cent were over weighted; 82% were smokers, Fumizono-cho, Moriguchi, Osaka, Japan 30% had a family history of HS. Age at onset was 23 years. Purpose: The aim of our study was to elucidate the Hurley’s class was I ¼ 72%; II ¼ 25 %; III ¼ 3%. Mean pathogenesis of hidradenitis suppurativa (HS) and pilonidal Sartorius’ score (1) was 22 (range: 3–72). Pain on VAS was sinus (PS) by evaluating their cytokeratin (CK) expression. 4.1 for lower localizations, and its duration was >15 days/ Methods: CK expression in HS (n ¼ 15) and PS (n ¼ 9) was month for 25% of patients. Patients with a Sartorius’ studied immunohistochemically using six anti-keratin antibodies. score > median were older (P ¼ 0.003), more frequently Results: In HS and PS, three types of epithelia were overweighted (0.02). The mean HSPAIN scored 7.9 (0–30). It identified: infundibular-like keratinized epithelium (type A), was highly correlated to Sartorius’ score (P < 10-3). This new non-infundibular keratinized epithelium (type B), and non- score has been used in therapeutic trials of dalacine – rifadin keratinized epithelium (type C). Type A contained CK1, 10, and combination and of zinc gluconate. Quality of life study of 61 14 similar to normal infundibulum, but it did not contain CK17. patients emphasizes the strong impact of HS compared with In types B and C, CK14, 16, and 17 were detected similar to that other dermatological diseases; patients with an early onset, in normal outer root sheath. In tumor nests of squamous cell long duration and continuous evolution are particularly carcinoma derived from HS, CK 14, 17, and 19 were detected. affected. 480
  • HSF Abstracts Conclusion: Surgery is not the only treatment for HS; we need Aim: To present and evaluate a series of 106 patients of outcome measures that are more adapted to medical treatment, hidradenitis suppurativa treated in the regional plastic surgery i.e. integrating pain which is the major burden of this disease. unit of southeast Scotland between January 1990 and December 2000. Reference: 1. Sartorius K, Lapins J, Emtestam L, Jemec GB. Patients: Median age at onset was 36 years (range 17– Suggestions for uniform outcome variables when reporting 70 years). The patients were predominantly females (88.78%) treatment effects in hidradenitis suppurativa. Br J Dermatol: and heavy smokers (98.1%). Median age before active surgical 2003: 149: 193–227. intervention was six years (range 1–30 years). The patients were divided into a mild group (n ¼ 64) and a severe group (n ¼ 42). All patients had broad-spectrum antibiotics and multiple Surgical treatment options for hidradenitis suppurativa and incision and drainage procedures before referral from general critical review of own experience practitioners, dermatologists or general surgeons. One hundred Wolfgang Christian Marsch* sites were managed by primary closure (mostly in the ‘mild’ Universitatsklinik und Poliklinik fur Dermatologie und group); 29 resurfaced with split skin grafts and 14 with local, Venerologie, Martin-Luther-Universitat Halle-Wittenberg, fasciocutaneous or musculocutaneous flaps (mostly in the Halle, Saale, Germany ‘severe’ group). Median postoperative follow-up period was 3 years (range 1–7 years). HS (acne inversa) is a chronic, progressive, initially inflammatory, Results: In the ‘primary closure’ series, recurrence rate ultimately a fistulating and scarring disease affecting apocrine requiring at least one secondary operation was 69.88%. There gland-bearing skin areas. Late phases afford a broad surgical was no recurrence, no serious complications and no revision removal of affected skin areas including subcutaneous fatty operations in the ‘graft’ and the ‘flap’ series. tissue, with secondary mesh grafting after a period of Conclusion: A high index of suspicion is required before granulation tissue formation. Fifty-three patients have been contemplating primary closure in hidradenitis suppurativa, treated surgically at our Dermatology Department. Long-term even in the ‘mild’ variety. This condition should always be results are excellent concerning satisfaction of the patients and treated aggressively by radical excision of all hair-bearing functional objectives. Local recurrences or development of new areas and reconstructed with a graft or a flap. lesions in formerly unaffected areas were noticed only in some patients who did not stop smoking. Patient details were as follows: gender distribution: male (M) Surgical treatment of acne inversa (hidradenitis suppurativa): a 20 (38%), female (F) 33 (62%), age: M 19–62 (average 40.7), F 20-year experience 15–56 (average 35.4), onset: M 16–57 (32.2), F 8–50 (25.5), Wolfgang Hartschuh* duration: 3 months to 37 years (8.0), F 6 months to 37 years Department of Dermatology, University of Heidelberg, (9.9). Sites mainly affected: axillary and perigenital. Specific Heidelberg, Germany regions for men: perineum and rima ani, for women: inguinal, submammary and abdominal. Multiple anatomical regions Acne inversa (AI) is caused by follicular hyperkeratosis in involved: men 40%, women 91%. Familiarity 0.4%. Associated intertriginous areas rich in apocrine glands followed by acne papulo-pustulosa or nodulo-cystica (¼conglobata): 19%. occlusion and rupture of the follicle and inflammation. Sinus Cigarette smokers: men 100%, women 67%. tracts, scarring and often contraction with limitation in mobility Excised material from each operation was carefully examined may occur. There is a world-wide consensus that in chronic histologically. The results endorse the concept of ‘acne inversa’ by disease surgical removal of all involved tissue as early as recognizing a perifollicular accumulation of lymphocytes possible is the most effective treatment modality. The aim of simultaneously at different infrainfundibula of terminal hair this study is to demonstrate our operative strategy, including follicles. However, a follicular hyperkeratosis seems secondary postoperative wound care and prevention, the results and to this, follicular perforation, and a combination of sinus, pitfalls in the treatment of AI. abscess and scar formation are most obviously tertiary events. The operations are increasingly performed in tumescence Therefore, HS seems to be an inflammatory, probably an anaesthesia, followed by secondary healing. Only removal of immunological disease with an initially strictly dermal target, extended skin areas in the inguino-genital and ano-perineal even followed by an intradermal horizontal propagation. Laser regions demand general anaesthesia. In axillary regions, all flux imaging could visualize the subclinical peripheral extension of hair-bearing skin including the sweat glands is removed to the basically dermal perifollicular inflammation. Biologics may obtain a hair-free, dry skin area. In the other regions with ill- have a beneficial effect on these early or perpetuating defined hair and apocrine gland areas, only involved indurated inflammatory events; however, thus far surgery remains the skin is excised. For early limited disease with fluctuant abscesses, first-line therapy in late phases of the disease. unroofing instead of mere incision and drainage is a good first option. Professional wound care with shaving and stretching of the wound margins is very important to avoid premature wound Surgical treatment options for hidradenitis suppurativa and closure. Locally applied disinfectant soaps and 2% triclosane critical review of own experience† ointments are effective in pre- and postoperative skin care. Anirban Mandal* and J. Watson Follow-up evaluation and collaboration among surgeons Department of Plastic and Reconstructive Surgery, St. John’s and dermatologists and an improved insight in the Hospital, Livingston, Scotland pathogenesis of AI are mandatory for the successful long- term management of patients afflicted with this complex and Background: Hidradenitis suppurativa has always been a debilitating disease. challenging area to the plastic and reconstructive surgeon. The aetiology is idiopathic, and medical treatment offers temporary relief only. Radical surgical excision, therefore, is the mainstay Biologics in the treatment of hidradenitis suppurativa: etanercept of active management. (preliminary report on clinical trial) and infliximab (case study) David Richard Adams* Penn State Hershey Medical Center, Hershey, PA, USA †Abstract edited from: Experience with different treatment A teenager with significant hidradenitis suppurativa (HS) and modules in hidradenitis suppurativa: a study of 106 cases. ulcerative colitis was treated with infliximab infusion. The Surgeon 2005: 3: 23–26. response was rapid and dramatic. This response led us to 481
  • HSF Abstracts develop and gain approval for a 20 patient, 6-month tried to use such cells to stimulate healing in a purulent investigator-initiated study using another anti-tumor necrosis environment. alpha agent, etanercept. The study is double-blinded and Methods: In the beginning, we designed a phase I clinical placebo controlled using etanercept 50 mg subcutaneous trial, involving five patients with Crohn’s disease. We injection twice weekly. For the first 3 months, one-half of the inoculated nine fistulas in four patients with autologous patients will receive placebo and other half active drug. Neither adipose-derived stem cells (ADSC) and were followed at least patient nor investigator will know whether active drug or 8 weeks. Seventy-five percent became healed, and 25% showed placebo is used. In the last 3 months, all patients will receive a decrease in output flow. No adverse effects were observed in active drug. The purpose of the study is to evaluate etanercept any patient. This study evidenced that such cells are safe. Then, effectiveness and adverse effects when used for HS. The study is we started a research line using SC in different suppurative still in progress, but preliminary results are encouraging. environments. During the course of these studies, we had the opportunity to treat a patient with perianal hidradenitis suppurativa using our current protocol of ADSC transplantation. Eight weeks after injection, patient had no Infliximab for hidradenitis suppurativa: report of seven perianal suppuration, and a year later remains well. consecutive patients Discussion: The biological mechanism that underlies the Alain Dupuy*, Laurence Fardet, Delphine Kerob, Annabelle therapeutic success of ADSC transplantation is unknown. Cell Levy, Herve´ Bachelez, Patrice Morel and Celeste Lebbe differentiation, secretion of growth factors or Department of Dermatology, Hopital Saint-Louis, Paris, France immunomodulatory effects have been suggested. No ethical Purpose: To evaluate all consecutive patients treated with conflicts were identified by our Ethics Committee, because the infliximab for hidradenitis suppurativa (HS). cells were autologous. Patients and methods: Within 1 year, all consecutive patients Conclusions: Our study shows that ADSC are safe for the seen in our department for HS (1) resistant to usual medical treatment of suppurative processes. The actual number of therapies (2) which could not be easily cured by surgery (3) not patients included and the uncontrolled nature of these pilot treated with new medication within 2 months before inclusion studies do not allow demonstration of the effectiveness of the were treated intravenously with infliximab (5 mg/kg) without treatment. However, the results encourage the performance of corticosteroids premedication. Four infusions were planned further studies. (week 0, week 2, week 6 and week 10) before the interruption of therapy and follow-up. Clinical activity of HS and quality of Acknowledgements life of the patients were assessed immediately before the first, the third and the fourth infusions of infliximab. The HSF would like to thank the Dessau Medical Center for Results: Seven patients were included. Five completed the co-hosting the event, and the Task Force ‘‘Acne and Rosacea’’ of four infusions. Two patients received only three infusions the European Academy of Dermatology and Venerology because of severe side effects. The Sartorius score moderately (EADV) for co-organizing the event. ‘‘Directions 2006’’ was also improved with infliximab (mean score at week 0: 94 + 39, at supported in part by unrestricted gifts from Creative Fusion, Inc., week 6: 71 + 38 and at week 10: 83 + 48). At week 6, patients DVV Stadtwerke Dessau, Abbott Immunology, Labcatel judged the efficacy of therapy as marked (n ¼ 1), moderate Dermatology, and by private donations. The HSF would like to (n ¼ 4) or null (n ¼ 2). At week 10, five patients were evaluated extend special thanks to Mayor Otto and the City of Dessau, the and judged this efficacy as marked (n ¼ 2), moderate (n ¼ 2) or Departments of Dermatology and Immunology at the Dessau null (n ¼ 1). The mean Skindex-29 score varied from 22 + 11 (E: Medical Center, the helpful staff at the Steigenberger Hotel, and 25 + 9, S: 13 + 5, F: 28 + 12) at week 0 to 18 + 10 (E: 22 + 8, the residents of Dessau for their hospitality and support. S: 12 + 8, F: 22 + 12) at week 10. Donations to the Hidradenitis Suppurativa Foundation, Inc. Conclusion: The efficacy of infliximab in severe HS is partial. (HSF), are strongly encouraged and warmly welcomed. Gifts to More experience is needed before finding a place for infliximab the HSF will help promote innovative basic and clinical research in the therapeutic armamentarium for HS. into the as yet obscure cause(s) and into the clinically unsatisfactory management of Hidradenitis Suppurativa, which affects about 1% of the population worldwide. The HSF is a The role of stem cells in suppurative environments non-profit organization registered in the state of California, Dolores Herreros*, Mariano Garcı´a-Arranz, Isabel Pascual and USA. For more information, please visit http://www. Damia´n Garcı´a-Olmo hs-foundation.org. Cathedra UAM-CELLERIX for Cell Therapy and Regenerative Medicine, La Paz University Hospital and Universidad San Diego, CA, April 2006 Auto´noma de Madrid, Madrid, Spain Hidradentitis Suppurativa Foundation, Inc. Purpose: The management of suppurative perianal lesions Michelle Barlow, President presents an extremely challenging problem. Stem cells (SC) Rob Howes, Vice President extracted from certain tissues, such as adipose tissue, Ralf Paus, Director of Basic Research can differentiate into various cell types. Therefore, we have Christos C. Zouboulis, Director of Clinical Research 482