Acta Dermato-Venereologica 99-12CompleteContent

1170 SHORT COMMUNICATION Thalidomide in Severe Hidradenitis Suppurativa: A Therapeutic Option Claire HOTZ 1 , Emilie SBIDIAN 1–3 , Saskia INGEN-HOUSZ-ORO 1,3 , Olivier CHOSIDOW 1–3# and Pierre WOLKENSTEIN 1,3# 1 Department of Dermatology, Assistance Publique-Hôpitaux de Paris, Henri-Mondor Hospital, Créteil, 2 Institut National de la Santé et de la Recherche Médicale, Centre d’Investigation Clinique 1430, and 3 EA EpiDermE Université Paris-Est Créteil-Val de Marne, Créteil, France # These authors contributed equally to the study. Accepted Jul 17, 2019; E-published Jul 17, 2019 Hidradenitis suppurativa (HS) or acne inversa (AI) is a debilitating chronic inflammatory skin disease (1). Treat- ment of HS is challenging because of its persistence and poor response to treatment. Although only surgery leads to complete remission, antibiotics or biologics may be helpful. The potential efficacy of anti-tumor necrosis factor (anti-TNF) α agents to treat HS was demonstrated in a pa- tient who received 5 mg/kg/day intravenous infliximab for Crohn’s disease (CD): HS lesions substantially improved after two doses (2). The use of anti-TNFα agents (inflixi- mab and adalimumab) to treat HS has since been evaluated (3, 4). European guidelines were recently published (5). CASE REPORTS We report retrospectively the cases of 6 HS patients who received thalidomide after failure to respond to multiple broad-spectrum antibiotics and anti-TNFα agents. Three of the 6 patients showed good response to the treatment. All patients (4 men, 2 women; age range 17–38 years) presented severe Hurley stage III HS and had a very severe hidradenitis sup- purativa-physicians global assessment (HS-PGA) score (Table I). All had chronic, disabling disease (duration from 6 to 20 years) with a major impact on quality of life and all requested analgesic treat- ment. For the two women, the body mass index was > 30 kg.m –2 , and two of the men were smokers. The phenotype, as described by Canoui-Poitrine et al. (6), was axillary-mammary in two patients, follicular in one patient and gluteal in 3 patients. No patient had inflammatory bowel disease or inflammatory arthritis, acne or diabetes mellitus. Most patients received multiple antibiotics, such as rifampicin- clindamycin, doxycycline, or amoxicillin-clavulanic acid, and anti-TNFα therapy. All treatments failed to control the disease. Only patient 1 had limited surgery. Only patient 6, with a gluteal phenotype, and associated verrucous genital lesions, did not re- ceive anti-TNFα therapy. Squamous cell carcinomas on HS lesions were previously reported; most cases were in men with a gluteal phenotype, and the role of human papillomavirus was suggested (7). Previously, we had a male patient with the gluteal phenotype and associated verrucous genital lesions who had received anti- TNFα agents; cutaneous squamous cell carcinoma developed secondary to the treatment. Because of this unfortunate event and after a collegial discussion, we decided not to reintroduce anti-TNFα agents in this patient. For all 6 patients, we introduced thalidomide 50 mg, taken daily. We increased the daily dose to 100 mg after 2 months of treatment because of no meaningful improvement. The use of thalidomide in severe cases HS cases was established in our department based on a collegial decision. An informative leaflet regarding thalidomide is given to the patients, and informed consent and signature was required. After 4 months of daily treatment with 100 mg thalido- mide, 3 patients showed improvement, with less inflammation and purulent discharge (Fig. 1). The visual analog scale score decreased and the patients no longer used analgesics. The improved condi- tion led us to consider axillary and perineal surgery in patient 1 Table I. Characteristics of hidradenitis suppurativa patients receiving thalidomide Sex/ Pat. age, No years Duration BMI of disease, Skin involvement/ –2 kg.m Tobacco years phenotype 1 M/30 19 Yes 2 F/20 31 No 3 M/38 ND Yes 4 F/17 32 No 6 5 M/17 27 No 5 6 M/24 19 No 6 10 7 20 Hurley stage/HS- PGA score/VAS CRP level (mg/l) Armpits, groin, perineal areas, back and face/follicular phenotype III/very severe/ 9/10 55 Armpits, breast, groin/axillary- mammary phenotype Groin, perineal areas/ gluteal phenotype III/very severe/ 5/10 46 III/very severe/ 8/10 170 Armpits, breast, groin, perineum areas/ axillary-mammary phenotype Perineum areas/ gluteal phenotype III/very severe/ 9/10 ND III/very severe/ 6/10 18 Perineum areas/ gluteal phenotype III/very severe/na ND Past treatment -Broad-spectrum antibiotics (rifampicin-clindamycin doxycycline, sulfamethoxazole-trimethoprime, amoxicillin-clavulanic acid, azithromycin, rifampicin-clindamycin-metronidazole, ceftriaxon, rifampicin-metronidazole-moxifloxacin, ertapenem) -Zinc gluconate, steroids, retinoids, dapsone, pentasa -Axillary and perineal surgeries -Anti-TNFα therapy (infliximab 6 months, etanercept 3 months, adalimumab 5 months) -Broad-spectrum antibiotics (rifampicin-clindamycin, doxycycline, amoxicillin-clavulanic acid, azithromycin) -Anti-TBFα therapy (infliximab 3 months, adalimumab 7 months) -Broad-spectrum antibiotics (rifampicin-clindamycin, doxycyckin, amoxicillin-clavulanic-acid, pristinamycin, oxacillin) -Retinoids -Anti-TNFα therapy (infliximab 3 months), methotrexate -Broad-spectrum antibiotics (rifampicin-clindamycin, doxycycline, amoxicillin-clavulanic acid, azithromycin, rifampicin- metronidazole-moxifloxacin) -Anti-TNFα therapy (infliximab 4 months, adalimumab 4 months) -Broad-spectrum antibiotics (rifampicin-clindamycin, doxycycline, amoxicillin-clavulanic acid, azithromycin, rifampicin- metronidazole-moxifloxacin, ceftriaxon, sulfamethoxazole- trimethoprime) -Anti-TNFa therapy (infliximab 2 months) -Broad-spectrum antibiotics (rifampicin-clindamycin, doxycycline, amoxicillin-clavulanic acid, azithromycin) BMI: body mass index; HS-PGA scale: Hidradenitis Suppurativa-Physicians Global Assessment; VAS: visual analog scale; CRP: C-reactive protein (before start of thalidomide), ND: not done; NA: not applicable; TNF: tumor necrosis factor. doi: 10.2340/00015555-3268 Acta Derm Venereol 2019; 99: 1170–1171 This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta Journal Compilation © 2019 Acta Dermato-Venereologica.

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