Journal Identification = EJD Article Identification = 3329 Date: September 14, 2018 Time: 2:22 pm AB 1. Elkady A, Bonomo L, Amir Y, Vekaria AS, Guttman-Yassky E. Effec- tive use of ustekinumab in a patient with concomitant psoriasis, vitiligo, and alopecia areata. JAAD Case Rep 2017; 3: 477-9. 2. Muranski P, Boni A, Antony PA, et al. Tumor-specific Th17-polarized cells eradicate large established melanoma. Blood 2008; 112: 362-73. doi:10.1684/ejd.2018.3327 Granulomatous pigmented purpuric der- matosis containing Propionibacterium acnes Figure 1. Clinical manifestation before (A) and after (B) secukinumab administration. Granulomatous pigmented purpuric dermatosis (GPPD) is a rare variant of pigmented purpuric dermatoses [1]. Herein, we report a case of GPPD with localization of Propioni- bacterium acnes (P. acnes) in the granuloma, suggesting a Because the patient recognized hair growth after improve- sarcoidosis-like aetiology of GPPD. ment of psoriatic skin inflammation, not only on the trunk A 60-year-old woman consulted us for a rash on her extrem- and extremities but also on the scalp after secukinumab ities. Purpura had appeared on her legs two years before injection, our case suggests that recovery of hair growth her initial visit to a previous hospital. The lesions were could be expected in patients with psoriatic alopecia after refractory to topical steroids, and she was referred to our anti-IL-17A antibody treatment. Therefore, androgenetic hospital. Physical examination revealed purpuric plaques alopecia-like hair loss might be associated with psoriasis, on her extremities (figure 1A). Biopsy specimens from the manifesting as psoriatic alopecia in this case. Another pos- purpuric lesions on her leg and arm showed epithelioid sibility is that IL-17A could contribute to the pathogenesis granulomas with extravasation of red blood cells in the of alopecia itself. In a previously reported case, alopecia superficial dermis (figure 1C). Red blood cell extravasation complicated with psoriasis was documented to improve was particularly apparent around granulomas, involving after administration of the anti-IL-23p40 antibody, ustek- capillaries (figure 1D). The granulomas were not associ- inumab [1]. Therefore, inhibition of IL-17A might exert ated with hair follicles histopathologically and necrobiosis a further beneficial impact on alopecia. In addition, the was not found in the lesions. Histochemical staining (Fite’s role of IL-17 blockade on hair pigmentation has yet to acid-fast stain, Grocott stain, and PAS) was negative. Chest, be assessed. Although our patient’s hair was streaked with head, and neck X-ray and abdominal CT images did not dis- grey before secukinumab administration, he recognized an close obvious abnormalities. The acetylcholine esterase test increased proportion of black hair on the top of his scalp and beta-D glucan assay using the patient’s serum and the after treatment. Therefore, this case suggests a possible interferon-gamma release assay using the patient’s blood role of IL-17A in hair depigmentation. In a representative were negative. We suspected cutaneous sarcoidosis, and depigmentation disease, vitiligo, skin-homing Th17 cells the lesions were treated with photodynamic therapy, which were observed and adoptive transfer of melanocyte-reactive achieved resolution of the lesions. Th17 cells induced rapid depigmentation [2]. From these Five years later, the lesions recurred on the patient’s findings, we speculate that IL-17A might trigger depig- extremities (figure 1B). Skin biopsy specimens from the mentation of pigment cells in hair follicles, and IL-17A lesions from both the upper and lower limbs showed the blockade might cancel these effects, resulting in repigmen- same findings as those observed in the previous biop- tation of hair. sies (figure 1E, F). Immunohistochemical analysis with Taken together, these observations suggest that anti-IL-17A P. acnes-specific monoclonal antibodies (PAB antibodies), antibody treatment can cause unusual clinical manifesta- which specifically react with the P. acnes cell membrane tions. These unexpected responses due to IL-17A blockade [2], revealed small round bodies within the granulomas, increase our knowledge on the role of IL-17A in other skin which were assumed to be P. acnes (figure 1G, H). The diseases. PAB antibody used in this study has been proven to react with a P. acnes-specific epitope of lipoteichoic acid [2]. Disclosure. Financial support: none. Conflict of interest: In addition, the specificity and sensitivity of the antibody none. were confirmed previously [2, 3]. The patient refused our proposal of oral antibiotic treatment. Topical clindamycin Department of Dermatology, Emi MASHIMA gel was not effective for the exanthema. University of Occupational and Yu SAWADA To our knowledge, this is the first case report of GPPD Environmental Health, 1-1 Takashi YAMAGUCHI with localization of P. acnes in the granuloma. In 2011, Iseigaoka, Yahatanishi-ku, Haruna YOSHIOKA Bachmeyer et al. reported a case of systemic sarcoidosis Kitakyushu 807-8555, Japan Shun OHMORI with a purpuric skin manifestation that mimicked GPPD <[email protected]> Sanehito HARUYAMA [4]. It is difficult to distinguish cutaneous sarcoidosis from Etsuko OKADA pigmented purpuric lesions derived from GPPD based on Motonobu NAKAMURA clinicopathological features, and no clear criteria exist that 540 EJD, vol. 28, n◦ 4, July-August 2018 Journal Identification = EJD Article Identification = 3329 Date: September 14, 2018 Time: 2:22 pm ABCD EFGH Figure 1. A) Purpuric plaques on the lower leg at the initial hospital visit. B) Recurrent punctate purpura on the lower leg. C, D) Pathological findings from an exanthema on the leg at the initial hospital visit. C) A biopsy specimen taken from a recurrent exanthema on the arm shows epithelioid granuloma in the superficial dermis. D) Involvement of the dermal capillaries and extravasation of red blood cells are also demonstrated (haematoxylin-eosin; original magnification: ×100 [C] and ×200 [D]). E, F) Pathological findings from a recurrent exanthema on the arm show an epithelioid granuloma with giant cells (haematoxylin-eosin; original magnification: 400× [E] and 1,000× [F)]). G, H) Immunohistochemistry of the biopsy specimen using PAB antibodies showing small, round bodies within the sarcoid granuloma which are assumed to represent P.acnes (original magnification: 400× [G] and 1,000× [H]). may be used to distinguish between GPPD and cutaneous and sarcoidosis patients is needed to clarify the association sarcoidosis with purpuric manifestations if the patient has between P. acnes with GPPD and cutaneous sarcoidosis no other organ involvement [5]. Furthermore, systemic sar- with pigmented purpuric lesions. coidosis with asymptomatic lesions of other organs could be misdiagnosed as GPPD. Disclosure. Financial support: none. Conflict of interest: The localization of P. acnes in granulomas is known to be none. important in the pathogenesis of sarcoidosis [2, 6]. A recent meta-analysis of 11 studies revealed a significant associ- 1 1,2 ation between sarcoidosis and the presence of P. acnes, Department of Dermatology, Aichi Hiroyuki TAKAMA Medical University, Aichi, Japan Takeshi and the detection of P. acnes by immunohistochemistry 2 1 and western blotting analysis was remarkably specific for Department of Dermatology, YANAGISHISTA Nagoya University Graduate School Jun MUTO1 sarcoidosis [7]. Therefore, in this case, the localization of of Medicine, Aichi, Japan Yuichiro OHSHIMA1 P. acnes in granulomas suggests a role for sarcoidal reac- 3 Department of Surgical Pathology, Emiko TAKAHASHI3 tions in the development of GPPD lesions. Aichi Medical University, Aichi, Toyonori TSUZUKI3 To the best of our knowledge, positive cultures of P. acnes Japan Keisuke UCHIDA4 from skin lesions of cutaneous sarcoidosis have not been 4 Division of Surgical Pathology, Yoshinobu EISHI5 reported. Thus, to date, there is only indirect evidence of the Tokyo Medical and Dental Masashi AKIYAMA2 pathogenic role of P.acnes infection in cutaneous sarcoido- University Hospital, Tokyo, Japan Daisuke WATANABE1 sis. There is only one reported case of cutaneous sarcoidosis 5 Department of Human Pathology, associated with P. acnes which was a 25-year-old woman Graduate School of Medical with livedoid sarcoidosis on her back and lower legs; the Sciences, Tokyo Medical and Dental authors considered that specific staining for P. acnes within University, Tokyo, Japan granulomas around the vessels might suggest haematoge- <[email protected]> nous dissemination of P. acnes. Because our case had marked vascular involvement and was resistant to topical 1. Battle LR, Shalin SC, Gao L. Granulomatous pigmented purpuric clindamycin gel, P. acnes in the granuloma of our case was dermatosis. Clin Exp Dermatol 2015; 40: 387-90. also presumed to be disseminated haematogenously rather 2. Negi M, Takemura T, Guzman J, et al. Localization of Propionibac- terium acnes in granulomas supports a possible etiologic link between than transcutaneously. sarcoidosis and the bacterium. Mod Pathol 2012; 25: 1284-97. To detect the localization of P. acnes in GPPD lesions, 3. Suzuki Y, Uchida K, Takemura T, et al. Propionibacterium acnes- immunohistochemical analysis with PAB antibodies may derived insoluble immune complexes in sinus macrophages of lymph be a powerful tool. Further research including more GPPD nodes affected by sarcoidosis. PLoS One 2018; 13: e0192408. EJD, vol. 28, n◦ 4, July-August 2018 541 Journal Identification