Acta Dermato-Venereologica issue 50:1 98-1CompleteContent | Page 28

119 SHORT COMMUNICATION Extensive Eruptive Syringoma After Liver Transplantation Takuya MAEDA 1 , Ken NATSUGA 1 *, Wataru NISHIE 1 , Kenichiro YAMASHITA 2 and Hiroshi SHIMIZU 1 1 Department of Dermatology, and 2 Department of Transplant Surgery, Hokkaido University Graduate School of Medicine, N15 W7, Sapporo 060-8638, Japan. *E-mail: [email protected] Accepted Oct 2, 2017; Epub ahead of print Oct 3, 2017 A syringoma is a benign appendageal neoplasm that typically presents in adolescent females. Clinically, syringomas appear as multiple small papules with sym- metrical distribution and are normal skin-coloured or slightly pigmented. The 4 subtypes of syringoma are classified according to their clinical features: localized, familial, Down’s syndrome-associated and generalized (encompassing eruptive syringoma) (1–4). The eruptive variant of syringoma, which is observed less commonly than other subtypes, is 1–5 mm in size and is predomi- nantly distributed over the anterior trunk, neck, abdomen and axillae (2, 3). We present here a case of a 70-year-old woman with extensive eruptive syringomas that develo- ped after deceased donor liver transplantation (DDLT). CASE REPORT A 70-year-old Japanese woman was referred to our department with an 11-year history of papules on the trunk and upper limbs. The lesions appeared soon after DDLT for her primary biliary cirrhosis and subsequent immunosuppressive therapy (tacrolimus hydrate, myco­ phenolate mofetil and corticosteroids). No exacerbation or spontaneous remission had been observed during the 11 years. There was no family history of similar eruptions. Physical examination revealed multiple light- brown papules up to 7 mm in diameter disseminated on the trunk and upper extremities (Fig. 1A). There were no eruptions on the lower extremities, the oral mucosa or the genital area. Physical examination revealed no other significant symptoms, and laboratory results were unremarkable. In light of the immunosuppressive back- ground, she was initially suspected of having verruca plana. Histological examination of a biopsy specimen from a papule on the right forearm (Fig. 1B) revealed multiple small ducts embedded in fibrous stroma. The ducts were lined by 2 layers of epithelial cells and were filled with accumulated eosinophilic secretions. Some of the cells had a tadpole appearance (Fig. 1C). These findings led to a diagnosis of eruptive syringoma. DISCUSSION Since syringoma eruptions appear as non-specific pa- pules, clinical diagnosis of eruptive syringoma can be difficult. The differential diagnoses include xanthoma, verrucae, mastocytosis, milia, lichen planus, dissemina- ted molluscum contagiosum and viral-related trichostasis spinulosa. The diagnostic difficulty of the present case owed to the fact that the patient was referred 11 years after liver transplantation and each lesion was larger than a typical syringoma. Although the pathogenesis of eruptive syringoma remains unknown, some studies have suggested an association between syringomas and inflammatory reactions, such as contact dermatitis and drug hypersensitivity (4, 5). To our knowledge, this is the first case of eruptive syringoma occurring after liver transplantation. Apart from the current case, a case of eruptive syringoma that developed after kidney trans- plantation has been reported (6). These 2 cases might imply the involvement of immunosuppression in the pathogenesis of syringomas, in contrast to the previous Fig. 1. (A) Clinical presentation. Multiple brown papules distributed on the trunk and upper extremities. (B) A skin biopsy was performed from a papule on the right forearm. (C) Pathology of the biopsy specimen. The specimen reveals multiple small ducts embedded in fibrous stroma. Some of the cells show a tadpole appearance (haematoxylin and eosin staining, original magnification ×100). This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta Journal Compilation © 2018 Acta Dermato-Venereologica. doi: 10.2340/00015555-2814 Acta Derm Venereol 2018; 98: 119–120