Case report Autoerythrocyte sensitization syndrome (GardnereDiamond syndrome) associated with cutaneous vasculitis Do ¨ndu ¨ Cansu a , Timuc ¸in Kas xifo˘ glu a ,O ¨ zgu ¨l Pas xao˘ glu b , Cengiz Korkmaz a, * a Division of Rheumatology, Department of Internal Medicine, Eskis xehir Osmangazi University, Medical Faculty, Vis xnelik M. Alifuat Gu ¨ven C. Akasya S. 11/11, Eskis xehir 26480, Turkey b Department of Pathology, Eskis xehir Osmangazi University, Medical Faculty, Eskis xehir 26480, Turkey Accepted 23 October 2007 Available online 15 April 2008 Abstract Autoerythrocyte sensitization syndrome (ASS) (GardnereDiamond syndrome) is characterized by painful ecchymotic lesions affecting mostly women with emotional stress. Although it is widely accepted as a non-inflammatory disease, ASS can be accompanied by some autoimmune diseases. In this case report, we present a case with ASS associated with cutaneous vasculitis. We also briefly discuss the possible inflammatory features of ASS. Ó 2008 Elsevier Masson SAS. All rights reserved. Keywords: Autoerythrocyte sensitization syndrome; GardnereDiamond syndrome; Inflammation; Stress; Vasculitis 1. Introduction Autoerythrocyte sensitization syndrome (ASS) and Gard- nereDiamond syndrome are characterized by painful ecchy- motic lesions that mostly affect women suffering from emotional stress or psychiatric disorders [1]. Although its pathogenesis is not exactly known, some speculations have been suggested so far [1,2]. Most of the patients with ASS suf- fering psychiatric disorders, ASS is defined as a psychogenic purpura. This syndrome was first described by Gardner and Diamond who postulated it as an autoerythrocyte sensitization syndrome [1]. It was shown that the reaction occurs against the stromal erythrocyte phospholipids, particularly phosphatidyl- serine [3]. Although ASS is not accepted as an inflammatory disease, some patients with ASS can present with immuno- inflammatory features [4e8]. In this case report, we present a case with ASS associated with cutaneous vasculitis. We also briefly discuss possible inflammatory features of ASS. 2. Case report A 52-year-old woman was admitted to our hospital with spontaneous painful ecchymotic bruises over the anterior and posterior aspects of the thighs and forearms. Prior to ad- mission to our hospital, she had a history of two episodes of painful ecchymotic bruises over the same areas. Her lesions had resolved within 1e2 weeks. Her past medical history in- cluded psychotic depression for 3 years, for which she had been treated with antidepressive drugs. Her physical examination revealed painful bilateral ecchymotic bruises over the anterior aspects of the thighs and forearms (Fig. 1). She had fever (38.8  C). Her labora- tory findings were as follows: erythrocyte sedimentation rate 18 mm/h, C-reactive protein 7.19 mg/dl (n < 0.8), haemo- globin 13 g/dl, platelet count 233,000 mm 3 , and white blood cells count 4400 mm 3 . Other haematologic and rheumato- logic tests were normal. The punch biopsy obtained from lesions located in the left leg showed neutrophils infiltration in some small vessel walls at the dermis and subcutaneous fat associated with accumulation of fibrinous material (Fig. 2a, b). Only fibrinogen accumulation was noted upon * Corresponding author. Tel./fax: þ90 222 2392979. E-mail address: ckorkmaz@ogu.edu.tr (C. Korkmaz). 1297-319X/$ - see front matter Ó 2008 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.jbspin.2007.10.007 Available online at www.sciencedirect.com Joint Bone Spine 75 (2008) 721e724