Bekers Elise M, Eijkelenboom Astrid, Grünberg Katrien, Roverts Rona C, de Rooy Jacky W J, van der Geest Ingrid C M, van Gorp Joost M, Creytens David, Flucke Uta
Department of Pathology, Radboud University Medical Center, Nijmegen, The Netherlands; Department of Pathologie (Pathologie-DNA), Jeroen Bosch Hospital, Den Bosch, The Netherlands.
Department of Pathology, Radboud University Medical Center, Nijmegen, The Netherlands.
Ann Diagn Pathol. 2018 Jun;34:56-59. doi: 10.1016/j.anndiagpath.2018.01.006. Epub 2018 Jan 12.
Myositis ossificans is defined as a self-limiting pseudotumor composed of reactive hypercellular fibrous tissue and bone. USP6 rearrangements have been identified as a consistent genetic driving event in aneurysmal bone cyst and nodular fasciitis. It is therefore an integral part of the diagnostic workup when dealing with (myo)fibroblastic lesions of soft tissue and bone. Two cases of myositis ossificans with USP6 rearrangement were published so far. We determine herein the incidence of USP6 rearrangement in myositis ossificans using USP6 fluorescence in situ hybridization analysis (FISH). Of the 11 cases included, seven patients were female and four were male. Age ranged from 6 to 56 years (mean 27 years). Lesions were located in the thigh (n = 5), knee (n = 1), lower leg (n = 1), lower arm (n = 1), perineum (n = 1), gluteal (n = 1) and thoracic wall (n = 1). All assessable cases except one (8/9) showed rearrangement of USP6 providing evidence that myositis ossificans is genetically related to nodular fasciitis and aneurysmal bone cyst.
骨化性肌炎被定义为由反应性细胞增多的纤维组织和骨组成的自限性假瘤。USP6重排已被确定为动脉瘤样骨囊肿和结节性筋膜炎中一致的基因驱动事件。因此,在处理软组织和骨的(肌)纤维母细胞性病变时,它是诊断检查不可或缺的一部分。迄今为止,已发表了2例伴有USP6重排的骨化性肌炎病例。我们在此使用USP6荧光原位杂交分析(FISH)确定骨化性肌炎中USP6重排的发生率。在纳入的11例病例中,7例为女性,4例为男性。年龄范围为6至56岁(平均27岁)。病变位于大腿(n = 5)、膝关节(n = 1)、小腿(n = 1)、下臂(n = 1)、会阴(n = 1)、臀肌(n = 1)和胸壁(n = 1)。除1例(8/9)外,所有可评估病例均显示USP6重排,这表明骨化性肌炎在基因上与结节性筋膜炎和动脉瘤样骨囊肿相关。