McFadden Kathryn, Ranganathan Sarangarajan
Department of Pathology, Children's Hospital of University of Pittsburgh Medical Center, B250 Main Hospital, 4401 Penn Avenue, Pittsburgh PA 15224.
Pediatr Dev Pathol. 2015 May-Jun;18(3):228-30. doi: 10.2350/14-09-1551-CR.1. Epub 2015 Jan 8.
Metachromatic leukodystrophy (MLD) is an autosomal recessive lysosomal storage disease caused by deficient activity of arylsulfatase A, leading to an accumulation of sulfatide in myelinating cells and progressive central and peripheral demyelination. Sulfatide also accumulates in various organs, most notably the gallbladder. Gallbladder mucosal hyperplasia with papillomatosis, in the setting of abdominal pain and hemobilia, is often demonstrated by sonography. We present a histologic and ultrastructural description of these alterations in a 5-year-old girl with MLD who presented with abdominal pain and feeding intolerance. Gross and light microscopic analysis demonstrated diffuse villous hyperplasia of the gallbladder mucosa with intraepithelial and intravillous macrophage accumulations of metachromatic material. Electron microscopic analysis demonstrated this material to comprise numerous membrane-bound inclusions composed of concentrically lamellated, dense material. The rarity of descriptions of this finding prompted this case report.
异染性脑白质营养不良(MLD)是一种常染色体隐性溶酶体贮积病,由芳基硫酸酯酶A活性缺乏引起,导致髓鞘形成细胞中硫脂蓄积,以及中枢和周围神经系统进行性脱髓鞘。硫脂也在各种器官中蓄积,最显著的是胆囊。超声检查常显示,在腹痛和胆道出血的情况下,胆囊黏膜增生伴乳头瘤病。我们对一名5岁患有MLD且伴有腹痛和喂养不耐受的女孩的这些病变进行了组织学和超微结构描述。大体和光学显微镜分析显示胆囊黏膜弥漫性绒毛增生,上皮内和绒毛内巨噬细胞蓄积异染物质。电子显微镜分析显示,这种物质由许多膜结合包涵体组成,包涵体由同心层状致密物质构成。这一发现的罕见性促使了本病例报告的撰写。