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拇指末节指骨附属骨化中心:拉森综合征的一项额外影像学表现。

Terminal phalangeal accessory ossification center of the thumb: an additional radiographic finding in Larsen syndrome.

作者信息

Alanay Yasemin, Utine Gulen Eda, Lachman Ralph S, Krakow Deborah, Tuncbilek Ergul

机构信息

Clinical Genetics Unit, Department of Pediatrics, Faculty of Medicine, Hacettepe University, Ankara, Turkey.

出版信息

Pediatr Radiol. 2006 Sep;36(9):970-3. doi: 10.1007/s00247-006-0217-z. Epub 2006 May 20.

Abstract

Larsen syndrome is an autosomal-dominant disorder characterized by multiple joint dislocations, vertebral anomalies and dysmorphic facies. Both autosomal-dominant and autosomal-recessive forms of the disorder have been proposed. Individuals with autosomal-dominant Larsen syndrome have characteristic "cylindrical-shape" thumbs caused by broad, shortened phalanges. Autosomal-dominant Larsen syndrome results from heterozygosity for mutations in filamin B, a cytoskeletal protein involved in multicellular processes. We report here a patient with a duplicated or accessory distal thumb phalanx and multiple large joint dislocations who was shown to be heterozygous for a filamin B mutation predicting the amino acid substitution G1691S. This adds a new radiographic finding, duplicated or accessory distal phalanx, to the radiographic abnormalities seen in this rare dominant disorder.

摘要

拉森综合征是一种常染色体显性疾病,其特征为多处关节脱位、脊椎异常和面部畸形。该疾病已被提出存在常染色体显性和常染色体隐性两种形式。患有常染色体显性拉森综合征的个体,由于指骨宽阔、缩短而具有特征性的“圆柱形”拇指。常染色体显性拉森综合征是由细丝蛋白B(一种参与多细胞过程的细胞骨架蛋白)突变的杂合性引起的。我们在此报告一名患者,其拇指远端指骨重复或有副指骨,并有多处大关节脱位,经检测该患者细丝蛋白B突变杂合,预测氨基酸替代为G1691S。这为这种罕见的显性疾病的影像学异常增加了一项新的影像学发现,即拇指远端指骨重复或有副指骨。

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