| Literature DB >> 16715218 |
Yasemin Alanay1, Gulen Eda Utine, Ralph S Lachman, Deborah Krakow, Ergul Tuncbilek.
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.Entities:
Mesh:
Year: 2006 PMID: 16715218 DOI: 10.1007/s00247-006-0217-z
Source DB: PubMed Journal: Pediatr Radiol ISSN: 0301-0449