Annotation Detail

Information
Associated Genes
VHL
Associated Variants
VHL p.Arg167Gln (p.R167Q) ( ENST00000256474.3, ENST00000345392.3, ENST00000696143.2, ENST00000696153.1, ENST00000713811.1, ENST00000713812.1, ENST00000713815.1, ENST00000713982.1 )
VHL p.Arg167Gln (p.R167Q) ( ENST00000256474.3, ENST00000345392.3, ENST00000696143.2, ENST00000696153.1, ENST00000713811.1, ENST00000713812.1, ENST00000713815.1, ENST00000713982.1 )
Associated Disease
von Hippel-Lindau disease
Source Database
CIViC Evidence
Description
A study of 34 Polish families revealed germline mutations in 30 families. Mutations were not detected in 4 probands fulfilling clinical criteria for von Hippel-Lindau disease (VHLD). Arg167Trp and Arg167Gln mutations conferred a high (62%) risk for pheochromocytoma. Pheochromocytoma was diagnosed in all families with Arg167Gln mutation. This missense mutation was found in a VHL patient with retinal hemangioblastomas and pheochromocytoma (family no. 17). This mutation was also found in a VHL family of 8 affected patients (family no. 18). Seven had retinal hemangioblastomas, six had hemangioblastomas of the central nervous system, and 2 had pheochromocytoma. Cosegregation of the disease in multiple affected family members in a gene definitively known to cause the disease (ACMG code: PP1).
Variant Origin
germline
Variant Origin
Rare Germline
Evidence URL
https://civic.genome.wustl.edu/links/evidence_items/5487
Gene URL
https://civic.genome.wustl.edu/links/genes/58
Variant URL
https://civic.genome.wustl.edu/links/variants/1739
Rating
2
Evidence Type
Predisposing
Disease
Von Hippel-Lindau Disease
Evidence Direction
Supports
Evidence Level
C
Clinical Significance
Uncertain Significance
Pubmed
12114495
Drugs