Annotation Detail

Information
Associated Genes
VHL
Associated Variants
VHL p.Ala149Ser (p.A149S) ( ENST00000256474.3, ENST00000345392.3, ENST00000696143.2, ENST00000696153.1, ENST00000713811.1, ENST00000713812.1, ENST00000713815.1, ENST00000713982.1 )
VHL p.Ala149Ser (p.A149S) ( 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
49 family members from three generations of a Turkish family were examined. A clinical presentation of VHL type 2B was noted. Genetic testing was performed on 17 family members. 11 members with the mutation had manifestations, while 6 members with the mutation were asymptomatic. 1 of the asymptomatic individuals belonged to generation 2, while the remaining 5 belonged to the fourth generation, suggesting they may be too young to present manifestations. From the members that had manifestations: 9 had pheochromocytoma (5 bilateral), 1 had lumbar spine hemangioblastoma, 1 had retinal hemangioblastoma, 4 had renal cell carcinoma (2 bilateral), and 3 had pancreatic neuroendocrine tumours (1 also had a serous cystadenoma in addition to the neuroendocrine tumour). ACMG evidence codes: 'PP1' because of segregation results, 'PP2' because there is a missense variant in a gene that has a low rate of benign missense variation and in which missense variants are a common mechanism of disease, 'PP4' because the patient's phenotype or family history is highly specific for a disease with a single genetic etiology. Relevant HPO terms: Renal cell carcinoma, Spinal hemangioblastoma, Multiple pancreatic cysts, Pheochromocytoma, Neuroendocrine neoplasm, Retinal hemangioblastoma.
Variant Origin
germline
Variant Origin
Rare Germline
Evidence URL
https://civic.genome.wustl.edu/links/evidence_items/5679
Gene URL
https://civic.genome.wustl.edu/links/genes/58
Variant URL
https://civic.genome.wustl.edu/links/variants/820
Rating
3
Evidence Type
Predisposing
Disease
Von Hippel-Lindau Disease
Evidence Direction
Supports
Evidence Level
C
Clinical Significance
Uncertain Significance
Pubmed
23673869
Drugs