Skip to main content

Von Hippel-Lindau Syndrome

OMIM ID:

autosomal dominant

Von Hippel-Lindau Syndrome

Alternate Names

VHL
angiomatosis retinae
VHL syndrome

Defective Genes

VHL

Clinical Characteristics

Ocular Features

Retinal angiomas are a feature of this syndrome, occurring in up to 70% of patients and often diagnosed by about age 25 years.  These hemangioblastomas are often connected to prominent arterioles and venules indicative of their vascular nature.  Capillary hamartomas located on or near the optic nerve may mimic papilledema or papillitis.  However, they may also occur throughout the retina and visual morbidity often results from secondary damage due to hemorrhage, exudates, and traction on the retina. When they are bilateral and multicentric the diagnosis of VHL is highly likely.  Patients with VHL tend to develop such tumors at a younger age and have worse visual outcomes than those in patients without VHL.  The impact on vision is responsible for initial presentation in many patients.

Systemic Features

Clinical symptoms typically have their onset during the second decade of life.  These commonly (in 35% of patients) result from the presence of a cerebellar hemangioblastoma while overall more than 60% eventually develop this malignancy.  Up to 40% of patients develop renal cell carcinomas and these are a major cause of death.   However, benign and malignant tumors may appear in many organs including the adrenal glands, pancreas, and spinal cord.  Pheochromocytomas occur in 20-35% of individuals and may be bilateral and multifocal.  These can induce an erythrocythemia. Endolymphatic sac tumors occur in about 10% of patients.  Cystic lesions are often associated with the tumors, especially in the pancreas.

Several subtypes have been proposed based on the pattern of malignancies and the types of mutations found in patients.

Genetics

Inheritance

This is an autosomal dominant cancer susceptibility disorder caused by a mutation in the VHL gene located at 3p26-p25.

There is evidence that the phenotype can be modified by variations in the cyclin D1 gene (CCND1) located at 11q13.

Pedigree

Autosomal dominant

Autosomal dominant disorders require only one mutation for the disease to be expressed.  Since an affected parent has two chromosomes, only one of which has the mutant gene, parents can expect that half (50%) of their children will receive that one and inherit the disease.  It is common for individuals that inherit the mutation, however, to not have evidence of the disease (nonpenetrance).

Image
Sample pedigree of autosomal dominant inheritance

Autosomal dominant inheritance leads to a vertical pattern of transmission

Treatment & Management

Local excision of isolated lesions can be considered in selected cases.  Photocoagulation and cryotherapy of retinal hamartomas can be considered although outcomes are variable depending on location and size of the lesions.

Publications

Displaying 1 - 4 of 4

A genetic register for von Hippel-Lindau disease.

PubMedID: 8929948

Genetic analysis of von Hippel-Lindau disease

PubMedID: 20151405

Juxtapapillary capillary hemangiomas

PubMedID: 11097604

Von Hippel-Lindau Disease

PubMedID: 27153780