Von Hippel–Lindau disease (VHL), also known as Von Hippel–Lindau syndrome, is a rare genetic disorder with multisystem involvement. It is characterized by polycystic disease and benign tumors with potential for subsequent malignant transformation. It is a type of phakomatosis that results from a mutation in the Von Hippel–Lindau tumor suppressor gene on chromosome 3p25.3.
Signs and symptoms
Signs and symptoms associated with VHL disease include headaches, problems with balance and walking, dizziness, weakness of the limbs, vision problems, and high blood pressure. Six organ systems mainly are affected: the central nervous system; retina; pancreas; kidney; adrenal gland; epididymis. Conditions associated with VHL disease include angiomatosis, hemangioblastomas, pheochromocytoma, renal cell carcinoma, pancreatic cysts (pancreatic serous cystadenoma), endolymphatic sac tumor, and bilateral papillary cystadenomas of the epididymis (men) or broad ligament of the uterus (women). Angiomatosis occurs in 37.2% of patients presenting with VHL disease and usually occurs in the retina. As a result, loss of vision is very common. However, other organs can be affected: strokes, heart attacks, and cardiovascular disease are common additional symptoms. Approximately 40% of VHL disease presents with CNS hemangioblastomas and they are present in around 60–80%. Spinal hemangioblastomas are found in 13–59% of VHL disease and are specific because 80% are found in VHL disease. Although all of these tumors are common in VHL disease, around half of cases present with only one tumor type. Most people with VHL develop symptoms in their mid-twenties.
Pathogenesis The disease is caused by mutations of the Von Hippel–Lindau tumor suppressor (VHL) gene on the short arm of chromosome 3 (3p25-26). There are over 1500 germline mutations and somatic mutations found in VHL disease.
Every cell in the body has two copies of every gene (bar those found in the sex chromosomes, X and Y). In VHL disease, one copy of the VHL gene has a mutation and produces a faulty VHL protein (pVHL). However, the second copy still produces a functional protein. The condition is inherited in an autosomal dominant manner – one copy of the faulty gene is sufficient to increase the risk of developing tumours. Approximately 20% of cases of VHL disease are found in individuals without a family history, known as de novo mutations. An inherited mutation of the VHL gene is responsible for the remaining 80 percent of cases. Of mutations in the VHL gene, 30–40% consist of 50-250kb deletion mutations that remove either part of the gene or the whole gene and flanking regions of DNA. The remaining 60–70% of VHL disease is caused by the truncation of pVHL by nonsense mutations, indel mutations or splice site mutations.
VHL protein
The VHL protein (pVHL) is involved in the regulation of a protein known as hypoxia inducible factor 1α (HIF1α). This is a subunit of a heterodimeric transcription factor that at normal cellular oxygen levels is highly regulated. In normal physiological conditions, pVHL recognizes and binds to HIF1α only when oxygen is present due to the post translational hydroxylation of two proline residues within the HIF1α protein. pVHL is an E3 ligase that ubiquitinates HIF1α and causes its degradation by the proteasome. In low oxygen conditions or in cases of VHL disease where the VHL gene is mutated, pVHL does not bind to HIF1α. This allows the subunit to dimerise with HIF1β and activate the transcription of a number of genes, including vascular endothelial growth factor, platelet-derived growth factor B, erythropoietin and genes involved in glucose uptake and metabolism.
Diagnosis The detection of tumours specific to VHL disease is important in the disease's diagnosis. In individuals with a family history of VHL disease, one hemangioblastoma, pheochromocytoma or renal cell carcinoma may be sufficient to make a diagnosis. As all the tumours associated with VHL disease can be found sporadically, at least two tumours must be identified to diagnose VHL disease in a person without a family history. Genetic diagnosis is also useful in VHL disease diagnosis. In hereditary VHL disease, techniques such as the Southern blot and gene sequencing can be used to analyse DNA and identify mutations. These tests can be used to screen family members of those afflicted with VHL disease; de novo cases that produce genetic mosaicism are more difficult to detect because mutations are not found in the white blood cells that are used for genetic analysis.
Classification Von Hippel-Lindau (VHL) disease is classified into two main types based on the presence or absence of pheochromocytoma (pheo). VHL type 1 is characterized by the absence of pheo, while VHL type 2 encompasses individuals with pheo and is further divided into three subcategories: type 2A, type 2B, and type 2C. Diagnosis of VHL is guided by two key criteria. The first involves patients with a family history of developing hemangioblastomas (HB) in the central nervous system (CNS) or retinal angiomas (RA), pheo, pancreatic tumors or cysts, or epididymal cystadenomas. The second criterion applies to patients without a family history of VHL disease who present with hemangioblastomas or retinal angiomas in conjunction with other tumors such as renal cell carcinoma (RCC), pheo, pancreatic tumors or cysts, or epididymal cystadenomas.
Classification terms Pheo - A pheochromocytoma is an adrenal tumor that makes and releases excess catecholamines. These tumors can cause serious health problems including stroke, heart attack, and even death. Hemangioblastomas - a hemangioblastoma is a tumor that grows in the blood vessels of your brain, spinal cord or retina. It isn't cancerous, but it may grow and press on surrounding tissues. Usually, healthcare providers recommend removing a hemangioblastoma with surgery. After removal, a hemangioblastoma is unlikely to grow back. Retina angiomas - retinal capillary hemangiomas also known as retinal hemangioblastomas, occur most frequently in conjunction with von Hippel-Lindau (VHL) syndrome. These lesions are characterized by plump, but otherwise normal, retinal capillary endothelial cells with normal pericytes and basement membrane. Epidydimal cystadenomas- A cyst that grows on the male testes. Renal cell carcinoma- The most common type of kidney cancer.
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