| Literature DB >> 29619618 |
Eryn Dow1, Ingrid M Winship2,3.
Abstract
Hemangioblastomas are rare vascularized central nervous system tumors, which can occur sporadically or be associated with von Hippel Lindau Syndrome. The pathogenesis of hemangioblastomas in von Hippel Lindau Syndrome is proposed to involve a pseudohypoxic intracellular state induced by dysregulation of hypoxia inducible factor alpha due to the absence of von Hippel Lindau protein complex mediated destruction. Dysregulation of fumarate hydratase, a tricarboxylic acid cycle enzyme, occurs in Hereditary Leiomyomatosis and Renal Cell Cancer Syndrome due to germline fumarate hydratase gene mutations, and also results in oncogenesis via hypoxia inducible factor alpha dysregulation. We present a case study of hemangioblastoma occurrence in a Hereditary Leiomyomatosis and Renal Cell Cancer Syndrome patient and propose it as possible evidence of a phenotypic overlap between von Hippel Lindau and Hereditary Leiomyomatosis and Renal Cell Cancer Syndromes due to their overlapping role in the biochemical regulation of hypoxia inducible factor alpha.Entities:
Keywords: Hemangioblastoma; Hereditary Leiomyomatosis and Renal Cell Cancer Syndrome (HLRCC); Hypoxia Inducible Factor alpha (HIFα); Von Hippel Lindau Syndrome (VHL)
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Year: 2019 PMID: 29619618 DOI: 10.1007/s10689-018-0081-7
Source DB: PubMed Journal: Fam Cancer ISSN: 1389-9600 Impact factor: 2.375