Key result
Multidisciplinary management is essential for the surveillance, treatment, and follow-up of multiple primary tumors in patients with von Hippel-Lindau disease, given the limited available evidence.
Why the study?
Very limited evidence is available to guide the surveillance, treatment, and follow-up of the multiple primary tumors developed by patients diagnosed with von Hippel-Lindau disease.
This review highlights the limited evidence available for the multidisciplinary management of von Hippel-Lindau disease, a rare hereditary disorder.
Highlights VHL management challenges amid limited evidence; leaves open need for prospective trials on diagnostics and therapies.
Objective: The aim of the current review is to summarize the available evidence to aid clinicians in the surveillance, treatment and follow-up of the different primary tumors developed by patients diagnosed with von Hippel-Lindau (VHL) syndrome. Methods: A non-systematic narrative review of original articles, meta-analyses, and randomized trials was conducted, including articles in the pre-clinical setting to support relevant findings. Results: VHL disease is the most common rare hereditary disorder associated with clear cell renal cell carcinoma. Affected individuals inherit a germline mutation in one VHL allele, and any somatic event that disrupt the other allele can trigger mutations, chromosomal rearrangements, or epigenetic regulations leading to oncogenesis. From a clinical perspective, patients continuously develop multiple primary tumors. Conclusion: Because VHL is considered a rare disease, very limited evidence is available for diagnosis, surveillance, active treatment with local or systemic therapy and follow-up.
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Larcher et al. (2022) conducted a review in von Hippel-Lindau (VHL) disease. Multidisciplinary management was evaluated. Multidisciplinary management is essential for the surveillance, treatment, and follow-up of multiple primary tumors in patients with von Hippel-Lindau disease, given the limited available evidence.
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