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希佩尔-林道病与兄弟姐妹中的快速进展性嗜铬细胞瘤

Von Hippel-Lindau disease and rapidly progressing pheochromocytomas in siblings.

作者信息

Fugaru Ioana, Goudie Catherine, Capolicchio John-Paul

机构信息

Division of Urology, Department of Surgery, McGill University Health Centre, Montreal, QC, Canada.

Division of Hematology-Oncology, Department of Pediatrics, Montreal Children's Hospital, McGill University Health Centre, Montreal, QC, Canada.

出版信息

Fam Cancer. 2022 Apr;21(2):229-233. doi: 10.1007/s10689-021-00252-2. Epub 2021 Apr 20.

Abstract

Von Hippel-Lindau (VHL) disease is an autosomal dominant inherited condition with a predisposition to the development of a variety of tumors including pheochromocytomas. A number of cancer surveillance protocols for patients with VHL have been developed, all of which are based on expert opinion. We report a case of two brothers with a strong family history of VHL type 2 due to a pathogenic germline VHL variant, specifically, a surface missense substitution, with a rapidly progressive clinical course that both presented with a large adrenal mass. Both brothers presented with large pheochromocytomas, the earliest presentation being at age 7, despite routine screening. The rapid progression and early presentation of these patients raises an important discussion around the commonly used surveillance protocols for pheochromocytoma in pediatric patients with VHL and missense mutations. We conclude that a more accelerated surveillance protocol may be adequate for VHL families with a high pheochromocytoma risk.

摘要

冯·希佩尔-林道(VHL)病是一种常染色体显性遗传性疾病,易患包括嗜铬细胞瘤在内的多种肿瘤。针对VHL患者,已经制定了许多癌症监测方案,所有这些方案均基于专家意见。我们报告了一例病例,两名兄弟因致病性胚系VHL变异,具体为表面错义替代,有VHL 2型的强烈家族病史,临床病程进展迅速,两人均表现为巨大肾上腺肿块。尽管进行了常规筛查,但两兄弟均患有巨大嗜铬细胞瘤,最早发病年龄为7岁。这些患者的快速进展和早期发病引发了关于VHL和错义突变小儿患者嗜铬细胞瘤常用监测方案的重要讨论。我们得出结论,对于嗜铬细胞瘤风险高的VHL家族,更加速的监测方案可能是合适的。

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