Assistance Publique-Hôpitaux de Paris, Hôpital de Bicêtre, Service d'Endocrinologie et des Maladies de la Reproduction, Centre de Référence des Maladies Rares de l'Hypophyse, Le Kremlin-Bicêtre, France.
Service d'Endocrinologie, Maladies Métaboliques et Nutrition, CHU Amiens Picardie, Amiens, France.
Nat Rev Endocrinol. 2021 Apr;17(4):207-224. doi: 10.1038/s41574-021-00468-3. Epub 2021 Feb 9.
Multiple endocrine neoplasia type 1 (MEN1) is a rare syndrome characterized by the co-occurrence of primary hyperparathyroidism, duodenopancreatic neuroendocrine tumours (NETs) and/or pituitary adenomas. MEN1 can predispose patients to other endocrine and non-endocrine tumours, such as cutaneous tumours, central nervous system tumours and breast cancer. Endocrine tumours in patients with MEN1 differ from sporadic tumours in that they have a younger age at onset, present as multiple tumours in the same organ and have a different clinical course. Therefore, patients with overt MEN1 and those who carry a MEN1 mutation should be offered tailored biochemical and imaging screening to detect tumours and evaluate their progression over time. Fortunately, over the past 10 years, knowledge about the clinical phenotype of these tumours has markedly progressed, thanks to the implementation of national registries, particularly in France and the Netherlands. This Review provides an update on the clinical management of MEN1-related tumours. Epidemiology, the clinical picture, diagnostic work-up and the main lines of treatment for MEN1-related tumours are summarized. Controversial therapeutic aspects and issues that still need to be addressed are also discussed. Moreover, special attention is given to MEN1 manifestations in children and adolescents.
多发性内分泌腺肿瘤 1 型(MEN1)是一种罕见的综合征,其特征是甲状旁腺功能亢进、十二指肠胰腺神经内分泌肿瘤(NETs)和/或垂体腺瘤同时发生。MEN1 可使患者易患其他内分泌和非内分泌肿瘤,如皮肤肿瘤、中枢神经系统肿瘤和乳腺癌。MEN1 患者的内分泌肿瘤与散发性肿瘤不同,其发病年龄较小,同一器官存在多个肿瘤,且具有不同的临床病程。因此,对于显性 MEN1 患者和携带 MEN1 突变的患者,应提供量身定制的生化和影像学筛查,以检测肿瘤并评估其随时间的进展。幸运的是,在过去的 10 年中,由于实施了国家登记处,特别是在法国和荷兰,这些肿瘤的临床表型的知识有了显著的进展。这篇综述提供了 MEN1 相关肿瘤的临床管理的最新信息。总结了 MEN1 相关肿瘤的流行病学、临床表现、诊断方法和主要治疗方法。还讨论了有争议的治疗方面和仍需解决的问题。此外,还特别关注了儿童和青少年的 MEN1 表现。