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SDHA 相关嗜铬细胞瘤和副神经节瘤:综述与临床管理。

SDHA-related phaeochromocytoma and paraganglioma: review and clinical management.

机构信息

Kolling Institute of Medical Research, The University of Sydney, Sydney, Australia.

Department of Endocrinology, Royal North Shore Hospital, Sydney, Australia.

出版信息

Endocr Relat Cancer. 2024 Sep 21;31(10). doi: 10.1530/ERC-24-0111. Print 2024 Oct 1.

Abstract

Phaeochromocytomas and paragangliomas (collectively termed PPGL) are rare yet highly heritable neuroendocrine tumours, with over one-third of cases associated with germline pathogenic variants (PVs) in numerous genes. PVs in the succinate dehydrogenase subunit-A gene (SDHA) were initially implicated in hereditary PPGL in 2010, and SDHA has since become an important susceptibility gene accounting for up to 2.8% of cases. However, it remains poorly understood, particularly regarding the clinical nature of SDHA PPGL, rates of recurrence and metastasis, and the nature of metastatic disease. We present a narrative review of SDHA-related PPGL, covering pathophysiology, relevance to current clinical practice, and considerations for clinical genetics. We analyse a pool of 107 previously reported cases of SDHA-associated PPGL to highlight the spectrum of SDHA-related PPGL. Our analysis demonstrates that SDHA PPGL occurs across a wide age range (11-81 years) and affects men and women equally. SDHA PPGL typically presents as single tumours (91%), usually occurring in the head and neck (46%) or abdomen (43%, including 15% with phaeochromocytomas). Metastatic disease was reported in 25.5% of cases, with bone (82%) and lymph nodes (71%) being the most common sites of metastasis, often identified many years after the initial diagnosis. A family history of SDHA-related neoplasia was rare, reported in only 4% of cases. Understanding the clinical nature and risks associated with SDHA PVs is essential for facilitating the optimal management of patients and their families.

摘要

嗜铬细胞瘤和副神经节瘤(统称为 PPGL)是罕见但高度遗传性的神经内分泌肿瘤,超过三分之一的病例与许多基因中的种系致病性变异(PVs)有关。2010 年,琥珀酸脱氢酶亚单位-A 基因(SDHA)中的 PVs 最初被认为与遗传性 PPGL 有关,此后,SDHA 已成为一个重要的易感基因,占病例的 2.8%。然而,其仍知之甚少,特别是关于 SDHA PPGL 的临床性质、复发和转移率以及转移性疾病的性质。我们对 SDHA 相关的 PPGL 进行了叙述性综述,涵盖了病理生理学、与当前临床实践的相关性以及临床遗传学的考虑因素。我们分析了一组 107 例先前报道的 SDHA 相关 PPGL 病例,以突出 SDHA 相关 PPGL 的范围。我们的分析表明,SDHA PPGL 发生在广泛的年龄范围内(11-81 岁),男女发病率相等。SDHA PPGL 通常表现为单发肿瘤(91%),通常发生在头颈部(46%)或腹部(43%,包括 15%的嗜铬细胞瘤)。转移性疾病在 25.5%的病例中被报道,骨(82%)和淋巴结(71%)是最常见的转移部位,通常在初始诊断多年后才被发现。仅有 4%的病例报告了 SDHA 相关肿瘤的家族史。了解与 SDHA PVs 相关的临床性质和风险对于促进患者及其家属的最佳管理至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ab07/11466202/22207eef499e/ERC-24-0111fig1.jpg

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