与 SDHA 相关的嗜铬细胞瘤和副神经节瘤:综述和临床治疗。

Endocrine-related cancer Pub Date : 2024-09-21 Print Date: 2024-10-01 DOI:10.1530/ERC-24-0111
Adam I Kaplan, Trisha Dwight, Catherine Luxford, Diana E Benn, Roderick J Clifton-Bligh
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摘要

辉铬细胞瘤和副神经节瘤(统称为 PPGL)是一种罕见但高度遗传的神经内分泌肿瘤,超过三分之一的病例与许多基因的种系致病变异(PVs)有关。2010 年,琥珀酸脱氢酶亚基-A 基因(SDHA)的致病变体首次被认为与遗传性 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,人们对它的了解仍然很少,尤其是对SDHA PPGL的临床性质、复发率和转移率以及转移性疾病的性质了解甚少。我们对与 SDHA 相关的 PPGL 进行了叙述性综述,内容包括病理生理学、与当前临床实践的相关性以及临床遗传学方面的注意事项。我们分析了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 的发病年龄跨度很大(11-81 岁),男性和女性的发病率相当。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)。25.5%的病例出现转移性疾病,骨(82%)和淋巴结(71%)是最常见的转移部位,通常在初次诊断多年后才被发现。与SDHA相关的肿瘤家族史很少见,仅占4%。了解与SDHA PV相关的临床性质和风险对于促进患者及其家属的最佳治疗至关重要。
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SDHA-related phaeochromocytoma and paraganglioma: review and clinical management.

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.

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