Endocrinology & Nutrition Department, Ramón y Cajal University Hospital, Madrid, Spain.
Instituto Ramón y Cajal de Investigación Sanitaria IRYCIS, Madrid, Spain.
Front Endocrinol (Lausanne). 2022 Aug 5;13:913253. doi: 10.3389/fendo.2022.913253. eCollection 2022.
Cushing´s syndrome (CS) secondary to bilateral adrenal cortical disease may be caused by bilateral macronodular adrenal cortical disease (BMACD) or by bilateral micronodular adrenal cortical disease (miBACD). The size of adrenal nodules is a key factor for the differentiation between these two entities (>1cm, BMACD and <1cm; miBACD). BMACD can be associated with overt CS, but more commonly it presents with autonomous cortisol secretion (ACS). Surgical treatment of BMACD presenting with CS or with ACS and associated cardiometabolic comorbidities should be the resection of the largest adrenal gland, since it leads to hypercortisolism remission in up to 95% of the cases. Medical treatment focused on the blockade of aberrant receptors may lead to hypercortisolism control, although cortisol response is frequently transient. miBACD is mainly divided in primary pigmented nodular adrenocortical disease (PPNAD) and isolated micronodular adrenocortical disease (i-MAD). miBACD can present at an early age, representing one of the main causes of CS at a young age. The high-dose dexamethasone suppression test can be useful in identifying a paradoxical increase in 24h-urinary free cortisol, that is a quite specific in PPNAD. Bilateral adrenalectomy is generally the treatment of choice in patients with overt CS in miBACD, but unilateral adrenalectomy could be considered in cases with asymmetric disease and mild hypercortisolism. This article will discuss the clinical presentation, genetic background, hormonal and imaging features and treatment of the main causes of primary bilateral adrenal hyperplasia associated with hypercortisolism.
库欣综合征(CS)继发于双侧肾上腺皮质疾病,可能由双侧大结节性肾上腺皮质疾病(BMACD)或双侧小结节性肾上腺皮质疾病(miBACD)引起。肾上腺结节的大小是区分这两种疾病的关键因素(>1cm,BMACD 和 <1cm;miBACD)。BMACD 可伴有明显 CS,但更常见的是伴有自主皮质醇分泌(ACS)。对于表现为 CS 或 ACS 并伴有心脏代谢合并症的 BMACD,手术治疗应切除最大的肾上腺,因为这可使高达 95%的病例皮质醇过度症得到缓解。针对异常受体的药物治疗可能会导致皮质醇过度症得到控制,尽管皮质醇反应通常是短暂的。miBACD 主要分为原发性色素性结节性肾上腺皮质疾病(PPNAD)和孤立性小结节性肾上腺皮质疾病(i-MAD)。miBACD 可在年轻时发病,是年轻人 CS 的主要原因之一。大剂量地塞米松抑制试验有助于识别 24 小时尿游离皮质醇的反常增加,这在 PPNAD 中是相当特异的。在 miBACD 中,对于明显 CS 的患者,双侧肾上腺切除术通常是治疗的首选,但对于疾病不对称和轻度皮质醇过度症的患者,可以考虑单侧肾上腺切除术。本文将讨论与皮质醇过度症相关的原发性双侧肾上腺增生的主要病因的临床表现、遗传背景、激素和影像学特征及治疗。