Unilateral adrenalectomy partially improved hyperglycemia in a patient with primary bilateral macronodular adrenal hyperplasia.
Unilateral adrenalectomy partially improved hyperglycemia in a patient with primary bilateral macronodular adrenal hyperplasia.
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单侧肾上腺切除术部分改善了原发性双侧肾上腺大结节增生患者的高血糖症状。
DOI：
10.1007/s13340-021-00503-8
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发表时间：
2021
影响因子：
2.2
通讯作者：
Fujimoto S
中科院分区：
文献类型：
--
作者：
Nishiyama M;Karashima T;Iwasaki Y;Terada Y;Fujimoto S
原发性双侧肾上腺大结节增生（PBMAH）的特征是双侧肾上腺多发性大结节，常引起轻度皮质醇过度分泌，表现为亚临床库欣综合征。我们在此描述一个病例，其中单侧肾上腺切除术部分改善了PBMAH患者的高血糖，并提出了这种手术策略的有用性和局限性。一名64岁的女性2型糖尿病患者偶然诊断为双侧肾上腺病变。她有2型糖尿病家族史，胰岛素治疗时HbA 1c水平为8.9%。她没有出现任何与库欣综合征相关的症状。基础皮质醇水平在正常范围内（12.0 μg/dL）;然而，促肾上腺皮质激素（ACTH）水平受到抑制（2.1 pg/mL），地塞米松试验中血清皮质醇水平未受到抑制。计算机断层扫描和磁共振成像显示双侧肾上腺大结节和131 I-肾上腺甾醇积聚在双侧肾上腺病变。总的来说，她被诊断为亚临床库欣综合征，由于PBMAH并发糖尿病、高血压和血脂异常。行腹腔镜左肾上腺切除术，病理结果与PBMAH一致。单侧肾上腺切除术后，血清皮质醇水平下降，高血压改善。HbA 1c水平和胰岛素需求也下降，但仍需要持续胰岛素治疗。应该注意的是，PBMAH患者的高血糖症在手术成功后可能无法治愈。如果单侧肾上腺切除术不能纠正PBMAH患者的皮质醇增多症，应考虑额外的手术或药物治疗。
Primary bilateral macronodular adrenal hyperplasia (PBMAH) is characterized by bilateral multiple adrenal macro-nodules that often cause mild over-secretion of cortisol in the form of subclinical Cushing’s syndrome. We herein describe a case, wherein unilateral adrenalectomy partially improved hyperglycemia in a patient with PBMAH and suggest the usefulness and limitations of this surgical strategy. A 64-year-old woman with type 2 diabetes had an incidental diagnosis of bilateral adrenal lesions. She had a family history of type 2 diabetes, and her HbA1c level was 8.9% under insulin therapy. She did not present with any symptoms associated with Cushing’s syndrome. The basal cortisol level was in the normal range (12.0 μg/dL); however, the adrenocorticotropic hormone (ACTH) level was suppressed (2.1 pg/mL) and the serum cortisol level was not suppressed in the dexamethasone test. Computed tomography and magnetic resonance imaging showed bilateral adrenal macro-nodules and131I-adosterol accumulated in the bilateral adrenal lesions. Collectively, she was diagnosed with subclinical Cushing’s syndrome due to PBMAH complicated with diabetes mellitus, hypertension, and dyslipidemia. Laparoscopic left adrenalectomy was performed, and the pathologic findings were consistent with PBMAH. After unilateral adrenalectomy, serum cortisol levels decreased, and hypertension improved. Both HbA1c levels and insulin requirement also decreased, but insulin therapy was continuously needed. It should be noted that hyperglycemia may not be cured after successful surgery in a patient with PBMAH. Additional operation or medical therapy should be considered if unilateral adrenalectomy is unable to correct hypercortisolism in PBMAH patients.