Aiga Ko, Kometani Mitsuhiro, Aono Daisuke, Yoneda Takashi
Department of Health Promotion and Medicine of Future, Kanazawa University Graduate School of Medicine, Kanazawa, Ishikawa 920-8641, Japan.
JCEM Case Rep. 2023 Jan 11;1(1):luac032. doi: 10.1210/jcemcr/luac032. eCollection 2023 Jan.
Primary aldosteronism (PA) is a major cause of secondary hypertension. Aldosterone-producing adenoma (APA) is a subtype of PA, and adrenalectomy is the gold-standard treatment. Recently, a high prevalence of the gene mutation has been reported in APA, particularly in Japan. Herein, we present 2 extremely rare cases of PA recurrence more than 10 years after adrenalectomy for APA. In the first case, a 52-year-old woman was examined for hypertension 22 years after total adrenalectomy of the right adrenal gland. Recurrent PA was diagnosed based on high aldosterone-renin-ratio (ARR), identification of left adrenal gland tumor by computed tomography (CT), and a confirmatory test. In the second case, a 65-year-old man was examined for hypertension 17 years after total adrenalectomy of the left adrenal gland. He had maintained his blood pressure using medication since the onset of hypertension 4 years after the surgery. A year later, a high ARR was observed. PA recurrence was determined by a right adrenal gland tumor noted on CT and a confirmatory test. Somatic mutations in were detected in the resected tissues in both cases. We recommend careful follow-ups after adrenalectomy in APA cases, especially in those with a gene mutation.
原发性醛固酮增多症(PA)是继发性高血压的主要病因。醛固酮瘤(APA)是PA的一种亚型,肾上腺切除术是其金标准治疗方法。最近,有报道称APA中该基因突变的发生率很高,尤其是在日本。在此,我们报告2例极为罕见的病例,患者在因APA接受肾上腺切除术后10多年出现PA复发。第一例中,一名52岁女性在右侧肾上腺全切术后22年因高血压接受检查。根据高醛固酮-肾素比值(ARR)、通过计算机断层扫描(CT)发现左侧肾上腺肿瘤以及确诊试验,诊断为复发性PA。第二例中,一名65岁男性在左侧肾上腺全切术后17年因高血压接受检查。自手术后4年高血压发病以来,他一直通过药物控制血压。一年后,观察到高ARR。通过CT发现右侧肾上腺肿瘤以及确诊试验确定为PA复发。在这两例患者的切除组织中均检测到该基因的体细胞突变。我们建议对APA病例,尤其是那些有该基因突变的患者,在肾上腺切除术后进行仔细随访。