Babaya Naru, Okuda Yuki, Noso Shinsuke, Hiromine Yoshihisa, Taketomo Yasunori, Niwano Fumimaru, Ueda Kazuki, Tanaka Yumiko, Yamazaki Yuto, Sasano Hironobu, Kawabata Yumiko, Ohno Yasuhiro, Ikegami Hiroshi
Department of Endocrinology, Metabolism and Diabetes, Kindai University Faculty of Medicine, Osaka, Japan.
Department of Surgery, Kindai University Faculty of Medicine, Osaka, Japan.
J Endocr Soc. 2020 Nov 27;5(2):bvaa184. doi: 10.1210/jendso/bvaa184. eCollection 2021 Feb 1.
Characterization of adrenocortical disorders is challenging because of varying origins, laterality, the presence or absence of hormone production, and unclarity about the benign or malignant nature of the lesion. Histopathological examination in conjunction with immunohistochemistry is generally considered mandatory in this characterization. We report a rare case of bilateral adrenocortical adenomas associated with unilateral adrenal endothelial cysts in a 65-year-old woman whose condition was not diagnosed before surgery. Detailed histological examination of the resected adrenal glands revealed hyperplasia in the zona glomerulosa. Despite hyperplasia, the patient had normal serum aldosterone levels and renin activity without clinical evidence of hypertension. The patient was treated with a sodium-glucose cotransporter protein 2 (SGLT2) inhibitor. This may have stimulated the renin-angiotensin-aldosterone system. To the best of our knowledge, this is the first case in which both relatively large bilateral adrenocortical adenomas and unilateral adrenal endothelial cysts were detected. This case also highlights the complexity and difficulty of preoperative diagnosis. Furthermore, this case reports the first detailed histopathological examination of adrenal lesions with SGLT2 treatment and the possibility of SGLT2 inhibitor treatment resulting in histological hyperplasia in the zona glomerulosa; however, it is difficult to prove a causative relationship between SGLT2 inhibitors and hyperplasia of the zona glomerulosa based on the data of this case. It can be confirmed only under limited conditions; therefore, further studies on adrenal gland histology employing SGLT2 inhibition are warranted.
肾上腺皮质疾病的特征描述具有挑战性，这是因为其起源、单侧性、激素分泌情况各异，且病变的良恶性性质不明。在这种特征描述中，组织病理学检查结合免疫组织化学通常被认为是必不可少的。我们报告了一例罕见病例，一名65岁女性患有双侧肾上腺皮质腺瘤并伴有单侧肾上腺内皮囊肿，其病情在手术前未被诊断出来。对切除的肾上腺进行详细的组织学检查发现球状带增生。尽管有增生，但患者血清醛固酮水平和肾素活性正常，且无高血压的临床证据。该患者接受了钠-葡萄糖协同转运蛋白2（SGLT2）抑制剂治疗。这可能刺激了肾素-血管紧张素-醛固酮系统。据我们所知，这是首例同时检测到相对较大的双侧肾上腺皮质腺瘤和单侧肾上腺内皮囊肿的病例。该病例还凸显了术前诊断的复杂性和困难。此外，本病例首次报告了对接受SGLT2治疗的肾上腺病变进行详细的组织病理学检查，以及SGLT2抑制剂治疗导致球状带组织学增生的可能性；然而，根据本病例的数据难以证明SGLT2抑制剂与球状带增生之间存在因果关系。只有在有限的条件下才能得到证实；因此，有必要对采用SGLT2抑制的肾上腺组织学进行进一步研究。
