Demartin Sophie, Goffette Pierre, Christ Emanuel, Freitag Martin T, Maiter Dominique, Maria Furnica Raluca
Department of Endocrinology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium.
Department of Radiology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium.
Endocrinol Diabetes Metab Case Rep. 2022 Nov 1;2022. doi: 10.1530/EDM-22-0325.
A 52-year-old female presented with recurrent episodes of fasting or post-absorptive hypoglycemia. A 72-h fasting test confirmed endogenous hyperinsulinemia. Conventional imaging was unremarkable. Selective pancreatic arterial calcium stimulation and hepatic venous sampling showed a maximum calcium-stimulated insulin concentration from several pancreatic areas, mainly the proximal splenic artery and the proximal gastroduodenal artery, suggesting the presence of one or more occult insulinoma(s) in the region of the pancreatic body. 68Ga-DOTA-exendin-4 PET/CT showed however generalized increased uptake in the pancreas and a diagnosis of nesidioblastosis was therefore suspected. The patient has been since successfully treated with dietetic measures and diazoxide. Treatment efficacy was confirmed by a flash glucose monitoring system with a follow-up of 7 months.
Adult nesidioblastosis is a rare cause of endogenous hyperinsulinemic hypoglycemia. The distinction between insulinoma and nesidioblastosis is essential since the therapeutic strategies are different. 68Ga-DOTA-exendin-4 PET/CT emerges as a new noninvasive diagnostic tool for the localization of an endogenous source of hyperinsulinemic hypoglycemia. Medical management with dietetic measures and diazoxide need to be considered as a valuable option to treat patients with adult nesidioblastosis. Flash glucose monitoring system is helpful for the evaluation of treatment efficacy.
一名52岁女性出现反复的空腹或吸收后低血糖发作。72小时禁食试验证实存在内源性高胰岛素血症。传统影像学检查无异常。选择性胰动脉钙刺激和肝静脉采血显示，几个胰腺区域（主要是脾动脉近端和胃十二指肠动脉近端）的钙刺激胰岛素浓度最高，提示胰体区域存在一个或多个隐匿性胰岛素瘤。然而，68Ga-DOTA-艾塞那肽-4 PET/CT显示胰腺普遍摄取增加，因此怀疑为胰岛细胞增殖症。此后，该患者通过饮食措施和二氮嗪成功治疗。通过闪光葡萄糖监测系统进行7个月的随访，证实了治疗效果。
成人胰岛细胞增殖症是内源性高胰岛素血症性低血糖的罕见原因。区分胰岛素瘤和胰岛细胞增殖症至关重要，因为治疗策略不同。68Ga-DOTA-艾塞那肽-4 PET/CT成为一种用于定位内源性高胰岛素血症性低血糖来源的新的非侵入性诊断工具。饮食措施和二氮嗪的药物治疗应被视为治疗成人胰岛细胞增殖症患者的一种有价值的选择。闪光葡萄糖监测系统有助于评估治疗效果。
