Abdul-Hafez Hamza A, Rabi Karam, Sarama Asmaa, Melhem Layan, Abed Waddah, Maree Mohammed
Department of Medicine, Faculty of Medicine and Health Sciences, An-Najah National University, Nablus, Palestine.
Department of General Surgery, Ibn Sina Specialized Hospital, Jenin, Palestine.
Ann Med Surg (Lond). 2025 Mar 28;87(5):3006-3011. doi: 10.1097/MS9.0000000000003198. eCollection 2025 May.
Adult-onset nesidioblastosis is an exceedingly rare yet significant cause of persistent hyperinsulinemic hypoglycemia. This condition is often associated to bariatric surgeries such as Roux-en-Y gastric bypass and sleeve gastrectomy. Characterized by abnormal β-cell hyperplasia and hypertrophy, its diagnosis presents a unique challenge due to overlapping features with insulinomas and post-bariatric hypoglycemia syndrome (PBHS).
We report a 55-year-old woman with a history of gastric sleeve and Roux-en-Y gastric bypass surgeries who presented with a 1.5-year history of recurrent hypoglycemic episodes. Her symptoms, including blurred vision, tremors, and altered consciousness, persisted despite medical therapy with octreotide, acarbose, and nifedipine. Extensive imaging, including magnetic resonance imaging and endoscopic ultrasound, ruled out insulinomas, raising suspicion of non-insulinoma pancreatogenous hypoglycemia syndrome. The patient underwent laparoscopic subtotal distal pancreatectomy with spleen preservation. Histopathological examination confirmed nesidioblastosis, revealing irregular islet distribution and β-cell hypertrophy. Post-surgery, the patient achieved normoglycemia without recurrence of hypoglycemic episodes during follow-up.
This case highlights the complexity of diagnosing nesidioblastosis in adults, especially following bariatric surgeries. Nesidioblastosis involves β-cell hyperplasia and hypertrophy driven by hormonal factors like GLP-1, whereas PBHS results from altered incretin patterns causing excessive insulin release. Advanced imaging and multidisciplinary collaboration are important for accurate diagnosis. Surgical management remains a cornerstone for refractory cases, as demonstrated in this patient's remarkable recovery.
Nesidioblastosis is a rare condition but should be considered in the differential diagnosis of post-bariatric surgery hypoglycemia. This case highlights the importance of distinguishing nesidioblastosis from PBHS to ensure appropriate and effective management strategies.
成人隐匿性自身免疫性糖尿病是持续性高胰岛素血症性低血糖症极为罕见但重要的病因。这种情况常与减肥手术相关,如Roux-en-Y胃旁路手术和袖状胃切除术。其特征为β细胞异常增生和肥大,由于与胰岛素瘤及减肥后低血糖综合征(PBHS)有重叠特征,其诊断面临独特挑战。
我们报告一名55岁女性,有袖状胃切除术和Roux-en-Y胃旁路手术史,出现复发性低血糖发作1.5年。尽管使用奥曲肽、阿卡波糖和硝苯地平进行药物治疗,她的症状,包括视力模糊、震颤和意识改变仍持续存在。包括磁共振成像和内镜超声在内的广泛影像学检查排除了胰岛素瘤,怀疑为非胰岛素瘤性胰源性低血糖综合征。该患者接受了保留脾脏的腹腔镜远端胰腺次全切除术。组织病理学检查确诊为成人隐匿性自身免疫性糖尿病,显示胰岛分布不规则和β细胞肥大。术后,患者血糖恢复正常,随访期间低血糖发作未复发。
该病例突出了成人隐匿性自身免疫性糖尿病诊断的复杂性,尤其是在减肥手术后。成人隐匿性自身免疫性糖尿病涉及由GLP-1等激素因素驱动的β细胞增生和肥大,而PBHS是由肠促胰岛素模式改变导致胰岛素过度释放引起的。先进的影像学检查和多学科协作对于准确诊断很重要。手术治疗仍然是难治性病例的基石,如该患者的显著康复所示。
成人隐匿性自身免疫性糖尿病是一种罕见疾病,但在减肥手术后低血糖的鉴别诊断中应予以考虑。该病例突出了区分成人隐匿性自身免疫性糖尿病与PBHS以确保采取适当有效管理策略的重要性。