Department of Diabetes, Endocrinology and Metabolism and Department of Rheumatology and Clinical Immunology, Gifu University Graduate School of Medicine, Gifu, 501-1194, Japan.
Department of Diabetes and Endocrinology, Gifu Prefectural General Medical Center, Gifu, Japan.
BMC Endocr Disord. 2023 Aug 10;23(1):168. doi: 10.1186/s12902-023-01415-1.
Insulinoma in women during pregnancy and postpartum is very rare; approximately 65% of cases are diagnosed early in pregnancy and ~ 35% immediately after delivery, few being found in middle or late pregnancy, likely due to increased insulin resistance seen after early-stage pregnancy. We successfully treated a case of insulinoma in which severe hypoglycemic coma immediately after delivery occasioned detailed investigation and diagnosis.
Our patient experienced hypoglycemic coma in the 3 month of pregnancy (initially considered due to her hyperemesis gravidarum) that improved spontaneously during the gestational period. No abnormalities of plasma glucose or body weight were found in regular checkups during her pregnancy; however, recurrence of hypoglycemic coma after delivery led us to suspect insulinoma. While contrast enhanced computer tomography and endoscopic ultrasonography (EUS) initially failed to detect a tumor in the pancreas, selective arterial calcium stimulation test revealed an insulin-secreting tumor localized in the pancreatic body. She then underwent spleen-preserving distal pancreatectomy; a 10-mm tumor positive for chromogranin A, synaptophysin and insulin was identified.
Although pregnancy can mask insulinoma-associated symptoms and make diagnosis challenging, hypoglycemic episodes during early pregnancy, which were observed in this case, are suggestive of insulinoma. Importantly, in this case, accurate preoperative localization of the tumor enabled prompt curative surgery after delivery. Thus, clinical vigilance for the occurrence of insulinoma and its localization is appropriate for pregnant women suffering severe hypoglycemia.
妊娠和产后期间的女性胰岛素瘤非常罕见；大约 65%的病例在妊娠早期诊断，约 35%在分娩后立即诊断，少数在妊娠中期或晚期发现，这可能是由于妊娠早期后出现的胰岛素抵抗增加所致。我们成功治疗了一例产后严重低血糖昏迷的胰岛素瘤病例，该病例引起了详细的调查和诊断。
我们的患者在妊娠 3 个月时出现低血糖昏迷（最初认为是由于妊娠剧吐），在妊娠期间自发改善。在妊娠期间的常规检查中，未发现血浆葡萄糖或体重异常；然而，分娩后低血糖昏迷的复发使我们怀疑是胰岛素瘤。虽然增强计算机断层扫描和内镜超声检查（EUS）最初未能在胰腺中检测到肿瘤，但选择性动脉钙刺激试验显示胰腺体部存在胰岛素分泌肿瘤。随后她接受了保脾胰体尾部切除术；发现一个 10 毫米大小的肿瘤，免疫组化染色阳性提示为嗜铬粒蛋白 A、突触素和胰岛素阳性。
尽管妊娠可以掩盖与胰岛素瘤相关的症状并使诊断变得具有挑战性，但在本病例中观察到的妊娠早期的低血糖发作提示可能是胰岛素瘤。重要的是，在这种情况下，肿瘤的准确术前定位使分娩后能够及时进行根治性手术。因此，对于患有严重低血糖的孕妇，应保持对胰岛素瘤发生和定位的临床警惕。
