Department of General Surgery, Boston Children's Hospital, 300 Longwood Avenue, Fegan 3, Boston, MA, 02115, USA.
Department of Pathology, Boston Children's Hospital, Boston, MA, 02115, USA.
BMC Pediatr. 2022 Jan 20;22(1):52. doi: 10.1186/s12887-022-03122-7.
Triple gallbladder is a rare congenital anomaly of the biliary tract that can be associated with heterotopic tissue. Gallbladder triplication results from the failure of rudimentary bile ducts to regress during embryological development, and can be difficult to distinguish from Todani type II choledochal cysts and biliary duplication cysts.
A 2-year-old patient presented to our institution with intermittent abdominal pain for 1 year. She had elevated transaminases with imaging concerning for a choledochal cyst. After assessment with magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography, she was diagnosed with a gallbladder multiplication and a common bile duct stricture. She underwent laparoscopic cholecystectomy, which confirmed the diagnosis of triple gallbladder. One of the three gallbladders demonstrated heterotopic gastric mucosa on final pathology, including at the cystic duct margin. Follow up testing with a technetium 99 m scan demonstrated a subtle focus of increased activity in the right upper abdomen at the expected location of the common bile duct, concerning for the presence of residual gastric mucosa. The patient remains well without abdominal pain.
We describe the first case of heterotopic gastric mucosa in a triple gallbladder in a young patient presenting with chronic abdominal pain. We also demonstrate the safety and feasibility of laparoscopic cholecystectomy in young children with triple gallbladder. Finally, we propose an interdisciplinary approach to the management of common bile duct strictures in the setting of ectopic acid secretion, involving a combination of medical management, endoscopic intervention, and possible salvage laparoscopic Roux-en-Y hepaticojejunostomy.
三胆囊是一种罕见的先天性胆道异常,可伴有异位组织。胆囊三倍体是由于胚胎发育过程中原始胆管退化失败所致,难以与 Todani Ⅱ型胆总管囊肿和胆管重复囊肿相区别。
一名 2 岁患者因间歇性腹痛 1 年来我院就诊。她的转氨酶升高,影像学检查提示胆总管囊肿。经磁共振胰胆管成像和内镜逆行胰胆管造影评估后,诊断为胆囊增殖和胆总管狭窄。她接受了腹腔镜胆囊切除术,证实了三胆囊的诊断。三个胆囊中有一个在最终病理上显示异位胃黏膜,包括胆囊管边缘。锝 99m 扫描的随访检查显示右上腹部在预期的胆总管位置有一个细微的放射性活性增加焦点,提示存在残留的胃黏膜。患者无腹痛,恢复良好。
我们描述了首例在年轻患者中出现慢性腹痛的三胆囊中存在异位胃黏膜的病例。我们还证明了腹腔镜胆囊切除术在患有三胆囊的幼儿中的安全性和可行性。最后,我们提出了一种针对异位酸分泌情况下胆总管狭窄的多学科管理方法,包括药物治疗、内镜干预和可能的挽救性腹腔镜 Roux-en-Y 肝肠吻合术。