Extrahepatic bile duct fibroma in an infant: a case report

IF 0.4 Q4 PEDIATRICS Annals of Pediatric Surgery Pub Date : 2023-11-16 DOI:10.1186/s43159-023-00272-w
Md. Samiul Hasan, S. M. Nazmul Islam, Umama Huq, Mashud Parvez, Hasiba Mahshed Khan, Refoyez Mahmud, Ashrarur Rahman
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Abstract

Extrahepatic bile duct tumor in neonates and infants is extremely rare. Fibroma in neonates and infants has been reported in different body parts but not in the bile duct. We report a 2-month-old girl with common bile duct (CBD) fibroma. A 2-month-old girl presented with jaundice and pale stool for 15 days. Her perinatal period was uneventful. On examination, we found the girl deeply icteric and having hepatomegaly but no palpable lump. Laboratory investigations revealed direct hyperbilirubinemia with elevated liver enzymes. Ultrasonography revealed hepatomegaly with focal dilatation of the extrahepatic bile duct (2.1 × 0.5 cm). Our preliminary diagnosis was a type I choledochal cyst. Considering the clinical scenario, we planned for exploration without further imaging. On exploration, we found the liver blackish and a hard mass involving the common CBD and cystic duct. The gall bladder was filled with clear mucus. After excising the mass, the gall bladder, and CBD, we reconstructed the anatomy with Roux-N-Y hepaticojejunostomy. The postoperative period was uneventful except for minor bile leakage during the first four postoperative days, which stopped spontaneously. Histopathology revealed spindle-shaped fibrous tissue admixed with collagenous tissue, compatible with a fibroma. A liver biopsy showed the features of cirrhosis. However, liver function tests were normal 2 weeks after surgery. The baby was thriving at 6 months of follow-up, hepatomegaly was resolved, and there was no clinical or radiological sign of recurrence. CBD fibroma is a rare cause of obstructive jaundice in neonates and infants. The earliest possible excision of the tumor and bile drainage restoration are necessary to halt the progression of liver damage.
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婴儿肝外胆管纤维瘤1例报告
肝外胆管肿瘤在新生儿和婴儿中极为罕见。纤维瘤在新生儿和婴儿已报道在不同的身体部位,但没有在胆管。我们报告一个2个月大的女孩与胆总管(CBD)纤维瘤。2个月大的女婴黄疸,大便苍白15天。她的围产期平安无事。在检查中,我们发现女孩深度黄疸和肝肿大,但没有可触及的肿块。实验室调查显示直接高胆红素血症伴肝酶升高。超声示肝肿大伴肝外胆管局灶性扩张(2.1 × 0.5 cm)。初步诊断为I型胆总管囊肿。考虑到临床情况,我们计划不做进一步的影像学检查。在检查中,我们发现肝脏发黑,硬块累及总CBD和囊管。胆囊充满了透明的粘液。在切除肿物、胆囊和CBD后,我们采用Roux-N-Y肝空肠吻合术重建解剖结构。除术后4天发生轻微胆漏外,术后无大碍,胆漏自行停止。组织病理学显示纺锤形纤维组织与胶原组织混合,与纤维瘤相容。肝活检显示肝硬化的特征。术后2周肝功能检查正常。随访6个月时,婴儿发育良好,肝肿大消退,无临床或放射学复发迹象。CBD纤维瘤是一种罕见的原因梗阻性黄疸在新生儿和婴儿。尽早切除肿瘤和恢复胆汁引流对于阻止肝损伤的进展是必要的。
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来源期刊
CiteScore
0.60
自引率
0.00%
发文量
74
审稿时长
9 weeks
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