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新生儿舌和胃肠道毛霉病一例——低位肛门直肠畸形的罕见表现。

Neonatal lingual and gastrointestinal mucormycosis in a case of low anorectal malformation-a rare presentation.

机构信息

Department of Pediatric Surgery, King Edward Memorial Hospital, Mumbai, India.

Department of Pediatric Surgery, King Edward Memorial Hospital, Mumbai, India.

出版信息

J Pediatr Surg. 2011 Apr;46(4):745-748. doi: 10.1016/j.jpedsurg.2010.12.024.

Abstract

We report a 1.7 kg male infant with a low anorectal malformation treated at an outside facility and referred to us on postoperative day 11. At presentation, his upper abdomen was distended, and he had perianal mucoid discharge. The tongue had a blackish discoloration. An erect abdominal radiograph showed a few fluid-filled bowel loops in the upper abdomen with a gasless lower abdomen and pelvis, suggestive of upper small bowel obstruction. There were no specific radiological features of necrotizing enterocolitis. He underwent laparotomy and bowel resection for perforated jejunum. Histopathology of the tissue specimen was suggestive of mucormycosis. Postoperatively, he received intravenous amphotericin B (liposomal) and was started on liquid enteral nutrition after 2 weeks. However, the anterior two thirds of his tongue gradually sloughed off. He is awaiting reconstruction of the tongue. The purpose of this report is to emphasize that physicians should have a high index of suspicion for oral and gastrointestinal tract mucormycosis in neonates with metabolic disturbances who present with a discolored oral mucosa and an abdominal mass with intestinal obstruction. Early diagnosis and an aggressive approach of combined medical and surgical treatment may improve the outcome of patients with this potentially lethal invasive disease.

摘要

我们报告了一例 1.7 公斤男性婴儿,患有低位肛门直肠畸形,在外部医疗机构接受治疗,并在术后第 11 天转至我们医院。就诊时,他的上腹部膨隆,有肛周黏液性分泌物。舌呈黑褐色变色。直立腹部 X 光片显示上腹部有几个充满液体的肠袢,下腹部和骨盆无气,提示上小肠梗阻。没有坏死性小肠结肠炎的特定放射学特征。他因穿孔空肠而行剖腹手术和肠切除术。组织标本的组织病理学检查提示为毛霉菌病。术后,他接受了静脉注射两性霉素 B(脂质体)治疗,并在 2 周后开始进行肠内液体营养。然而,他的舌头前 2/3 逐渐脱落。他正在等待舌头重建。本报告的目的是强调,对于代谢紊乱、口腔黏膜变色和肠梗阻伴腹部肿块的新生儿,医生应高度怀疑口腔和胃肠道毛霉菌病。早期诊断和积极的联合药物和手术治疗方法可能会改善这种潜在致命性侵袭性疾病患者的预后。

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