Pandey Diwakar, Verma Ankur, Akhtar Azaz, Arsia Ashish, Singh Nain
Post Graduate Student, Department of Surgery, Lady Hardinge Medical College , New Delhi, India .
Senior Resident, Department of Surgery, Lady Hardinge Medical College , New Delhi, India .
J Clin Diagn Res. 2015 May;9(5):XD03-XD04. doi: 10.7860/JCDR/2015/13318.5933. Epub 2015 May 1.
Malignant Peripheral Nerve Sheath Tumours (MPNST) arises from a peripheral nerve or exhibit nerve sheath differentiation on histology. Proximal portions of the upper and lower extremities and the trunk are the most common sites of occurrence. Around 50% are associated with Neurofibromatosis Type 1 (NF1) with incidence of two to five per cent in patients with NF1. The estimated incidence in general population without NF1 is 0.0001% of which gastrointestinal MPNST are extremely rare. A 45-year-old lady without pathological antecedent for NF1 was admitted with pain in right lower abdomen and multiple episodes of vomiting for 3 months. Preoperatively intussusception was diagnosed in the small bowel with USG and CECT abdomen showing characteristic target sign. On laparotomy Ileo-ileal intussusception (proximal ileum telescoping into distal ileum) was found 2 feet proximal to ileo-caecal junction with surrounding inflammed mesentery and presence of intraluminal tumour as lead point. Resection of involved segment of ileum along with its mesentery was done followed by ileo-ileal anastomosis. Histopathology was suggestive of high grade MPNST. Postoperative course and follow up for last 10 month is uneventful. This case is unique in terms of a rare tumour presenting with unusual complication and only one case had been reported so far in western literature.
恶性周围神经鞘膜瘤(MPNST)起源于周围神经,或在组织学上表现出神经鞘分化。上肢、下肢近端以及躯干是最常见的发病部位。约50%的病例与1型神经纤维瘤病(NF1)相关,NF1患者的发病率为2%至5%。在无NF1的普通人群中,估计发病率为0.0001%,其中胃肠道MPNST极为罕见。一位45岁无NF1病史的女性因右下腹痛和多次呕吐3个月入院。术前通过超声和腹部CT检查诊断为小肠套叠,显示特征性的靶征。剖腹探查时,在回盲部交界处近端2英尺处发现回肠-回肠套叠(近端回肠套入远端回肠),伴有周围肠系膜炎症,腔内肿瘤作为套叠头部。切除受累的回肠段及其系膜,随后进行回肠-回肠吻合术。组织病理学提示为高级别MPNST。术后病程及过去10个月的随访情况良好。该病例独特之处在于一种罕见肿瘤伴有不寻常的并发症,西方文献中迄今为止仅报道过一例。