Kharbuja Punyaram, Thakur Raghvendra, Suo Jian
Department of Gastrointestinal-Colorectal Surgery, First Hospital of Jilin University, 71th Xin Min Street, Changchun, Jilin 130021, China.
Case Rep Surg. 2013;2013:906457. doi: 10.1155/2013/906457. Epub 2013 Apr 30.
Background. Visceral myopathy is rare pathological condition of gastrointestinal tract with uncertain clinical presentation and unknown etiology. It often presents with symptoms of chronic intestinal pseudoobstruction of colon. We report a case of visceral myopathy which presented to us as acute appendicitis and Ogilvie syndrome, and we managed it surgically. Method and Result. A case report of 20-year female clinically presented as acute appendicitis and we performed laparoscopic exploration which revealed inflamed appendix with grossly dilated ascending colon. We performed laparoscopic appendectomy and postoperatively managed the patients with IV fluids, antibiotics, neostigmine, and extended length rectal tube for enema and decompression. During postoperative period, she developed abdomen distension and peritonitis, and we ordered abdomen CT which revealed colon pseudo- obstruction. We performed right hemicolectomy with permanent ileostomy, and the histopathology reports of resected colon were visceral myopathy. Conclusion. Visceral myopathy is very rare group of disease and poorly understood condition that may present with chronic or acute intestinal pseudo-obstruction and often mimic other more common gastrointestinal disease. VM should be considered as differential diagnosis whenever the patient presents with acute appendicitis, uncharacteristic abdominal symptoms, recurrent attacks of abdominal distention, and pain with no radiological evidence of intestinal obstruction.
背景。内脏肌病是一种罕见的胃肠道病理状况,临床表现不明确,病因不明。它常表现为结肠慢性假性肠梗阻的症状。我们报告一例以内脏肌病表现为急性阑尾炎和奥吉尔维综合征的病例,并对其进行了手术治疗。方法与结果。一名20岁女性的病例报告,临床表现为急性阑尾炎,我们进行了腹腔镜探查,发现阑尾发炎且升结肠明显扩张。我们进行了腹腔镜阑尾切除术,术后通过静脉输液、抗生素、新斯的明以及延长直肠管灌肠和减压对患者进行治疗。术后,她出现腹胀和腹膜炎,我们安排了腹部CT检查,结果显示结肠假性梗阻。我们进行了右半结肠切除术并做了永久性回肠造口术,切除结肠的组织病理学报告显示为内脏肌病。结论。内脏肌病是一组非常罕见且了解甚少的疾病,可能表现为慢性或急性肠假性梗阻,常与其他更常见的胃肠道疾病相似。每当患者出现急性阑尾炎、不典型腹部症状、反复发作的腹胀以及无肠梗阻影像学证据的疼痛时,都应考虑内脏肌病作为鉴别诊断。