Lenzhofer R, Lindner M, Moser A, Berger J, Schuschnigg C, Thurner J
Interne Abteilung, Kardinal Schwarzenberg'sches Krankenhaus Schwarzach.
Clin Investig. 1993 Jul;71(7):568-71. doi: 10.1007/BF00208483.
A 26-year-old patient presented with epigastric pain of sudden onset and severe puffy swelling of both legs and forearms. An irregularly shaped nodular filling defect on selective jejunal films, severe hypoproteinemia, low IgG concentration, and lymphopenia were suggestive of primary intestinal lymphangiectasia with protein-losing enteropathy, and the patient was placed on a low-fat diet with medium-chain triglycerides. This initially improved his condition, but some weeks later he developed obstructive ileus of the small intestine. On laparotomy yellowish to whitish deposits were found to be present in some segments of the small intestine and a fist-sized mass 100 cm distal to the duodenojejunal flexure was resected without complications. Histologically, the submucosal lymphatics were dilated, and the jejunal wall showed extensive pseudocystic, intramural submucosal lymph edema with secondary bleeding and tight stenosis of the jejunal lumen. During the 14-month follow-up time after discharge the patient has been asymptomatic and working, on no treatment other than a low-fat diet with medium-chain triglycerides.
一名26岁患者出现突发上腹部疼痛,双腿和前臂严重肿胀。选择性空肠造影显示不规则形结节状充盈缺损、严重低蛋白血症、低IgG浓度及淋巴细胞减少,提示原发性肠淋巴管扩张症伴蛋白丢失性肠病,患者接受了含中链甘油三酯的低脂饮食。这最初改善了他的病情,但几周后他出现了小肠梗阻。剖腹手术时发现小肠部分节段有淡黄色至白色沉积物,在十二指肠空肠曲远端100 cm处切除了一个拳头大小的肿块,无并发症。组织学检查显示黏膜下淋巴管扩张,空肠壁呈现广泛的假囊肿形成、壁内黏膜下淋巴水肿伴继发性出血及空肠管腔严重狭窄。出院后的14个月随访期间,患者无症状且能正常工作,除了含中链甘油三酯的低脂饮食外未接受其他治疗。