Department of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey.
Department of Pathology, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey.
J Coll Physicians Surg Pak. 2022 Sep;32(9):1222-1224. doi: 10.29271/jcpsp.2022.09.1222.
Lipodystrophy (LD) is an acquired or congenital rare condition consisting of hyperlipidaemia, glucose intolerance/ insulin resistance, and almost complete absence and storage of adipose tissue. Colon perforations can be observed in type 4 congenital LD. Here, we aimed to present a case of sigmoid colon perforation which developed in a young woman with the diagnosis of LD. Extensive purulent peritonitis, significant wall thickening, and oedema in the sigmoid colon were detected during surgical exploration. Anterior resection with end colostomy procedure was then performed. Although bowel perforation has been theoretically reported to occur in LD, the presented case is the first adult patient in the literature. These individuals tend to develop colon perforation as a result of histological changes in their gastrointestinal tract. This situation should always be taken into consideration in order to avoid delay in diagnosis, especially in patients who present with abdominal pain and have a history of LD. Key Words: Intestinal perforation, Congenital lipodystrophy, Peritonitis, Sigmoid colon.
脂肪营养不良(LD)是一种获得性或先天性罕见疾病,表现为高脂血症、葡萄糖耐量/胰岛素抵抗,以及几乎完全缺乏和储存脂肪组织。4 型先天性 LD 可观察到结肠穿孔。在此,我们旨在报告一例在 LD 诊断的年轻女性中发生的乙状结肠穿孔。术中探查发现广泛脓性腹膜炎、乙状结肠明显壁增厚和水肿。然后进行了乙状结肠前切除术加末端结肠造口术。虽然理论上有报道称 LD 会发生肠穿孔,但本例是文献中首例成人患者。这些患者由于胃肠道的组织学变化,易发生结肠穿孔。对于出现腹痛且有 LD 病史的患者,为避免延误诊断,应始终考虑到这种情况。
肠穿孔、先天性脂肪营养不良、腹膜炎、乙状结肠。