Reddington Hayley, Ballinger Zachary, Abghari Michelle, Modukuru Venkat, Wallack Marc
School of Medicine, New York Medical College, Valhalla, NY, USA.
Department of Surgery, Metropolitan Hospital, New York City, NY, USA.
Am J Case Rep. 2020 Oct 5;21:e926332. doi: 10.12659/AJCR.926332.
BACKGROUND Sclerosing mesenteritis is an inflammatory and fibrotic disease that affects the mesentery of the small intestine. This condition is non-neoplastic, although it is frequently associated with underlying malignancies. The overall etiology is unclear because of the limited number of cases available for review, yet a number of possible mechanisms have been described, including ischemia. Factor V (FV) Leiden is a hereditary condition causing hypercoagulability, thrombosis, and ischemia. Because ischemia is one of the proposed mechanisms for the fibrosis and sclerotic findings of sclerosing mesenteritis, this case explores a possible association between FV Leiden and sclerosing mesenteritis. CASE REPORT Herein, we describe a case of sclerosing mesenteritis in a patient heterozygous for FV Leiden, with a strong personal and family history of venous thromboembolism. This patient presented with acute worsening of chronic abdominal pain and was found to have a small bowel obstruction requiring acute surgical intervention. Imaging findings and pathologic examination of the ileum and mesentery conclusively diagnosed sclerosing mesenteritis. CONCLUSIONS This case serves to highlight a possible association between mesenteric ischemia secondary to chronic thrombotic activity and sclerosing mesenteritis. This patient's virgin abdomen and lack of additional risk factors for sclerosing mesenteritis make this case a unique presentation of the disorder. This case serves to update the literature at large, as only one prior case in a FV Leiden patient has been described, in which the patient had the additional risk factor of previous abdominal surgery.
硬化性肠系膜炎是一种影响小肠系膜的炎症性和纤维化疾病。这种情况是非肿瘤性的,尽管它经常与潜在的恶性肿瘤相关。由于可供审查的病例数量有限,总体病因尚不清楚,但已经描述了一些可能的机制,包括缺血。因子V(FV)莱顿是一种导致高凝状态、血栓形成和缺血的遗传性疾病。由于缺血是硬化性肠系膜炎纤维化和硬化表现的提出机制之一,本病例探讨了FV莱顿与硬化性肠系膜炎之间的可能关联。病例报告:在此,我们描述了一例FV莱顿杂合子患者的硬化性肠系膜炎病例,该患者有强烈的个人和家族静脉血栓栓塞病史。该患者出现慢性腹痛急性加重,被发现患有小肠梗阻,需要进行急性手术干预。回肠和肠系膜的影像学检查结果及病理检查确诊为硬化性肠系膜炎。结论:本病例旨在强调慢性血栓形成活动继发的肠系膜缺血与硬化性肠系膜炎之间的可能关联。该患者未做过腹部手术且缺乏硬化性肠系膜炎的其他危险因素,使得该病例成为该疾病的独特表现。本病例有助于更新整个文献,因为之前仅描述过一例FV莱顿患者的病例,该患者有腹部手术史这一额外危险因素。