Sclerosing mesenteritis successfully treated with a TNF antagonist

Abstract

SummaryA 29-year-old female presented with intermittent nausea, vomiting, fevers, abdominal pain and fatigue. CT scans of the abdomen revealed inflammatory changes within the mesentery and small bowel. Histopathology of the mesentery and omentum showed chronic inflammation and fibrosis, supporting a diagnosis of sclerosing mesenteritis. Over the past 2 years, the patient suffered debilitating paroxysmal abdominal pain despite treatment with prednisone, azathioprine, sulfasalazine and narcotics. Additionally, she developed sacroiliitis diagnosed clinically and on radiographs. Intravenous infliximab (5 mg/kg intravenous) was initiated and continued every 6 weeks for 3 years. The patient has since had a dramatic improvement in her back and abdominal symptoms and has tapered off of prednisone, azathioprine and narcotics. Erythrocyte sedimentation rate, anaemia, leukocytosis and radiographic findings improved after initiation with infliximab. In conclusion, the authors report successfully treating sclerosing mesenteritis with sacroiliitis by the addition of infliximab. This may implicate a role for tumour necrosis factor α in disease pathogenesis. BACKGROUND

Debilitating Manifestation of a Disease with Multiple Names: A Severe Case of Sclerosing Mesenteritis

“…There are a few case reports written about successful SM treatment with TNF-alpha inhibitors, such as infliximab and adalimumab. Rothlein et al described a case report about a patient with sacroiliitis and similar presentation of SM with debilitating paroxysmal abdominal pain, who was successfully treated with significant improvement in abdominal symptoms with infliximab treatment [ 10 ]. In another study, Nyberg et al presented 27 patients with SM.…”

Sclerosing Mesenteritis in a Patient Heterozygous for Factor V Leiden

“…In a systematic review of sclerosing mesenteritis by Yale New-Haven Hospital’s Dr. Prabin Sharma in 2017, 192 cases from the literature were examined to gain a better understanding of sclerosing mesenteritis’s epidemiology, risk factors, diagnostic methods, and other parameters [ 6 ]. Among these 192 cases and others presented in the literature, only one prior case of sclerosing mesenteritis in a patient heterozygous for FV Leiden has been described, in a 29-year-old female patient in 2010 [ 3 ]. In that case, the patient had a history of cholecystectomy within a year of presentation, which is described in the report as a possible contributing factor to her presentation.…”

“…Eventually, the chronic fibrosis of his mesentery resulted in a small bowel obstruction, necessitating the extensive surgical resection performed in this case. Especially considering this patient’s virgin abdomen, this case serves to update the literature as a comparison to the patient in the 2010 case, in which the patient had a prior abdominal surgery in addition to FV Leiden [ 3 ].…”

“…Any of these forms of sclerosing mesenteritis can potentially have a mass effect that impedes the integrity of the gastrointestinal lumen and mesenteric vessels. However, only one case report, from 2010, discussed chronic thrombotic activity in the mesentery as a risk factor for the development of sclerosing mesenteritis, as was seen in the present case [ 3 ].…”

“…As the etiology of sclerosing mesenteritis appears to be broad and multifactorial based on literature review, we present this case of sclerosing mesenteritis in a hypercoagulable patient to support chronic thrombotic activity as a viable pathogenic mechanism. This case also serves to update the literature at large, as only one prior case in a patient heterozygous for FV Leiden has been described, which occurred in a 29-year-old woman in 2010 [ 3 ].…”