Abstract
BackgroundIdiopathic myointimal hyperplasia of the mesenteric vein (IMHMV) is a rare, often undiagnosed pathology affecting the colon. Patients typically present with severe abdominal pain and inflammation caused by smooth muscle proliferation of the veins, leading to arterialization, stenosis, and potential occlusion. The etiology remains unclear, but it has been hypothesized that an arteriovenous connection may be associated with the pathology. This is the first reported case indicating such an association. This case additionally highlights the potential utility of endovascular treatment, as endovascular embolization is generally a less invasive alternative to surgical resection in the treatment of such vascular disorders.Case PresentationThis report describes a 24-year-old female patient with findings of colitis and an abnormal arteriovenous connection of the inferior mesenteric arterial and venous systems. Partial embolization of this arteriovenous connection temporarily improved the patient’s condition, but her symptoms ultimately returned due to the presence of multiple smaller feeder vessels not amenable to embolization, necessitating colonic resection for definitive treatment. Although prior reports have hypothesized that arterial pressurization of the veins may precipitate myointimal hyperplasia, to the authors’ knowledge, this is the first report of IMHMV with an associated abnormal arteriovenous connection.ConclusionsThis case illustrates the possibility of an association between an arteriovenous connection and IMHMV. This rare diagnosis should be considered in patients with a similar presentation of abdominal pain after common etiologies like IBD have been excluded.
Highlights
Idiopathic myointimal hyperplasia of the mesenteric vein (IMHMV) is a rare, often undiagnosed pathology affecting the colon
Prior reports have hypothesized that arterial pressurization of the venous vasculature may cause venous myointimal hyperplasia, but to the authors’ knowledge, (2021) 4:88 there have been no reports of IMHMV in a patient with angiographic evidence of an abnormal arteriovenous connection
Patients present with nonspecific symptoms that mimic inflammatory bowel disease (IBD), including severe abdominal pain, weight loss, rectal bleeding, and diarrhea or constipation (Song and Shroff 2017)
Summary
IMHMV has classically been reported in otherwise healthy young to middle-aged men, recent cases, including this one, have identified the disease in females or older patients (Platz 2012). Prior reports have hypothesized that IMHMV results from an arteriovenous connection that leads to increased pressurization of the veins, resulting in venous arterialization Standard treatment for both IMHMV and AVM is surgical resection of the affected section of bowel. The diagnosis is missed, and surgery is performed after significant morbidity or failure to respond to IBD medical treatments (Bronswijk et al 2019) This case illustrates the possible significance of an arteriovenous connection in the setting of ischemic colitis with a plausible pathogenic relationship with IMHMV. While embolization did not yield long-term benefit in the presented case, further study is needed to investigate the utility of endovascular approaches as a viable treatment alternative to resection for IMHMV with an abnormal arteriovenous connection, when diagnosed early, as endovascular treatment offers significant benefits (Hendy et al 2018). Author details 1Sidney Kimmel Medical College, Thomas Jefferson University, 1025 Walnut St, 19107 Philadelphia, PA, USA. 2Department of Interventional Radiology, Hospital of the University of Pennsylvania, 3400 Spruce St, 19104 Philadelphia, PA, USA
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