Pneumatosis cystoides intestinalis following alpha-glucosidase inhibitor treatment: A case report and review of the literature

Abstract

A 69-year-old man was diagnosed as having myasthenia gravis (MG) in September 2004, and treated with thymectomy and prednisolone. He was then diagnosed as having steroid-induced diabetes mellitus, and received sulfonylurea (SU) therapy in May 2005. An alpha-glucosidase inhibitor (alphaGI) was added in March 2006, resulting in good glycemic control. He experienced symptoms of abdominal distention, increased flatus, and constipation in October 2007, and was admitted into our hospital in late November with hematochezia. Plain abdominal radiography revealed small linear radiolucent clusters in the wall of the colon. Computed tomography (CT) showed intramural air in the sigmoid colon. Colonoscopy revealed multiple smooth surfaced hemispherical protrusions in the sigmoid colon. The diagnosis of pneumatosis cystoides intestinalis (PCI) was made on the basis of these findings. As the alphaGI voglibose was suspected as the cause of this patient's PCI, treatment was conservative, ceasing voglibose, with fasting and fluid supplementation. The patient progressed well, and was discharged 2 wk later. Recently, several reports of PCI associated with alphaGI therapy have been published, predominantly in Japan where alphaGIs are commonly used. If the use of alphaGIs becomes more widespread, we can expect more reports of this condition on a global scale. The possibility of PCI should be considered in diabetic patients complaining of gastrointestinal symptoms, and the gastrointestinal tract should be thoroughly investigated in these patients.

Pneumatosis Cystoides Intestinalis Induced by the Alpha-glucosidase Inhibitor Miglitol

“…In an earlier case report and literature review, we analyzed the radiological findings of the previously reported cases (9). Plain abdominal radiography demonstrated linear radiolucent gas collections along the bowel wall in most cases, and abdominal CT scanning demonstrated pneumatosis within or along the bowel wall.…”

“…In recent years, cases of PCI induced by alpha-glucosidase inhibitors (αGIs), a new class of antidiabetic agents, have been reported. Our PubMed search yielded only 9 cases of PCI associated with αGI therapy (2)(3)(4)(5)(6)(7)(8)(9)(10). Of the different αGIs, reports of PCI induced by voglibose and acarbose have been published, but this is the first report of miglitol causing PCI.…”

Segmental pneumatosis cystoides coli: Computed tomography-facilitated diagnosis

“…On the other hand, causative treatment, conservative or surgical if necessary, is recommended in symptomatic patients and involves therapeutic means tailored according to PCC etiology, including oxygen hyperbaric therapy or antibiotics in individual cases (17,18). Review of the literature shows that in case of α-glucosidase inhibitor-related PCC, discontinuation of the drug is the treatment of choice that usually leads to the complete recovery within 5 to 28 days (6,19). Such therapeutic option proved to be successful also in the case of the patient described.…”

A Rare Case of Pneumatosis Cystoides Intestinalis After Appendicectomy

“…Conservative methods include hyperbaric oxygen as routinely used. 12,13 For secondary pneumatosis cystoides intestinalis with or without complication, surgery is indicated. 11 Diagnosing rare benign conditions can be difficult, especially where bowel cancer is part of the differential diagnosis.…”