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.

Figure 1

Plain radiography of the abdomen on admission revealing small linear and round radiolucent clusters in the wall of the colon (black arrows).

Figure 2

Computed tomography (CT) scanning of the abdomen on admission revealing intramural gas in the sigmoid colon (white arrows).

Figure 3

Colonoscopy on admission showing multiple round and smooth-surfaced elevated lesions like submucosal tumors in the sigmoid colon (A) and endoscopic ultrasonography (EUS) revealing hyperechoic lesions and acoustic shadows in the submucosal layer (B).