Pneumatosis cystoides intestinalis accompanied by intestinal spirochetosis

Pneumatosis cystoides intestinalis (PCI) is a rare but well-recognized entity characterized by multiple gas-filled cysts in the intestinal wall. Although the pathogenesis of PCI remains unclear, several theories, including a bacterial theory, have been postulated. Intestinal spirochetosis (IS) is an uncommon condition defined by the presence of spirochetes attached to the surface of the colonic epithelium. The nature of IS as a commensal or pathogenic process remains debatable. However, recent evidence supports the idea that IS can be invasive and highly pathogenic in both immunocompromised and immunocompetent individuals. We present the case of a 35-year-old asymptomatic and immunocompetent man who underwent colonoscopy because of a positive fecal blood test. Multiple submucosal cystic lesions were detected accompanied by erythematous areas along the ascending colon. Computed tomography-colonography and biopsy specimens from the erythematous areas confirmed coexisting PCI and IS. Both PCI and IS recovered completely 3 months after administration of metronidazole. To the best of our knowledge, this case represents only the second report of the extremely rare concurrence of PCI with IS. Taking into account the published literature, we also discuss the possibility that the development of PCI may be related to IS.