Pneumatosis cystoides intestinalis associated with sunitinib and a literature review

Abstract

Background: Pneumatosis cystoides intestinalis (PCI) is a rare self-limiting condition characterized by air-filled cysts within intestinal walls. Diagnosis should be prudent because it can mimic pneumoperitoneum leading to unnecessary treatment such as surgical exploration. Although various drugs including anti-neoplastic agents have been suggested as etiologies, cases related to sunitinib are sparse. Because of the rarity of this unusual side effect by sunitinib, we report the case report.

Case presentation: A 68-year-old female with pancreatic neuroendocrine tumor who was treated with sunitinb for 4 months visited to our hospital complaining of severe diarrhea and mild abdominal discomfort. The abdominal X-ray showed subdiaphragmatic air mimicking intestinal perforation. After the meticulous evaluation including abdomino-pelvic computed tomography, the patient was diagnosed of PCI induced by sunitinib and fully recovered with conservative management.

Conclusions: It is important to note that PCI can develop after treatment with sunitinib because PCI has not been widely known as an adverse event caused by the agent. Furthemore, emergent surgery while sunitinib was administrated without adequate washout period can result in substantial surgical complications which could be avoided with the precise diagnosis.

Keywords: Perforation; Pneumatosis cystoides intestinalis; Sunitinib.

Fig. 1

Erect view of abdominal X-ray at initial presentation

Fig. 2

Abdominal CT at initial presentation. Note the air collection within ileal loops and colonic wall on initial CT (Left). PCI was severe, thus air-containing cysts (arrows and circle) were distributed at both of mesenteric and anti-mesenteric border. On the follow-up CT (Right) taken 1 week later, improving PCI was observed. Cysts at mesenteric border and near mesenteric vessels were predominant (arrows)

Fig. 3

Follow-up abdominal CT after 2 week shows nearly resolved state of PCI