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Case Reports
. 2012 Apr;77(2):60-3.
doi: 10.12659/pjr.882972.

Pneumatosis cystoides interstitialis: A complication of graft-versus-host disease. A report of two cases

Katarzyna Laskowska  1 , Małgorzata Burzyńska-Makuch, Anna Krenska, Sylwia Kołtan, Małgorzata Chrupek, Elżbieta Nawrocka, Władysław Lasek, Zbigniew Serafin
Affiliations
Case Reports

Pneumatosis cystoides interstitialis: A complication of graft-versus-host disease. A report of two cases

Katarzyna Laskowska et al. Pol J Radiol. 2012 Apr.

Abstract

Background: Pneumatosis cystoides intestinalis (PCI) is a rare disorder characterized by the presence of multiple gas collections in the subserosal or submucosal intestinal wall of the large or small intestine. We report two cases of PCI in the course of chronic graft-versus-host disease.

Case report: A 5-year-old girl was treated for acute lymphoblastic leukemia. Twenty-four months after the hematopoietic stem cell transplantation, in the course of graft-versus-host disease, she developed subcutaneous emphysema of the right inguinal and pudendal region. PCI was diagnosed based on a CT examination. A 3-year-old boy was treated for juvenile myelomonocytic leukemia. Fourteen months after the hematopoietic stem cell transplantation he presented with an increased severity of intestinal symptoms, including intermittent bleeding from large intestine. PCI was diagnosed based on a CT exam and was confirmed by a colonoscopy.

Conclusions: Pneumatosis cystoides interstitialis in the course of chronic graft-versus-host disease has a heterogeneous clinical presentation that does not correlate with results of imaging. CT is a method of choice to diagnose PCI. In patients with PCI, the presence of free air in the peritoneal cavity does not confirm an intestinal perforation.

Keywords: computed tomography; graft-versus-host disease; pneumatosis cystoides intestinalis.

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Figures

Figure 1
Figure 1
Patient 1. Numerous air collections and air in the inguinal region and soft tissues of the right pudendal labium visible on a CT scan (arrow) (A). A CT image at the level of mid-abdomen shows multi-cystic air collections in intestinal walls, free air in peritoneal cavity and single air collections in the extraperitoneal space (B). A CT scan of the lesser pelvis reveals air in the wall of ileum (arrow) (C).
Figure 2
Figure 2
Patient 2. In computed tomography air collections are visible in the periintestinal fat (A) and gastrointestinal wall (B).
Figure 3
Figure 3
Patient 2. Endoscopic Picture of PCI with a submucosal air collection (arrow).
See this image and copyright information in PMC

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