AT THE FOCAL POINT Lawrence J. Brandt, MD, Associate Editor for Focal Points

Primary pneumatosis cystoides intestinalis (with video)

A 58-year-old man was referred for screening colonoscopy because of a family history of colon cancer. He did not describe any symptoms other than intestinal meteorism. At the first colonoscopy, multiple “sessile polyps” were seen in the sigmoid colon. Repeat colonoscopy showed that the previously seen “polyps” were, in fact, sessile, ball-shaped subepithelial nodules that were clustered in streaks (A), mainly in the left side of the colon, and suggestive of pneumatosis cystoides intestinalis. Some of the nodules were covered with a reddish mucosa (B), but the mucosa overlying most of the nodules was normal. Biopsies were performed with the intention of confirming the nature of the cystic lesions (C), but the result of histologic examination was nonspecific. Some lesions deflated after being punctured by a needle, with resolution of the cystic cavities (Video 1, available online at www.giejournal.org).

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Abdominopelvic CT confirmed the presence of air “bubbles” in the intestinal wall (D). No other abnormalities were identified. No underlying disease was found, and the patient remains asymptomatic. DISCLOSURE All authors disclosed no financial relationships relevant to this publication. Eduardo Rodrigues-Pinto, MD, Pedro Pereira, MD, Guilherme Macedo, MD, PhD, Gastroenterology Department, Centro Hospitalar São João, Porto, Portugal http://dx.doi.org/10.1016/j.gie.2014.01.027

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At the Focal Point

Commentary Pneumatosis intestinalis comes in two sorts: cystoides (PC) and linearis. I’d rather have the former because the latter most often is seen in conditions associated with intestinal necrosis, and its presence usually mandates surgery. This patient was asymptomatic, except for meteorism (gaseous drumlike distention of the abdomen), and the condition was diagnosed serendipitously. In other patients, and depending on the location of the cysts, constipation, diarrhea, abdominal pain, hematochezia, mucorrhea, and urgency may prompt medical attention. Hematochezia is from ulceration of the mucosa overlying the cysts, whereas obstructive symptoms may be caused by cyst encroachment on the lumen, volvulus as the “lightness of being” causes the bowel to float and twist, or by adhesions formed as the cysts collapse. Benign pneumoperitoneum also may occur when subserosal cysts rupture; such rupture does not cause peritonitis and does not mandate surgery. In PC, thin-walled, endothelial-lined cysts may be located in the mucosa, submucosa, or serosa. Typically, the endothelial lining collapses, forming multinucleated giant cells; the cyst undergoes fibrosis and is eventually sloughed. The connective tissue surrounding the cysts may show a granulomatous inflammatory reaction made up of a variety of cells, including eosinophils, lymphocytes, plasma cells, and macrophages. Mucosal histology may show mild focal nonspecific abnormalities, as in this patient, to substantial changes including granulomas, abnormal crypt branching, cryptitis, and crypt abscesses. Although PC in this patient was first misidentified as polyps, a more experienced eye made the diagnosis by colonoscopy and confirmed it with CT. The presence of bluish or reddish thin walled–appearing nodules evokes few diagnostic possibilities: colon ischemia with subepithelial hemorrhage, varices, perhaps varicoid carcinoma or melanoma, arteriovenous malformation, and the blue rubber bleb syndrome. An injection needle or biopsy forceps can be used to puncture the cyst and diagnose the nature of the target by provoking release of “air” and collapse of the cyst; actually, these are not air-filled but rather contain a mixture of gases including nitrogen, oxygen, carbon dioxide, and up to 25% hydrogen. Why should an asymptomatic and apparently healthy person have pneumatosis? Recognizing that the subtlety of nature is many times over our understanding (Francis Bacon), I advise a careful search for chronic pulmonary and collagen vascular disease, sigmoid volvulus, and rectal prolapse. Idiopathic is idiopathic only if one has exhausted all diagnostic considerations. Lawrence J. Brandt, MD Associate Editor for Focal Points

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Primary pneumatosis cystoides intestinalis (with video).

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