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Pneumatosis cystoides intestinalis is a rare disease characterized by the presence of multiple intramural gas-filled cysts in the gastrointestinal tract. The etiology remains unknown, but the disease can present with profound disturbances of bowel function. We report the successful management of pneumatosis cystoides intestinalis of the small intestine with the use of hyperbaric oxygen.
The authors report a case of pneumatosis cystoides intestinalis which, after failure of medical treatment, was treated by laparoscopic partial colectomy. Surgical treatment is a last resort and the laparoscopically-assisted approach seems to be a good indication in colectomy for pneumatosis cystoides intestinalis.
A case of pneumatosis cystoides intestinalis with pneumoperitoneum and volvulus complications is reported. After careful examination of the literature, etiopathogenic, pathophysiological and clinical features of the disease are analyzed by the Authors, also in the light of recent findings. Major diagnostic and therapeutic implications are discussed regarding possible complications requiring a different therapeutic approach. PCI, a lesion which can be benign, primary or secondary in many conditions, may be characterized by the presence of spontaneous pneumoperitoneum which only requires a careful, but not aggressive therapy, unlike other complications such as volvulus or intestinal occlusion which, instead, require timely surgical action. The Authors describing the therapy adopted, highlight the positive role of the enteral nutrition (NE) and oxygen therapy in the clinical course of the disease.
Pneumatosis cystoides intestinalis (PCI) is a rare disorder of the medical management and clinical outcome of which largely depend on the severity of its clinical manifestation. A limited number of cases have been reported in the literature although it is suggested that the true incidence of this disorder is higher than observed in clinical practice. This is the case of a 76-year-old woman with a past medical history of Crohn's disease found to have PCI. The patient initially complained of abdominal pain, distention, and weight loss. Chest and abdominal radiographs demonstrated free intraperitoneal air. CT scans revealed characteristic air-filled cysts in the intestinal wall, which established the diagnosis of PCI. Because the patient did not have an acute abdomen or findings requiring emergency laparotomy she was treated nonoperatively with supportive care. Her symptoms resolved gradually over several days. The patient was discharged home in stable condition tolerating a regular diet and was doing well at follow-up. The sole finding of free air with PCI does not mandate exploratory laparotomy.
Pneumatosis cystoides intestinalis (PCI) is a rare condition characterized by the presence of gas-filled cysts in the submucosa or subserosa of gastrointestinal tract. PCI has been widely recognized as a late manifestation of systemic sclerosis but seldom reported to take place in patients with systemic lupus erythematosus (SLE). We reported here a 13-year-old female who had been diagnosed to have SLE based on the following findings; malar rash, discoid erythema, proteinuria, positive antinuclear antibody and anti-DNA antibody. She had been treated with various immunosuppressive drugs including pulse use of corticosteroid, cyclophosphamide and cyclosporin A. She was referred to our hospital because of proteinuria and numbness on her right fifth toe, refractory to above treatment. On admission, the activity of her disease was already low and she had no abdominal symptoms. Plain X-ray film showed multiple round translucencies along the wall of the ascending and transverse colon. Colonoscopy revealed multiple firm-walled cysts distributing in the terminal ileum as well. A diagnosis of PCI was made and she was successfully treated with oral antibiotics and laxatives. The association of PCI with SLE is reviewed briefly.
Pneumatosis cystoides intestinalis (PCI) is still a poorly understood phenomenon, currently considered to result from primary mucosal insult from varying causes. We report a case of severe PCI in a patient with chronic GVHD after bone marrow transplantation (BMT) performed to treat secondary AML. Post BMT, the patient suffered acute intestinal and cutaneous GVHD, eventually developing intestinal and biopsy-proven cutaneous chronic GVHD, which necessitated continuous steroid therapy. Chronic pancreatitis associated with GVHD was diagnosed by explorative surgery in February 2000 on the basis of increasing epigastric discomfort, tumour marker (CA 125) increase and the CT finding of a suspicious mass in the pancreas. Readmission occurred in April 2000 for rapid onset of inferior abdominal pain with distinct peritoneal signs. Relaparotomy, deemed necessary on the grounds of both clinical and radiological findings, revealed marked PCI of the ascending and transverse colon and attached mesentery in an otherwise intact gastrointestinal tract. Post-operative reconvalescence was uneventful, with no clinical or radiological recurrence of PCI in the following 10 months. In the context of a review of the relevant literature, this case report illustrates the complex underlying pathophysiology, and difficulty in making a differential diagnosis and treating PCI.
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A case of a woman with spontaneous pneumoperitoneum as result of pneumatosis cystoides intestinalis is reported. A brief review of this rare disease and its pathogenetic mechanisms is presented. The clinical course of the pneumatosis cystoides intestinalis was characterized by abdominal pain and distention, vomiting. The disease was proved by contrast X-ray examination. The pneumoperitoneum was the result of subserous cyst rupture and had a benign course. It was treated successfully with chemotherapeutics. The participation of extensive bacterial growth in the pathogenesis of the pneumoperitoneum is discussed.
Four cases of pneumatosis cystoides intestinalis following ileojejunal by-pass in 37 patients are described and the literature reviewed. This complication is usually a self-limited condition, responding to conservative management.
A patient with antinuclear antibody (ANA) negative systemic lupus erythematosus (SLE) developed pneumatosis cystoides intestinalis and spontaneous pneumoperitoneum. The literature of pneumatosis cystoides intestinalis and spontaneous pneumoperitoneum is reviewed, the benignancy of these conditions is emphasized, and particular attention directed to their association with and implications in the collagen vascular diseases. This is the first reported case of pneumatosis cystoides intestinalis and pneumoperitoneum in a patient with ANA negative SLE.
The use of hyperbaric oxygen for the treatment of pneumatosis cystoides intestinalis is reported. The pathophysiology, etiology and previous treatment reports are discussed, as are the advantages of hyperbaric oxygen therapy over normobaric oxygen therapy or surgery. The use of hyperbaric oxygen appears to represent a significant advance in the treatment of pneumatosis cystoides intestinalis.
A case of fatal air embolism caused by pneumatosis cystoides intestinalis is presented. To our knowledge, there has been no previous report of fatal air embolism associated with this disease. The underlying pathology was not known premortem and the finding of air both in the mesenteric venous bed and in the right side of the heart is consistent with a collateralizing phenomenon due to portal vein obstruction.
Intestinal pseudoobstruction and pneumatosis cystoides intestinalis are uncommon complications of progressive systemic sclerosis. We report a 26-year-old woman with this disorder who responded poorly to conventional treatment. Subcutaneous administration of octreotide, a long-acting somatostatin analogue, at a dose of 50 micrograms/day for 3 weeks, relieved symptoms such as nausea and bloating. There was also a marked decrease of intestinal gas accumulation, as documented on X-ray films.
Pneumatosis cystoides intestinalis (PCI), a disorder in which gas filled cysts develop within the bowel wall, has been associated with collagen vascular disease, usually progressive systemic sclerosis (PSS). We have extended the spectrum of collagen vascular disease associated with PCI by this report of a patient with mixed connective tissue disease (MCTD) who developed PCI and recurrent spontaneous pneumoperitoneum (SP). Possible pathogenetic mechanisms are reviewed and the management of the asymptomatic patient with pneumoperitoneum, PCI and collagen vascular disease is discussed.
A rare association of pneumoperitoneum induced by pneumatosis cystoides intestinalis with chronic mesenteroaxial volvulus of an irreducible stomach in a paraesophageal hiatus hernia is described. The importance of accurate radiologic work-up is emphasized. The possible significance of this association to explain the pathogenesis of pneumatosis cystoides intestinalis is discussed.
A case of chronic pneumatosis cystoides intestinalis, complicating jejunoileal by-pass procedure for morbid obesity, is described. Grave clinical symptoms associated with irreversible segmental mucosal atrophy and mural fibrosis resulting from untreated long-standing pneumatosis intestinalis, required resection of the whole by-passed small intestine. It is suggested that this simple and safe surgical procedure is justified in selected cases. Conservative treatment of asymptomatic patients with pneumatosis intestinalis, however, may prevent further surgical intervention in these patients.
Four cases of pneumatosis cystoides intestinalis associated with unexplained ascites in Ethiopian patients are described. The relationship between the two conditions has not previously been reported.
The authors present a case of pneumatosis cystoides intestinalis, observed by chance in a patient previously subjected to end-to-end jejunoileal bypass for severe obesity. Using their observation and the cases reported in the literature as a starting point, the authors discuss the etiopathogenetic and clinical aspects of the disease, and briefly indicate what is, at present considered the correct therapeutic approach.