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Description
A previously healthy adolescent girl presented to the emergency department with intermittent, periumbilical pain of 3 days’ duration. Her visit was prompted by exacerbation and increasing frequency of the pain. She denied other symptoms, such as vomiting, diarrhoea and bloody stool as well as a history of surgery, trauma and recent consumption of raw food. Her vital signs were normal, and a physical examination revealed pain on compressing the periumbilical area. There were no peritoneal signs. The intestines were not able to be clearly visualised on abdominal ultrasound. Abdominal radiography revealed multiple, round lucencies distributed in a crescent-like pattern in the left upper quadrant and absence of air in the right side of the abdomen (figure 1).
Abdominal radiograph revealed round lucencies distributed in a crescent-like pattern in the left upper quadrant (white arrow).
Non-contrast abdominal CT revealed colocolic intussusception with multiple, intramural, air-filled cysts at the lead point within the transverse colon (figure 2). Based on these findings, intussusception secondary to pneumatosis cystoides intestinalis (PCI) was diagnosed. The patient successfully underwent radiological hydrostatic reduction. An abdominal radiograph taken 7 hours after the reduction revealed multiple, colonic cysts mainly in the caecum (figure 3). At the 3-month follow-up, the PCI was no longer visible on an abdominal radiograph.
Non-contrast abdominal CT revealed colocolic intussusception with multiple, intramural, air-filled cysts at the lead point within the transverse colon (white arrows).
Post reduction abdominal radiograph revealed multiple, colonic cysts mainly in the caecum (white arrow).
PCI is intestinal intramural air-filled cysts, most often developing in patients in their 40–50s.1 Although the majority of PCI develops secondary to other intestinal or pulmonary diseases, around 15% of them develops in patients without any background medical conditions.1 Although CT showed high sensitivity in detecting PCI, diagnosis is often delayed due to its non-specific symptoms.1
Intussusception, or the invagination of a part of the intestine (the intussusceptum) into the adjacent part (the intussuscipiens), is a common abdominal emergency in children.2 It is usually diagnosed by ultrasonography, and delayed diagnosis can lead to ischaemia of the intestine. The most common age range at presentation is 3 months to 36 months.2 If intussusception develops in a patient outside this age range, a pathological lesion should be sought as the lead point. In our patient, PCI developing in the caecum acted as the lead point, telescoping the caecum into the transverse colon. Although most PCI cases can be managed conservatively, complications, including intussusception, are sometimes known to occur.3
In the present case, abdominal radiography played a major role in diagnosing intussusception because the intramural air in the large intestine hampered ultrasonographic evaluation. On abdominal radiograph, delineation of the tip of the intussusceptum outlined by the intestinal air within the intussuscipiens is called the meniscus sign, which is known to be highly specific to ileocecal intussusception.4 Although the air within the transverse colon was not seen on abdominal radiograph in our patient, intramural air in the inverted caecum outlined the crescent-shaped contour of the intussusceptum, leading to the suspicion of intussusception. In patients with PCI, a crescent-like distribution of cysts on an abdominal radiograph may serve as an important clue to detecting this complication.
Learning points
Intussusception is a rare complication of pneumatosis cystoides intestinalis.
Since ultrasonographic evaluation could be hampered by intramural air, an abdominal radiography may serve as an important clue to detecting intussusception in patients with pneumatosis cystoides intestinalis.
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Footnotes
Contributors KI drafted the manuscript, and YH revised it. All authors approved the final version of the manuscript.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.