A rare association of intussusception and celiac disease in a child

Abstract

CONTEXT: Intussusception is a common cause of acute intestinal obstruction in the pediatric population and it is normally idiopathic. Rare cases of chronic intussusception require investigation with greater attention. CASE REPORT: We present a clinical case of a three-year-old boy with aqueous diarrhea, abdominal distension, vomiting and weight loss over a two-month period. During the investigation, abdominal ultrasound showed imaging of intussusception. The intraoperative findings showed the intussusception had resolved spontaneously. In further investigation, it was found that the diarrhea was malabsorptive and, after the patient underwent upper gastrointestinal endoscopy, a diagnosis of celiac disease was made. After a gluten-free diet was introduced, the patient showed complete remission of symptoms and regained weight, and normal growth was reestablished. CONCLUSION: If the clinical presentation of intussusception is unusual, etiological investigation should be undertaken. In this case report, celiac disease was the underlying cause.RESUMO CONTEXTO: Intussuscepção é uma causa frequente de obstrução intestinal aguda em pacientes pediátricos, e geralmente é idiopática. Raros casos de intussuscepção crônica devem ser investigados com maior atenção. RELATO DE CASO: Apresentaremos um caso clínico de um menino de três anos com diarreia aquosa, distensão abdominal, vômito e perda de peso por dois meses, cuja investigação por ultrassonografia abdominal revelou imagem de intussuscepção. Os achados intraoperatórios mostraram que a intussuscepção havia se resolvido espontaneamente. Em investigação subsequente, foi identificado que a diarreia era malabsortiva e, após a realização de endoscopia digestiva alta, foi feito diagnóstico de doença celíaca. Foi iniciada dieta com restrição de glúten e o paciente teve remissão completa dos sintomas, recuperou peso e o crescimento foi restabelecido. CONCLUSÃO: Caso a apresentação clínica de intussuscepção não seja a habitual, deve-se prosseguir a investigação etiológica. No caso clínico apresentado, doença celíaca foi a causa subjacente.I MD, MSc. Attending Physician,

Intussusception associated with celiac disease in a child: case report and review of literature

“…Surgical intervention needed in case of prolonged intussusception, shock, peritonitis and perforation or when non-operative intervention is either unsuccessful or lead to perforation, or in situations of lack of a trained professional for non-invasive approaches. 1,12,13 Gonda TA et al, determined 14 cases of intussusception among a cohort of 880 patients with CD (mean age of 47±17.5 years). 9 Eleven of them underwent imaging for abdominal pain and three were incidental.…”

“…But multiple enlarged mesenteric lymph nodes as reported during surgery, diffuse chronic inflammation with stiffened and thickened bowel, disturbed peristalsis in the hypotonic intestinal loop, dilated bowel, ulceration and weakening of intestinal wall may act as lead point for intussusception. 5,6,8,13 Intussusception in celiac patients are transient and may be chronic and painless, so can easily be missed in single examination and thus mostly underdiagnosed. In adults, most cases of intussusception are investigated because it occurs due to pathological condition.…”

“…While in children, mostly they idiopathic and identified by chance when undergoes imaging for abdominal pain, otherwise investigated only when an abnormal clinical presentation or physical examination ensues. 6,8,13 Whether spontaneous resolution in CD is more frequent, will be determined only when routine abdominal ultrasound becomes protocol in CD workup and a large serial study on pediatric populations might help in resolving this question.…”

Surgical Aspects of Celiac Disease

“…Поскольку инвагинация у пациентов с целиакией может быть хронической и безболезненной, вероятно, ее частота недооценивается. По данным литературы, инвагинация у пациентов с целиакией может разрешаться без оперативного вмешательства, в том числе и у детей [13,17,20]. V. Borkar и соавт.…”

“…[7]. В доступной литературе имеются публикации, касающиеся ассоциации целиакии с инвагинацией и другой хирургической патологией у взрослых [8][9][10][11] и детей [5,[12][13][14][15][16][17][18][19][20]. В отечественной литературе мы не встретили ни одной публикации по данному вопросу.…”