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Journal of Indian Association of Pediatric Surgeons
     Journal of Indian Association of Pediatric Surgeons
Official journal of the Indian Association of Pediatric Surgeons         
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Year : 2007  |  Volume : 12  |  Issue : 4  |  Page : 221-223

Recurrent intussusception, coeliac disease and cholelithiasis: A unique combination

1 The Children's Hospital, University Hospitals of Leicester, United Kingdom
2 Northampton General Hospital, United Kingdom

Correspondence Address:
S Nour
Department of Paediatric Surgery, The Children's Hospital, Leicester Royal Infirmary, University Hospitals of Leicester NHS Trust, Leicester, LE1 5WW
United Kingdom
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0971-9261.40840

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Authors report an 11-month-old female child, who presented with recurrent episodes of colicky abdominal pain and diarrhea. An abdominal ultrasound revealed small bowel intussusception. She was also noted to have a thick walled gall bladder and a solitary gallstone. Further investigations confirmed the diagnosis of coeliac disease. The combination of small bowel intussusception, coeliac disease and cholelithiasis is unique and has not been reported in the literature.

Keywords: Child, cholelithiasis, coeliac disease, intussusception

How to cite this article:
Sinha C K, Haider N, Zaw W, Nour S. Recurrent intussusception, coeliac disease and cholelithiasis: A unique combination. J Indian Assoc Pediatr Surg 2007;12:221-3

How to cite this URL:
Sinha C K, Haider N, Zaw W, Nour S. Recurrent intussusception, coeliac disease and cholelithiasis: A unique combination. J Indian Assoc Pediatr Surg [serial online] 2007 [cited 2023 Mar 22];12:221-3. Available from: https://www.jiaps.com/text.asp?2007/12/4/221/40840

   Introduction Top

The association of small bowel intussusception and celiac disease is rare, but known. [1],[2],[3] However, a combination of recurrent small bowel intussusception, coeliac disease and cholelithiasis has never been described in literature. We report an infant with this unique combination.

   Case History Top

An 11-month-old female child was referred with a six-week history of recurrent abdominal distension and diarrhea. This was associated with excessive cry, which seemed to resolve spontaneously after several hours. Pale, loose stools without mucus or blood were features in most of the episodes. She was admitted to her local hospital and was undergoing investigations for the above symptoms. It was initially thought that she was lactose intolerant and, so, was taken off dairy products. With this intervention, there was a transient improvement, but her symptoms returned after some time. So, a coeliac screen was requested along with other investigations.

At her most recent admission, she presented with high fever, lethargy, irritability and clinical signs of shock. On examination, there was tenderness in the right upper quadrant along with moderate hepatomegaly and a palpable gall bladder. Hematological tests were done for white cell count (33.5x10 9 /l), hemoglobin (11.7 g/dl), platelets (438 x10 9 /l) and CRP (229 mg/l). An abdominal X-ray was suggestive of bowel obstruction and an ultrasound showed some free fluid in the right iliac fossa. A possibility of acute appendicitis was raised and she was transferred to the regional pediatric surgical unit for further assessment. After initial resuscitation, she underwent a repeat ultrasound, which showed no features of appendicitis. Her clinical condition improved slowly over the next few days with conservative management. A repeat ultrasound done two days later (for excessive crying and increased abdominal distension) showed a thick walled gall bladder with a solitary gallstone [Figure - 1]. On prolonged ultrasound examination, a spontaneously resolving small bowel intussusception was noticed [Figure - 2]. Simultaneously, her coeliac screen antibody (IgA anti-endomysial) came back positive. She was discharged a week later on gluten free diet with plans to perform an upper gastrointestinal endoscopy and duodenal biopsies at a later date. Ever since the commencement of gluten free diet, she has remained well, with no recurrence of abdominal pain and diarrhea. Astonishingly, there was no stone in the gall bladder on repeat ultrasound! Her parents are not keen for her to have an upper gastrointestinal endoscopy and biopsies in view of her clinical improvement on gluten free diet. One year on, she is thriving well and is asymptomatic.

   Discussion Top

Small bowel intussusception comprises of less than 10% of all pediatric intussusceptions. [4] Although about 18% of the children with coeliac disease show radiological features of small bowel intussusception, clinical association is rare. [1],[2],[3],[5] Transient small bowel intussusception is considered to be a specific radiological sign of coeliac disease; possibly due to hypotony of bowel loops and/or intestinal motor abnormalities. [6],[7] Cucchiara et al . described motor abnormalities in active coeliac disease that suggested a marked derangement of the neural control of gut motility, which improved after a six-month period of gluten free diet. [8] This is possibly due to increased expression of cytokines within the mucosa. [9] The gallbladder has a ganglionic plexus similar to the enteric nervous system. Nerves arising from the myenteric plexus of the duodenum directly supply the gall bladder wall. [10] Few authors have reported gallbladder inertia in coeliac disease, which causes decreased contractility and defective emptying of the gall bladder. [11],[12],[13] This is due to decreased cholecystokinin secretion from the already damaged mucosa of the small bowel and also a poor response of the gallbladder wall to the endogenous cholecystokinin. [14],[15],[16] The resulting effect is distension of the gall bladder, which predisposes to stone formation (as seen in our case). Although, recurrent small bowel intussusceptions hypothetically appear to be a common physiological event in coeliac disease, the clinical presentation is rarely mentioned in pediatric literature. However, the few case reports that have mentioned the clinical occurrence of intussusception in coeliac disease suggest, that most of them resolve spontaneously with conservative management i.e. the optimum nutritional support. [7],[17]

A single instance of gallbladder distension (but not cholelithiasis) in association with small bowel intussusception and coeliac disease has been published so far. [7] Our case report is the first of its kind to present the rare triad of cholelithiasis, small bowel intussusception and coeliac disease. It also emphasizes the fact that coeliac disease may predispose to recurrent intussusception, as well as gallbladder dysmotility and stone formation.

   References Top

1.Al Furaikh S, Al Zaben. Recurrent small bowel intussusceptions: An uncommon presentation of celiac disease in an Arab child. Trop Gastroenterol 2005;26:38-9.  Back to cited text no. 1    
2.Lastennet F, Piloquet H, Camby C. Bowel intussusception revealing coeliac disease in a nine month old infant. Arch Pediatr 2002;9:151-4.  Back to cited text no. 2    
3.Martinez G, Israel NRB, White JJ. Coeliac disease presenting as entero-enteral intussusception. Pediatr Surg Int 2001;17:68-70.  Back to cited text no. 3    
4.Cerrah CA, Celayir S, Kutlu T. The role of surgery in the treatment of intussusception in celiac disease. Turk J Gastroenterol 2002;13:63-5.  Back to cited text no. 4    
5.Burrows FG, Toye DK. Coeliac disease: Barium studies. Clin Gastroenterol 1974;3:91-107.  Back to cited text no. 5  [PUBMED]  
6.Masterson JB, Sweeney EC, Path MC. The role of small bowel follow through examination in the diagnosis of coeliac disease. Br J Radiol 1976;49:660-4.  Back to cited text no. 6    
7.Gonzalez JA, Gonzalez JB, Crespo MJ. Acute gallbladder distension and recurrent small bowel intussusception in a child with coeliac disease. J Pediatr Gastroenterol Nutr 1998;27:444-5.  Back to cited text no. 7    
8.Cucchiara S, Bassoti G, Castellucci G. Upper gastrointestinal motor abnormalities in children with active coeliac disease. J Pediatr Gastroenterol Nutr 1995;21:435-42.  Back to cited text no. 8    
9.Kontakou M, Przemioslo RT, Sturges RP. Cytokine mRNA expression in the mucosa of treated coeliac patients after wheat peptide challenge. Gut 1995;37:52-7.  Back to cited text no. 9    
10.Mawe GM, Gershon MD. Structure, afferent innervation and transmitter content of ganglia of the guinea pig gallbladder: Relationship to the enteric nervous system. J Comp Neurol 1989;283:374-90.  Back to cited text no. 10  [PUBMED]  
11.Delamarre J, Capron JP, Joly JP. Atonie vesiculaire et maladie coeliaque de l'adulte. Etude radiographique et echographique de 15 cas. J Radiol 1984;65:133-6 [Article not English].  Back to cited text no. 11    
12.Low-Beer TS. Galbladder inertia and sluggish enterohepatic circulation of bile-salts in coeliac disease. Lancet 1971;7707:991-4.  Back to cited text no. 12    
13.Fraquelli M, Pagliarulo M, Colucci A, Paggi S, Conte D. Gallbladder motility in obesity, diabetes mellitus and coeliac disease. Dig Liver Dis 2003;35:S12-6.  Back to cited text no. 13  [PUBMED]  
14.Masclee AA, Jansen JB, Driessen WM. Gallbladder sensitivity to cholecystokinin in coeliac disease- Correlation of gallbladder contraction with plasma cholecystokinin-like immunoreactivity during infusion of cerulein. Scand J Gastroenterol 1991;26:1279-84.  Back to cited text no. 14    
15.Maton PN, Seldon AC, Fitzpatrick ML. Defective gallbladder emptying and cholecystokinin release in coeliac disease- Reversal by gluten- free diet. Gastroenterology 1985;88:391-6.  Back to cited text no. 15    
16.Deprez P, Sempoux C, Van Beers BE, Jouret A, Robert A, Rahier J, et al . Persistent decreased plasma cholecystokinin levels in celiac patients under gluten-free diet: Respective roles of histological changes and nutrient hydrolysis. Regul Pept 2002;110:55-63.  Back to cited text no. 16  [PUBMED]  [FULLTEXT]
17.Germann R, Kuch M, Prinz K. Celiac disease: An uncommon cause of recurrent intussusception. J Pediatr Gastroenterol Nutr 1997;25:415-6.  Back to cited text no. 17    


  [Figure - 1], [Figure - 2]

This article has been cited by
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