Adenomyoma of the small intestine a rare pathological lead point for intussusception in an infant
© Bak et al.; licensee Springer. 2014
Received: 24 July 2014
Accepted: 8 October 2014
Published: 18 October 2014
Intussusception is a typical abdominal emergency in early childhood.
We report a case of an infant in the typically affected age group with an intussusception triggered by a rare benign intramural intestinal adenomyoma as a pathological lead point. The infant had the typical symptoms of a recurrent idiopathic ileocolic intussusception.
Discussion and evaluation
Idiopathic intussusception is frequent in the infant age group. Contrary to that, reports on pathological lead points for intussusceptions are sparse in the toddler age.
That case illustrates that even in intussusceptions in the typically affected age group, it is important to be aware of pathological lead points, especially if the intussusceptions are recurrent.
Intussusception occurs when a proximal part of the bowel invaginates into a more distal part, typically within the ileocoecal region, which occurs commonly in infants and children between 3 months and 4 years of age. Typical symptoms in these patients include a triad of acute abdominal pain, vomiting and bloody stools; however, regularly, patients present with variable, non-specific symptoms. Ultrasonography is the established standard for diagnosis of intussusception and has a high sensitivity and specificity (Lehnert et al. 2009). Idiopathic intussusception occurs due to swollen mesenteric lymph nodes in patients in the typically affected age group that have been affected by viral infection or non-specific immunologic factors. If recurrent intussusception or intussusception occur in older children, the presence of a pathological lead point must be considered. Herein, we report and discuss the case of an infant in the typically affected age group with an ileocolic intussusception triggered by an adenomyoma of the distal ileum wall, a rare benign intramural intestinal tumor, acting as pathological lead point.
Discussion and evaluation
Intussusception is a common cause of bowel obstruction in infants and toddlers, with the greatest incidence in infants aged 3–9 months (Lehnert et al. 2009, Gfrorer et al. 2009). There is a seasonal incidence, with peaks in spring and autumn resembling the most typical periods of seasonal gastroenteritis and respiratory tract infections. Most infants do not have a specific lead point. Hypertrophied Peyer’s patches and reactive lymph node hyperplasia, which result from prior viral infection, can serve as a lead point for idiopathic intussusception. Specific lead points (e.g., Meckel diverticulum, intestinal polyps, lymphomas, and intestinal duplication) are more commonly found in older children and adults. Ultrasonography is the preferred diagnostic tool in intussusception and has a sensitivity of 98-100% and a specificity of 88-100% (Lehnert et al. 2009, Gfrorer et al. 2009). Hydrostatic reduction under ultrasound control and contrast enema are established therapies for the treatment of intussusception, with a success rate of 70-90% (Lehnert et al. 2009, Gfrorer et al. 2009). Immediate surgery is indicated in patients who have peritonitis, sepsis, evidence of perforation, unsuccessful non-operative repositioning or a clear finding of pathological lead points. In cases occurring in individuals not in the typical age group or in cases of recurrent intussusceptions, a pathological lead point must be excluded.
Previous reported cases of adenomyoma in intussusception
Schwartz et al.
Gal et al.
Kim et al.
Gal et al.
Lamki et al.
Serour et al.
Chan et al.
Gonzalvez et al.
Yamagami et al.
Lee et al.
Park et al.
Mouravas et al.
Takeda et al.
here described case
Adenomyoma of the small bowel is a rare cause of intussusception in all age groups. The here presented case shows, that even in patients where intussusceptions occur in the typically affected age group, it is important to be aware of pathological lead points, especially in recurrent intussusceptions.
Written informed consent was obtained from the parents for the publication of this report and any accompanying images.
We wish to acknowledge ML Hansmann (Institute of Pathology, Goethe-University Frankfurt/M.) for providing the histological images.
- Chan YF, Roche D: Adenomyoma of the small intestine in children. J Pediatr Surg 1994, 29(12):1611-1612. 10.1016/0022-3468(94)90237-2View ArticleGoogle Scholar
- Gal R, Kolkow Z, Nobel M: Adenomyomatosis hamartoma of the small intestine: a rare cause of intussusception in an adult. Am J Gastroenterol 1986, 12: 1209-1211.Google Scholar
- Gal R, Rath-Wolfson GM, Kessler E: Adenomyoma of the small intestine. Histopathology 1991, 18: 369-371. 10.1111/j.1365-2559.1991.tb00862.xView ArticleGoogle Scholar
- Gfrorer S, Fiegel H, Rolle U: Invagination. Monatsschr Kinderheilkd 2009, 157: 917-924. 10.1007/s00112-009-2048-0View ArticleGoogle Scholar
- Gonzalvez J, Marco A, Andujar M, Iniguez L: Myoepithelial hamartoma of the ileum: a rare cause of intestinal intussusception in children. Eur J Ped Surg 1995, 5: 303-304. 10.1055/s-2008-1066232View ArticleGoogle Scholar
- Kim CJ, Choe GY, Chi JG: Foregut choristoma of the ileum (adenomyoma) – a case report. Ped Pathol 1990, 10: 799-805. 10.3109/15513819009064713View ArticleGoogle Scholar
- Lamki N, Woo CL, Watson AB Jr, Kim HS: Adenomyomatosis hamartoma causing ileoileal intussusception in a young child. Clin Imaging 1993, 17: 183-185. 10.1016/0899-7071(93)90106-WView ArticleGoogle Scholar
- Lee JS, Kim HS, Jung JJ, Kim YB: Adenomyoma of the small intestina in an adult: a rare cause of intussusception. J Gastroenterol 2001, 37(7):556-559.View ArticleGoogle Scholar
- Lehnert T, Sorge I, Till H, Rolle U: Intussusception in children – clinical presentation, diagnosis and management. Int J Colorectal Dis 2009, 24: 1187-1192. 10.1007/s00384-009-0730-2View ArticleGoogle Scholar
- Mouravas V, Koutsoumis G, Patoulias J, Kostopoulos I, Kottakidou R, Kallergis K, Kepertis C, Liolios N: Adenomyoma of the small intestine in children: a rare cause of intussusception: a case report. Turk J Pediatr 2003, 45(4):345-347.Google Scholar
- Park HS, Lee SO, Lee JM, Kang MJ, Lee DG, Chung MJ: Adenomyoma of small intestine: report of two cases and review of the literature. Pathol Int 2003, 53: 111-114. 10.1046/j.1440-1827.2003.01435.xView ArticleGoogle Scholar
- Schwartz SI, Radwin HM: Myoepithelial hamartoma of the ileum causing intussusception. AMA Arch Surg 1958, 77: 102-104. 10.1001/archsurg.1958.01290010104018View ArticleGoogle Scholar
- Serour F, Gorenstein A, Lipnitzky V, Zaidel L: Adenomyoma of the small bowel: a rare cause of intussusception in childhood. J Pediatr Gastroenterol Nutr 1994, 18(2):247-249. 10.1097/00005176-199402000-00021View ArticleGoogle Scholar
- Takeda M, Shoji T, Yamazaki M, Higashi Y, Maruo H: Adenomyoma of the jleum leading to intussusception. Case Rep Gastroenterol 2011, 5(3):602-609. 10.1159/000333400View ArticleGoogle Scholar
- Yamagami T, Tokiwa K, Iwai N: Myoepithelial harmartoma of the ileum causing intussusception in an infant. Pediatr Surg Int 1997, 12: 206-207. 10.1007/BF01350005View ArticleGoogle Scholar
- Zhu HN, Yu JP, Luo J, Jiang YH, Li JQ, Sun WY: Gastric adenomyoma presenting as melena; a case report and literature review. World J Gastroenterol 2010, 16(15):1934-1936. 10.3748/wjg.v16.i15.1934View ArticleGoogle Scholar
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