Double Ileoileal Intussusception Following Surgical Reduction of Ileocolic Intussusception in an 8-Month-old Female.

A previously healthy 8-month-old female presented with 1 day of bilious emesis and bloody stool. An abdominal radiograph was consistent with a small bowel obstruction (Figure 1; available at www.jpeds.com), and abdominal ultrasound scan revealed a target sign, consistent with an ileocolic intussusception (Figure 2). Attempted air enema reduction was unsuccessful, so the patient underwent an uncomplicated exploratory laparotomy with reduction of the ileocolic intussusception as well as an appendectomy. Intraoperative reports noted extensive intussusception of the small bowel to the level of the sigmoid colon, as well as a cecal serosal tear. On postoperative day 1, the patient had increasing abdominal distension with multiple episodes of bilious emesis and a bloody bowel movement. A nasogastric tube was placed for decompression, and a repeat abdominal ultrasound scan showed a double target sign, indicative of double recurrent intussusception, in the right lower quadrant with extensive bowel wall edema and complex ascites (Figure 3; available at www.jpeds.com). The patient went back to the operating room for a second laparotomy that revealed two additional and separate ileoileal intussusceptions that were reduced. No recurrence of the ileocolic intussusception was found. No pathologic lead points were identified on intraoperative examination. The child's past medical history was significant for a complex febrile seizure that occurred the day after receiving a combination vaccination against tetanus, diphtheria, pertussis, polio, and hepatitis B (2 weeks before this presentation). It is unclear whether vaccination or febrile seizure precipitated the event. The ultrasound appearance of intussusception is typically described as a target lesion with concentric hypoechoic and echogenic layers.1Hryhorczuk A.L. Strouse P.J. Validation of US as a first-line diagnostic test for assessment of pediatric ileocolic intussusception.Pediatr Radiol. 2009; 39: 1075-1079Crossref PubMed Scopus (117) Google Scholar In addition to being a safe diagnostic tool with no ionized radiation, ultrasonography has been shown to have high sensitivity and specificity for detecting intussusception (sensitivity of 97.9%, specificity of 97.8%, positive predictive value of 86.6%, and negative predictive value of 99.7%).1Hryhorczuk A.L. Strouse P.J. Validation of US as a first-line diagnostic test for assessment of pediatric ileocolic intussusception.Pediatr Radiol. 2009; 39: 1075-1079Crossref PubMed Scopus (117) Google Scholar Recurrent intussusception after a successful reduction can be seen in 3.0%-15.8% of patients, with up to one-half of these recurrences presenting within 48 hours of the index case.2Gray M.P. Li S.H. Hoffmann R.G. Gorelick M.H. Recurrence rates after intussusception enema reduction: a meta-analysis.Pediatrics. 2014; 134: 110-119Crossref PubMed Scopus (72) Google Scholar, 3Wang Z. He Q.M. Zhang H. Zhong W. Xiao W.Q. Lu L.W. et al.Intussusception patients older than 1 year tend to have early recurrence after pneumatic enema reduction.Pediatr Surg Int. 2015; 31: 855-858Crossref PubMed Scopus (21) Google Scholar, 4Niramis R. Watanatittan S. Kruatrachue A. Anuntkosol M. Buranakitjaroen V. Rattanasuwan T. et al.Management of recurrent intussusception: non-operative or operative reduction?.J Pediatr Surg. 2010; 45: 2175-2180Abstract Full Text Full Text PDF PubMed Scopus (75) Google Scholar Recurrences are less common in children under 1 year of age3Wang Z. He Q.M. Zhang H. Zhong W. Xiao W.Q. Lu L.W. et al.Intussusception patients older than 1 year tend to have early recurrence after pneumatic enema reduction.Pediatr Surg Int. 2015; 31: 855-858Crossref PubMed Scopus (21) Google Scholar as well as after an operative reduction (3%) compared with enema reduction (11.4%-15.8%).2Gray M.P. Li S.H. Hoffmann R.G. Gorelick M.H. Recurrence rates after intussusception enema reduction: a meta-analysis.Pediatrics. 2014; 134: 110-119Crossref PubMed Scopus (72) Google Scholar, 4Niramis R. Watanatittan S. Kruatrachue A. Anuntkosol M. Buranakitjaroen V. Rattanasuwan T. et al.Management of recurrent intussusception: non-operative or operative reduction?.J Pediatr Surg. 2010; 45: 2175-2180Abstract Full Text Full Text PDF PubMed Scopus (75) Google Scholar For recurrent intussusception, enema reduction is the standard approach to management and is highly successful, even after an initial operative reduction.4Niramis R. Watanatittan S. Kruatrachue A. Anuntkosol M. Buranakitjaroen V. Rattanasuwan T. et al.Management of recurrent intussusception: non-operative or operative reduction?.J Pediatr Surg. 2010; 45: 2175-2180Abstract Full Text Full Text PDF PubMed Scopus (75) Google Scholar, 5Fisher J.G. Sparks E.A. Turner C.G. Klein J.D. Pennington E. Khan F.A. et al.Operative indications in recurrent ileocolic intussusception.J Pediatr Surg. 2015; 50: 126-130Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar, 6Bai Y.Z. Chen H. Wang W.L. A special type of postoperative intussusception: ileoileal intussusception after surgical reduction of ileocolic intussusception in infants and children.J Pediatr Surg. 2009; 44: 755-758Abstract Full Text Full Text PDF PubMed Scopus (15) Google Scholar In our patient, repeat operative management was chosen over enema reduction because of the concern for necrotic bowel and risk of perforation. Overall, the vast majority of intussusceptions are ileocolic (98%), and only a small percentage are ileoileal (0.7%).7Hsu W.L. Lee H.C. Yeung C.Y. Chan W.T. Jiang C.B. Sheu J.C. et al.Recurrent intussusception: when should surgical intervention be performed?.Pediatr Neonatol. 2012; 53: 300-303Abstract Full Text Full Text PDF PubMed Scopus (46) Google Scholar One case series identified 6 patients with single ileoileal intussusception 2-4 days after surgical reduction of ileocolic intussusception.6Bai Y.Z. Chen H. Wang W.L. A special type of postoperative intussusception: ileoileal intussusception after surgical reduction of ileocolic intussusception in infants and children.J Pediatr Surg. 2009; 44: 755-758Abstract Full Text Full Text PDF PubMed Scopus (15) Google Scholar Patients in this study presented with progressive abdominal distension and bilious emesis like our patient. However, unlike our patient, none reported bloody stool. Double (simultaneous) intussusceptions are rare, with only 10 cases identified, none of which occurred after reduction of a primary intussusception.8Shiu J.R. Chao H.C. Chen C.C. Chi C.Y. Rare concurrent ileoileal and ileocolic intussusceptions in a child presenting with painless hematochezia.Pediatr Neonatol. 2010; 51: 359-362Abstract Full Text PDF PubMed Scopus (5) Google Scholar, 9Singh J.K. Bawa M. Kanojia R.P. Ghai B. Menon P. Rao K.L. Idiopathic simultaneous intussusceptions in a neonate.Pediatr Surg Int. 2009; 25: 445-447Crossref PubMed Scopus (8) Google Scholar, 10Pandey A. Rawat J.D. Wakhlu A. Kureel S.N. Gopal S.C. Simultaneous occurrence of jejuno-jejunal and ileo-ileal intussusception in a child: a rare occurrence.BMJ Case Rep. 2011; https://doi.org/10.1136/bcr.08.2010.3294Crossref Scopus (8) Google Scholar The reported cases included double ileoileal, double ileocolic, double colocolic, simultaneous ileocolic and ileoileal, and simultaneous jejunojejunal and ileoileal.8Shiu J.R. Chao H.C. Chen C.C. Chi C.Y. Rare concurrent ileoileal and ileocolic intussusceptions in a child presenting with painless hematochezia.Pediatr Neonatol. 2010; 51: 359-362Abstract Full Text PDF PubMed Scopus (5) Google Scholar, 9Singh J.K. Bawa M. Kanojia R.P. Ghai B. Menon P. Rao K.L. Idiopathic simultaneous intussusceptions in a neonate.Pediatr Surg Int. 2009; 25: 445-447Crossref PubMed Scopus (8) Google Scholar, 10Pandey A. Rawat J.D. Wakhlu A. Kureel S.N. Gopal S.C. Simultaneous occurrence of jejuno-jejunal and ileo-ileal intussusception in a child: a rare occurrence.BMJ Case Rep. 2011; https://doi.org/10.1136/bcr.08.2010.3294Crossref Scopus (8) Google Scholar Supported by the National Center for the Advancement of Translational Science (U1TR000038), National Institutes of Health (NIH) (U1TR000038) (to. G.G.-von S), and the National Institute for Diabetes and Digestive and Kidney Diseases (R25DK092170-05), NIH (R25DK092170-05) (PI G.G.-von S.). Figure 3Postoperative abdominal ultrasound scan showing a double target sign, consistent with double ileoileal intussusceptions. Within the right lower quadrant there is intussusception with markedly thickened and edematous loops of bowel measuring approximately 1.5 cm.View Large Image Figure ViewerDownload Hi-res image Download (PPT)