Volume 9, Issue 2 (4-2021)                   J. Pediatr. Rev 2021, 9(2): 163-166 | Back to browse issues page


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Kumar P, Kumar Sarin Y, Jain N. The Prolapsed Intussusceptum in a 4-month-old Male: A Case Report. J. Pediatr. Rev 2021; 9 (2) :163-166
URL: http://jpr.mazums.ac.ir/article-1-322-en.html
1- Department of Pediatric Surgery, Chacha Nehru Bal Chikitsalya, New Delhi, India.
2- Department of Pediatric Surgery, Maulana Azad Medical College, New Delhi, India. , yksarin@gmail.com
3- Department of Pediatric Surgery, Maulana Azad Medical College, New Delhi, India.
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1. Context
The intussusception is a surgical emergency resulting from the invagination of a bowel segment (intussusceptum) into the distal segment (intussuscipiens); if untreated, this condition leads to the catastrophic event of bowel ischemia resulting in necrosis and perforation.

2. Case Report
A 4-month-old boy presented to the emergency department with the complaint of a mass protruding out of the anus for one day. On further questioning, it was revealed that the patient had complaints of a painful abdomen, intermittent crying, and diarrhea for 7 days; for which, he was referred to a local practitioner 3 days back. Accordingly, symptomatic medications, oral rehydration solution, and zinc were prescribed for him. There was also a history of 2 episodes of bilious vomiting before presentation to us. On presentation, the infant was sick-looking, pale, and lethargic. He had tachypnea (respiratory rate: 37 min), tachycardia (heart rate: 136 min), deranged Capillary Filling Time (CFT) of >3 sec, and the signs of shock and dehydration. The abdomen was distended with sluggish bowel sounds and obliterated liver dullness. Perineal examination revealed necrotic bowel prolapsing out of the anus (Figure 1). 
Two wide bore peripheral intravenous lines were secured and 2 fluid boluses of 20 mL/kg normal saline were given to the patient. His laboratory investigations revealed leukocytosis, Total Leucocyte Counts (TLC) of 21000 cmm with neutrophilia, hemoglobin: 7.9 g%, blood urea: 32 mg/dL, serum creatinine: 1mg/dL, and normal platelets. The venous blood gas demonstrated acidosis with a pH of 7.1 and bicarbonate levels of 8 meq/L. Serum Na+ was equal to 121 meq/L and K+ equaled 2.5 meq/L. The X-ray of the abdomen confirmed free gas under the diaphragm. The administration of triple antibiotics ceftriaxone, amikacin, and metronidazole were initiated for the patient for the coverage of Gram-positive, negative, and anaerobic organisms. After aggressive resuscitation and starting on maintenance fluid DNS 100 ml/kg/day with 2:100 KCl, the acidosis component got corrected and CFT was measured as 3 seconds. The infant underwent exploratory laparotomy, which revealed terminal ileum prolapsed through large bowel and coming out of the rectum, with perforated ascending, transverse, and descending colon (Figure 2).
The intussusceptum had partially necrosed and shriveled. The unhealthy small bowel and perforated large bowel were resected and ileostomy and distal mucus fistula at sigmoid were created. The patient had an uneventful postoperative course and discharged satisfactorily on POD-7 after sepsis was controlled. He is awaiting ileocolic anastomosis at a later date. 

3. Discussion
The intussusception predominantly affects children aged 6 months to 3 years, with >90% of the cases in the first 2 years of life. Literature refutes the seasonal variations; however, geographic and demographic differences exist. The diagnosis of intussusception may be challenging in non-specific presentations and mimicking common conditions, like gastroenteritis, which may cause delayed or missed diagnosis. The clinical triad of abdominal pain, red currant jelly stool, and palpable mass may be present in less than half of the cases.
Ileo-colic intussusception is the most prevalent type of intussusception. The long mesentery allows for migration up to the distal large bowel and even transanal protrusion (1). Radiological assessment using X-rays help with ruling out perforation as in our case; however, target or meniscus signs may also be appreciated in a few. Ultrasonography has a sensitivity of 98%-100%, a specificity of about 88%, and a negative predictive value of 100%. The hydrostatic or pneumatic reduction may be tried at the initial presentation of ileocolic intussusception. A 2017 Cochrane meta-analysis indicated air enema may be more successful than a hydrostatic enema (2). The indications of surgical intervention include unsuccessful non-surgical reduction, hemodynamic instability, and perforation. Laparotomy is the only option left is delayed cases.
The prolapsed intussusception is a missed event and late complication. It is a rare scenario and the relevant incidence quoted in the literature is up to 16 % (3). Trans-Anal intussusception prolapse may be confused with simple rectal prolapse; however, it may be very well differentiated on Per-Rectal (PR) examination. In intussusception, the finger can be easily insinuated between the prolapse part and rectum. In the presented case, it was a long gangrenous prolapsed segment of the bowel obviating the need for PR examination. Obiora et al. also stated that the average age at presentation for the trans-anal protrusion of intussusception is >1 year for 40% of children (3). Additionally, it has a female predisposition, contrary to other intussusceptions (3). It requires prompt diagnosis and management to prevent morbidity and mortality (4). A tabular presentation of various reports in the literature is listed in Table 1.

Ugwu BT et al. explored high bowel resection rates in trans-anal protrusion cases (67%), compared to other forms (30%) (11). Our patient also underwent the resection of unhealthy and perforated bowel due to late presentation. 
The mortality rate varies from 1 % in developed countries to 9.4 % in resource-challenged nations (12). Chalya PL et al. inferred that high mortality rates are associated with less than one year of age, delayed presentation greater than 24 hours, associated peritonitis, bowel resection, and surgical site infection (13). Our patient presented most of these factors but could be salvaged due to aggressive management. 

4. Conclusion
A high index of suspicion and focused imaging allows the early identification and subsequent management. Young age, male gender, prolapsed necrosed intussusceptum, the perforation of the most of large bowel, and good final outcome merits the publication of index case. 

Ethical Considerations
Compliance with ethical guidelines

The participants were informed of the purpose of the research and its implementation stages. A written consent has been obtained from the subjects. They were also assured about the confidentiality of their information and were free to leave the study whenever they wished, and if desired, the research results would be available to them. The Helsinki Convention was also observed.

Funding
This research did not receive any grant from funding agencies in the public, commercial, or non-profit sectors. 

Authors' contributions
All authors equally contributed to preparing this article. 

Conflicts of interest
The authors declared no conflicts of interest.


References
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  2. Gluckman S, Karpelowsky J, Webster AC, McGee RG. Management for intussusception in children. The Cochrane database of systematic reviews. 2017; 6(6):CD006476. [DOI:10.1002/14651858.CD006476.pub3] [PMID] [PMCID]
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  5. Mutua I, Ransom J, Kiptoon D. Trans-anal prolapse of ileo-colic intussusception. Journal of Pediatric Surgery Case Reports. 2018; 38:1-3. https://www.sciencedirect.com/science/article/pii/S2213576618301519
  6. Basu S. Trans-anal protrusion of intussusception (TAPI) revisited: Managed successfully in a resource limited hospital setting. International Surgery Journal. 2019; 6:310-3. [DOI:10.18203/2349-2902.isj20185494]
  7. Ray A, Mandal KC, Shukla RM, Roy D, Mukhopadhyay B, Bhattacharya M. Neglected intussusception presenting as transanal prolapse of small bowel. Indian Journal of Pediatrics. 2012; 79(10):1370-1. [DOI:10.1007/s12098-012-0692-1] [PMID]
  8. Ibrahim IA. Prolapsed ileocolic intussusception. Annals of Pediatric Surger. 2011; 7(2):76-8. [DOI:10.1097/01.XPS.0000396409.62385.a7]
  9. Coghill J, Mensah. Anal protrusion of intussusception. BMJ Case Reports. 2009; 2009:bcr0920092314. [DOI:10.1136/bcr.09.2009.2314]
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  11. Ugwu BT, Legbo JN, Dakum NK, Yiltok SJ, Mbah N, Uba FA. Childhood intussusception: A 9-year review. Annals of Tropical Paediatrics. 2000; 20(2):131-5. [DOI:10.1080/02724936.2000.11748122] [PMID]
  12. Fraser JD, Aguayo P, Ho B, Sharp SW, Ostlie DJ, Holcomb III GW, et al. Laparoscopic management of intussusception in pediatric patients. Journal of Laparoendoscopic & Advanced Surgical Techniques. 2009; 19(4):563-5.[DOI:10.1089/lap.2009.0117] [PMID]
  13. Chalya PL, Kayange NM, Chandika AB. Childhood intussusceptions at a tertiary care hospital in northwestern Tanzania: A diagnostic and therapeutic challenge in resource-limited setting. Italian Journal of Pediatrics. 2014; 40(1):28. [DOI:10.1186/1824-7288-40-28] [PMID] [PMCID]
Type of Study: Case Report and Review of Literature | Subject: Pediatrics
Received: 2020/04/7 | Accepted: 2020/12/27 | Published: 2021/04/1

References
1. Ngom G, Kane A, Ndour O, Cissokho Can, Ndoye M. Prolapsed Intussusception in Children: clinical study and therapeutic aspects. J Pediatr Surg Specialities. 2013; 7(1):644.
2. Gluckman S, Karpelowsky J, Webster AC, McGee RG (2017) Management for intussusception in children. Cochrane Database Syst Rev (6) Art. No.: CD006476 [DOI:10.1002/14651858.CD006476.pub3] [PMID] [PMCID]
3. Obiora EU, Okwuchukwu ES, Ogundu II. Trans anal protrusion of intussusception in children. Afr J Paediatr Surg. 2014;11(3):229-32. [DOI:10.4103/0189-6725.137331] [PMID]
4. Frank LT, Benjamin MK, Christian AD, Valirie NA. Delayed diagnosis of transanal prolapse of an ileo- colic intussusception in a 10 month old infant in rural Cameroon: a case report. BMC Res Notes Oct 2017; 10:521. [DOI:10.1186/s13104-017-2838-8] [PMID] [PMCID]
5. Mutua I, Ransom J, Kiptoon D. Trans-anal prolapse of ileo-colic intussusception. Journal of pediatric surgery case reports. 2018; 38:1-3. [DOI:10.1016/j.epsc.2018.07.029]
6. Basu S. Trans-anal protrusion of intussusception (TAPI) revisited: managed successfully in a resource limited hospital setting. Int Surg J 2019; 6:310-3. [DOI:10.18203/2349-2902.isj20185494]
7. Tianyi FL, Kadia BM, Dimala CA, Agbor VN. Delayed diagnosis of transanal prolapse of an ileo-colic intussusception in a 10-month-old infant in rural Cameroon: a case report. BMC research notes. 2017; 10(1):1-5. [DOI:10.1186/s13104-017-2838-8] [PMID] [PMCID]
8. Ray A, Mandal KC, Shukla RM, Roy D, Mukhopadhyay B, Bhattacharya M. Neglected intussusception presenting as transanal prolapse of small bowel. Indian J Pediatr. 2012; 79(10):1370-1. [DOI:10.1007/s12098-012-0692-1] [PMID]
9. Ibrahim I. A; Prolapsed ileocolic intussusception. Ann Pediatr Surg. 2011; 7(2):76-8. [DOI:10.1097/01.XPS.0000396409.62385.a7]
10. Coghill J. Anal protrusion of intussusception. Case Reports. 2009 Jan 1;2009:bcr0920092314 [DOI:10.1136/bcr.09.2009.2314] [PMID] [PMCID]
11. Ameh EA, Mshelbwala PM. Transanal protrusion of intussusception in infants is associated with high morbidity and mortality. Ann Trop Paediatr.2008; 28(4):287-92. [DOI:10.1179/146532808X375459] [PMID]
12. Ugwu BT, Legbo JN, Dakam NK, Yiltok SJ, Mbah N, Uba FA. Childhood intussusception: a 9 year review. Ann Trop Paediatr 2000; 20:131-5. [DOI:10.1080/02724936.2000.11748122] [PMID]
13. Fraser JD, Aguayo P, Ho B, Sharp SW, Ostlie DJ, Holcomb III GW, et al. Laparoscopic management of intussusception in pediatric patients. Journal of Laparoendoscopic & Advanced Surgical Techniques. 2009; 19(4):563-5. [DOI:10.1089/lap.2009.0117] [PMID]
14. Chalya PL, Kayange NM, Chandika AB. Childhood intussusceptions at a tertiary care hospital in northwestern Tanzania: a diagnostic and therapeutic challenge in resource-limited setting. Ital J Pediatr.2014; 40(1):28. [DOI:10.1186/1824-7288-40-28] [PMID] [PMCID]

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