Intussusception is the most common, non-congenital, cause of intestinal obstruction in infants. There are various patterns of intussusception seen in children with the most common being ileo-colic variety. Compound intussusception in which intussusception involves more than one non-adjacent segment is a rare finding in the literature and it can be associated with worse morbidity than typically occurs, especially in a region where delayed presentation is a major contributor to morbidity and mortality in the treatment of intestinal obstruction.We report the first documented case of double compound intussusception in an African child and reviewed relevant literature.Keywords: Antegrade and retrograde; Children; Compound; Intussusceptio

Adeoye, A.O.

Egbuchulem, K.I.

Lawal, T.A.

Nweke, M.C.

English

AJOL - African Journals Online

INTRODUCTIONIntussusception is the invagination of a segment ofintestine into the lumen of an immediately adjoiningsegment, which is mostly a distal segment with resultantintestinal obstruction. It is the most common acquiredcause of intestinal obstruction in childhood1 and canoccur in variants including: ileo-ileal, colo-colic and ileo-colic, which are the most commonly seen type in 75to 80%.2, 3 Almost always, the invagination is fromproximal to more distal segments of  bowel and rarely,however, it may occur in a retrograde fashion. Therarest form of  the pathology is compoundintussusception in which intussusception involves bothantegrade and retrograde patterns on differentsegments of bowel. Compound antegrade andretrograde intussusceptions are very rare and there hadbeen no documentation in the English literature ofsuch variety in an African child. This report is aimed atestablishing the fact that while it is true that ileo-colicintussusception is the commonest form in children, ahigh index of suspicion should be developed byclinicians in looking out for rare forms as in our indexcase. We report the case of  an 11-month-old Nigerianmanaged on account of double compoundintussusception and reviewed relevant literature.CASE PRESENTATIONAn 11 month old boy was referred with a five dayhistory of passage of watery stools, postprandialvomiting and a four day history of progressiveabdominal distension associated with intermittentdiscomfort and inconsolable crying. There wasassociated high grade fever and he passed red currant,jelly stools a few times before presentation. The patienthad an episode of upper respiratory tract infection,which lasted a few days, two weeks prior topresentation.Examination revealed an acutely ill child who waslethargic, febrile, pale and dehydrated. He also hadtachycardia (140/minute) and tachypnoea (44/minute).The abdomen was distended with visible peristalsisbut no generalized abdominal tenderness. A sausageshaped mass was palpated on the left hypochondriumwith positive sign of Dance (absence of loops ofbowels in the right half of the abdomen) and bowelsounds were hyperactive. There was no mass palpablein the rectum but the gloved finger was stained withmucoid blood stained faeces. A diagnosis of  acuteintestinal obstruction secondary to intussusception wasmade and the child was resuscitated.Plain abdominal radiographs showed dilated loops ofbowel with multiple air fluid levels. He was resuscitatedwith intravenous fluids, electrolyte, intravenousantibiotics and worked up for emergency exploratorylaparotomy. The findings at surgery were: a retrogradecolo-colic intussusception (proximal sigmoid colon intodescending colon); antegrade ileo-colic intussusceptionas far distally as the proximal descending colon;perforations at the neck of both intussusceptions withdevitalized bowel in between them and an area ofnecrotic bowel 10 cm from the neck of the ileo-colicA CASE OF COMPOUND INTUSSUSCEPTIONS IN A NIGERIAN CHILD – A RAREFINDING IN A COMMON DISEASEK.I. Egbuchulem1, T.A. Lawal1,, M.C. Nweke2 and A.O. Adeoye21. Division of  Paediatric Surgery, Department of  Surgery, University College Hospital Ibadan, Ibadan, Nigeria2. Department of  Pathology, University College Hospital, Ibadan, NigeriaCorrespondence:Dr. T.A. LawalDepartment of  Surgery,University College Hospital,PMB 5116, Ibadan 200212,Nigeria+2348069614811Email: taiwo.lawal@hotmail.comABSTRACTIntussusception is the most common, non-congenital, cause of intestinalobstruction in infants. There are various patterns of  intussusception seen inchildren with the most common being ileo-colic variety. Compoundintussusception in which intussusception involves more than one non-adjacentsegment is a rare finding in the literature and it can be associated with worsemorbidity than typically occurs, especially in a region where delayed presentationis a major contributor to morbidity and mortality in the treatment of intestinalobstruction.We report the first documented case of  double compound intussusception in anAfrican child and reviewed relevant literature.Keywords: Antegrade and retrograde; Children; Compound; Intussusception                                                   Annals of Ibadan Postgraduate Medicine. Vol. 15 No. 1, June 2017 57Ann Ibd. Pg. Med 2017. Vol.15, No.1 57-60CASE REPORTintussusception. There was minimal peritoneal soilageand about 50 millilitres of sero-fibrinous peritonealfluid was drained. He had resection of bothintussusceptions (Figure 1) and intervening devitalizedbowel with an end to end primary ileo-sigmoidanastomosis.colic) and ileum up to the proximal descending colon(ileo-colic). There was marked distortion of thearchitecture of the appendix and intussuscipiens withextensive suppurative necrosis, vascular congestion andstromal oedema. Sections of the intussusceptionsshowed glandular epithelium overlying markedlyhyperplastic lymphoid follicles with prominentgerminal centres. Overall features were in keeping withintussusceptions with reactive follicular hyperplasia(Figure 2).DISCUSSIONIntussusception or ‘‘introsusception’’ as it was calledthen, was first detailed in the literature in 1789 by JohnHunter4 but the first surgery for a child with thecondition was not reported until 1871 by Sir JonathanHutchinson.5 Intussusception, especially the idiopathicvariety, is primarily a disease of  childhood and is thecommonest cause of intestinal obstruction betweenthe ages of  three months and two years.1 Whileintussusception is quite common in childhood, doubleintussusception in which the invagination of bowelsegment into adjacent ones occur at two segments ofthe intestine is rarely seen.6 Double intussusceptionscan be one of  four varieties. The first type is one inwhich two separate segments prolapse into the samedistal loop of intestine, resulting in a characteristic“triple-circle” sign when sonography and computedtomography are performed.6 The second type, alsocalled double compound intussusception7 involvesboth antegrade and retrograde intussusception in thesame patient at the same time. This variety is extremelyrare7 and mimics our index patient. The third type isPostoperatively, he had oliguria, hypoproteinaemia andsepsis, but made good clinical progress and wasdischarged home.. He was followed up in theoutpatient clinic over a three month period and hadsustained clinical improvement.Histopathology examinations of  the resected proximalileum and caecum showed multiple intussusceptionsinvolving the sigmoid and descending colon (colo-Figure 1: Resected segment of ileum and colonFigure 2: Section of the resected segment of the intussuscepted bowel showing a markedly hyperplasticlymphoid follicle with prominent germinal centres (arrow).                                                   Annals of Ibadan Postgraduate Medicine. Vol. 15 No. 1, June 2017 58double prolapse of the proximal and distal intestinethrough a patent vitello-intestinal duct8, 9 while thefourth type is antegrade intussusception involving twodifferent sites on the bowel.10In view of the rare nature of compoundintussusception, there have been occasions when thesecond intussusception was found to have been missedat the time of surgery for the first intussusception eventhough both occurred synchronously.11 This may belinked to the rarity of compound intussusception andthe assumption in most instances that idiopathicintussusception of childhood is ileo-colic in location.11For intussusception to occur there must be bowelirritability and or a lead point. The aetiology ofintussusception in early childhood remains a dilemma,as the majority of intussusception has no definiteaetiological factor or demonstrable pathologic leadpoint10, 12-14 unlike in older children and adults.15 Wecould not identify a pathologic lead point in our patient,rather the histological examination of the resectedsegment of bowel revealed markedly hyperplasticlymphoid follicles with prominent germinal centressuggestive of  a secondary inflammatory response.Often, intussusception occurs during the time ofweaning and may have a seasonal pattern that coincideswith attacks of viral upper respiratory tract infection.The latter was noted in the presentation of the patient.The resected specimen also showed extensivelymphocytic infiltration, which is suggestive of  a viralaetiology.The major challenge in the treatment of intussusceptionin Africa is late presentation with associatedcomplications such as dehydration, electrolyteimbalance, sepsis, bowel gangrene with peritonitis andshock16, which were present in our patient. In a relatedstudy on peritonitis in children, Osifo et al in BeninCity, Nigeria, reported that management of  childhoodperitonitis in an African setting is challenging, becauseof late presentation, and intussusception accounted for7.1% of all the causes of secondary bacterial peritonitisin that study.17The management of intussusception in children in sub-Saharan Africa has not changed significantly andexploratory laparotomy is routinely performedfollowing adequate resuscitation for such cases.18 Ourpatient who also had exploratory laparotomy and endto end bowel anastomosis was not an exemption. In arelated study, comparing the rate of  surgicalintervention between colo-colic and ileo-colicintussusceptions in paediatric age group, it wasobserved that both groups have similar incidence ofsurgical intervention (82%) while resection rate washigher in the ileo-colic group compared to the colo-colic group.19 In our patient, both intussusceptions wereresected, because there was clinical evidence ofgangrene and perforation, and an ileo-sigmoidanastomosis done.CONCLUSIONAlthough, ileo-colic intussusception is the commonestpattern of intussusception in children, doublecompound intussusception is a rare disease ofchildhood that if  not diagnosed early, may lead toextensive bowel loss. Intussusception is a paediatricsurgical emergency hence, the onus lies on clinicians tolook out for them and manage them effectively as thiscould affect the outcome.This study was carried out in the Departments ofSurgery and Histopathology, University CollegeHospital, Ibadan.REFERENCES1. Ogundoyin OO, Afolabi AO, Ogunlana DI, etal. Pattern and outcome of childhood intestinalobstruction at a Tertiary Hospital in Nigeria.African Health Sciences 2009; 9(3):170–173.2. Talabi AO, Sowande OA, Etonyeaku CA andAdejuyigbe O. Childhood intussusceptions in Ile-Ife: What has changed? Afr J Paediatr Surg 2013;10:239–242.3. Tandoh JFK. Intussusception. In: Badoe EA,Archampong EQ, Da-Rocha-Afodu JT. Principlesand practice of  Surgery in the tropics. Ghanapublishing corporation Accra: 2000. 3rd edition.Chapter 34, p.540–543.4. Hunter J. On introsusception (read Aug 18, 1789).In: Palmer JF, ed. The Works of  John Hunter,FRS London: Longman, Rees, Orme, Brown,Green, Longman, 1837:587–593.5. Hutchinson J. A successful case of abdominalsection for intussusception. Proc R Med Chir Soc1873; 7:195–198.6. Kazez A, Ozel SK, Kocakoc E and Kiris A.Double intussusceptions in a child: the triplecircle sign. J Ultrasound Med 2004; 23:1659–1661.7. Him FP, Weng YK and Hoi CW. A case ofdouble compound intussusception in an infant.Singapore Med J 1980; 21:540–541.8. Benson JM, Sparnon AL. Double intussusceptionof ileum through a patent vitello intestinal duct:report of  a case and literature review. Aust N Z JSurg 1992; 62: 411–413.9. Mustafa R. Double intussusception of the smallbowel through a patent vitello-intestinal duct. Br JSurg 1976; 63:452.10. Kiyan G, Tugtepe H, Iskit SH and Dagli TE.Double intussusception in an infant. J Pediatr Surg2002; 37: 1643–1644.                                                   Annals of Ibadan Postgraduate Medicine. Vol. 15 No. 1, June 2017 5911. Yi-Hsin C, Guan-Yeu D, Chih-Hau C, et al.Double site intussusception in a four-year-old girl.J Med Science 2006; 26(5):191–194.12. Kuremu RT. Childhood intussusceptions at theMoi Teaching and Referral Hospital, Eldoret:Management challenges in a rural setting. E AfrMed J 2004; 81: 443–446.13. Carneiro PMR and Kususi DM. Intussusceptionin children seen at Muhimbili National Hospital,Dar Es Salaam. E Afr Med J 2004; 81: 439–442.14. Edino ST, Ochicha O, Mohammed AZ andAnumah M. Intussusception in Kano: a 5 yearanalysis of  pattern, morbidity and mortality. NigerJ Med 2003; 12: 221–224.15. Agha FP. Intussusception in adult. AJR Am JRoentgenol 1986; 146 (3): 527–531.16. Gudeta B. Intussusception in children: A ten yearreview. E Afr Med J 1993; 70:730–731.17. Osifo OD, Ogiemwonyi SO. Peritonitis inchildren: Our experience in Benin City, Nigeria.Surg Infect (Larchmt) 2011; 12 (2): 127–130.18. Carneiro PMR Editorial. Intussusception in thepaediatric age group. E Afr Med J 2004; 81: 437–438.19. Grant HW, Baccimazza I, Hadley GP. Acomparism of colo-colic and ileo-colicintussusception. J Pediatr Surg 1996; 31(12):1607–1610.                                                   Annals of Ibadan Postgraduate Medicine. Vol. 15 No. 1, June 201 60          This is an open access article licensed under the terms of the Creative Commons Attribution Non-Commercial License(<uri xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">http://creativecommons.org/licenses/by-nc/3.0</uri>)which permits unrestricted, non-commercial use, distribution and reproduction in any medium, provided the work is properly cited

A case of compound intussusceptions in a Nigerian child – a rare finding in a common disease

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A case of compound intussusceptions in a Nigerian child – a rare finding in a common disease

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