Double-site intussusception in an infant: A case report with review of literature

Rahul Gupta; Monish Raj; Arvind Kumar Shukla; Ravindra Sevar

doi:10.4103/ipcares.ipcares_258_22

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CASE REPORT WITH REVIEW OF LITERATURE

Year : 2023 | Volume : 3 | Issue : 3 | Page : 150-153

Double-site intussusception in an infant: A case report with review of literature

Rahul Gupta, Monish Raj, Arvind Kumar Shukla, Ravindra Sevar
Department of Paediatric Surgery, SMS Medical College, Jaipur, Rajasthan, India

Date of Submission	02-Nov-2022
Date of Decision	28-Jun-2023
Date of Acceptance	30-Jun-2023
Date of Web Publication	14-Aug-2023

Correspondence Address:
Dr. Rahul Gupta
Associate Professor, Department of Paediatric Surgery, SMS Medical College, Jaipur, Rajasthan
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ipcares.ipcares_258_22

Abstract

Background: Although intussusception is a well-known phenomenon in childhood, double-site intussusception is rarely encountered. We report an interesting case of idiopathic double-site, two ileoileal intussusceptions. Clinical Description: An 11-month-old baby girl presented with acute-onset vomiting and abdominal distension over 3 days. There was no associated history of fever, respiratory tract infection, or gastroenteritis. The infant was otherwise healthy, thriving well, without any significant medical or surgical illness in the past. On examination, the infant was alert, but irritable, hemodynamically stable, with abdominal distension without any lump or organomegaly. Management: The infant was stabilized with supportive care initially. The abdominal radiograph revealed multiple air–fluid levels with a paucity of distal gas shadows. Ultrasound showed a single-target sign in the right lumbar region. At laparotomy, an ileoileal intussusception was detected. During manual reduction, another intussusception was identified; the distal one was also ileoileal at the terminal ileum with its apex at the ileocecal junction. Both intussusceptions were successfully reduced. No pathological lead point was observed for each intussusception. Symptoms resolved, and there was no recurrence. Conclusion: This case creates awareness about the rare occurrence of double-site intussusceptions, which are not always detected by imaging methods. A careful inspection of the entire gut intraoperatively is essential to avoid missing such unsuspected second intussusceptions.

Keywords: Idiopathic, infant, intestinal obstruction, pediatric

How to cite this article:
Gupta R, Raj M, Shukla AK, Sevar R. Double-site intussusception in an infant: A case report with review of literature. Indian Pediatr Case Rep 2023;3:150-3

How to cite this URL:
Gupta R, Raj M, Shukla AK, Sevar R. Double-site intussusception in an infant: A case report with review of literature. Indian Pediatr Case Rep [serial online] 2023 [cited 2023 Sep 26];3:150-3. Available from: http://www.ipcares.org/text.asp?2023/3/3/150/383623

Intussusception is a fairly well-known phenomenon, common among children, especially in those below 2 years of age,^[1],[2] majority being ileocolic.^[1],[2] Double-site intussusception is rare.^{[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16]} In this report, we present a case of double-site intussusception in an infant without any pathological lead point. We also present a review of literature of previously reported pediatric cases of double-site intussusceptions.

Clinical Description

An 11-month-old baby girl presented with complaints of bouts of pain with high-pitched cry, vomiting, and abdominal distension for the last 72 h. There was no antecedent history of fever, cough, coryza, feeding intolerance, or loose stools. The vomitus was initially nonbilious and contained curdled milk, later became bilious. There was no history of blood in stools. The infant was not passing stools for the last 3 days and had diminished oral acceptance for the same duration. The patient had received vaccination against measles, mumps, and rubella, 2 months back. There was no significant past medical or surgical illness in the past.

The infant was born full-term with a birth weight of 2.5 kg. This first born child was a product of a non-consanguineous marriage; her antenatal, natal, and postnatal periods were uneventful. She was on the second stage of weaning with a varied diet alongside the usual breast milk. The patient had received vaccination as per the Universal Immunization Programme (UIP). The patient did not receive rotavirus vaccination (not included in the UIP of the state) in the past.

At presentation, the infant weighed 7.5 kg, with a length of 72 cm. She was normothermic (temperature 98.8°F), with well-felt peripheral pulses, heart rate of 122/min, capillary filling time of <2 s, and respiratory rate of 40/min. General physical examination revealed mild pallor, without any cyanosis, icterus, clubbing, edema, or subcutaneous fat wasting.

The baby was alert but irritable. Abdominal examination revealed mild abdominal distension, without any fullness of flanks. The skin over the abdomen was normal, with the umbilicus being central and flat in appearance. There was no venous prominence. On palpation, the temperature of the abdominal wall was normal and there was tenderness in the right iliac fossa. There was neither palpable lump nor any organomegaly. Peristaltic sounds were present. Cardiovascular, respiratory, and neurological systems appeared clinically normal. On per rectal examination, the rectum was empty and mucus staining of the finger was present.

Management and Outcome

The infant was hospitalized and admitted to the intensive care unit. Supportive care in the form of nil per mouth with intravenous fluids, nasogastric aspiration, and antibiotics (third-generation cephalosporin, aminoglycoside, and metronidazole) was initiated. The nasogastric aspirates were bilious in nature. Baseline blood examination showed anemia (hemoglobin of 9.1 g/dL), total leukocyte count of 9500/cumm, with polymorphs 82%, lymphocytes 15%, and platelets 2.5 lakhs/cumm. Her blood urea was 17 mg/dl and serum creatinine was 0.8 mg/dL. She had a deranged coagulation profile (international normalized ratio 1.51) and hyponatremia (Na 121 mEq/L), with normal serum potassium (4.2 mEq/L) and normal liver enzymes (alanine transaminase 36 units/L and aspartate transaminase 40 units/L). Abdominal radiographs revealed multiple air–fluid levels with paucity of distal gas shadows. Ultrasound showed a single-target sign in the right lumbar region with dilated proximal ileal loops (fluid filled with to-and-fro appearance) suggestive of ileoileal intussusception.

Because of the delayed presentation of ileoileal intussusception, surgical correction was attempted. At laparotomy, an ileoileal (mid-ileal portion) intussusception was detected. During manual (milking) reduction, it was noted that two separate intussusceptions were present [Figure 1]. The proximal one was 15 cm in length. The distal one (4–5 cm in length) was also ileoileal, nontransient intussusception at the terminal ileum with its apex at the ileocecal junction (impending ileo-ileocecal compound intussusception). The intussusceptions, seen approximately 30 cm apart, were reduced separately without much difficulty. No pathological lead point was for any of the intussusceptions. The gut wall in both parts was healthy. The infant recovered and started enteral feeds, without any recurrence. The infant is under follow-up and is asymptomatic and growing well.

Figure 1: Intraoperative photographs showing (a) double-site intussusception with proximal mid-ileoileal intussusception (green arrow), terminal ileoileal intussusception (blue arrow) and cecum (red arrow); (b-d) manual reduction of proximal mid-ileoileal intussusception (green arrow); (e) terminal ileoileal intussusception (blue arrow) up to ileocecal junction

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Discussion with Review of Literature

The above case depicts an interesting picture of double-site intussusception, diagnosed only after laparotomy, which was managed successfully without any complications.

Intussusception usually refers to the telescoping of the proximal intestinal segment into the adjacent distal segment.^[1],[2] Although intussusception is a common entity in children, predominantly in children <2 years of age, with an incidence of 1.5–4/1000 children, simultaneous occurrence of intussusceptions in two different parts of the small intestine is rare. Moreover, the absence of any leading points or congenital anomalies makes the phenomenon even more rare.^{[1],[2],[3],[4]}

Double intussusception, better named as double-site intussusception, is a different entity from compound intussusception. While double-site intussusception is the presence of two separately existing intussusceptions with some length of normal bowel present between them,^{[3],[4],[5],[6]} compound intussusception is where an intussusception itself acts as a lead point and is the intussusceptum of a second intussusception. Although there is not one definition for double compound intussusception, it is a combination of anterograde and retrograde intussusception or double anterograde.^[1],[7],[9]

Single-site intussusceptions are usually idiopathic, rarely associated with a lead point, such as Meckel's diverticulum, polyps, intestinal duplications, mesenteric cysts, intramural hematoma, and lymphoma.^{[8],[9],[10],[11],[12],[13]} Of the 15 cases of double-site intussusception described by Yu et al., 5 were idiopathic, 8 showed lead points, and 2 developed postoperatively.^[14] No lead point was identified in our case.

Less than 20 cases with double-site simultaneous intussusception have been reported in the pediatric age group [Table 1].^{[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17]} The age of presentation varied from the neonatal age of 1 day–16 years, with an almost similar distribution among females and males. Interestingly, the 8-year-old girl with multiple small-bowel intussusceptions with chronic gastrointestinal symptoms, reported by Hillier et al., was found to be suffering from cystic fibrosis, confirmed genetically.^[17] On the other hand, Chiew et al. found double-site intussusception to be associated with multiple large polyps of varying sizes, consistent with Peutz–Jeghers syndrome.^[16]

Table 1: Review of literature depicting demographic, clinical, management, and outcome details of double-site intussusception cases reported in children (including the present case)

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Clinically, double-site intussusception presents with abdominal pain, abdominal distension, vomiting, and bloody stools, similar to typical intussusception. It is usually impossible to diagnose double-site intussusception clinically.^{[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17]} On examination, the only useful sign is the presence of an abdominal mass, though it has been reported rarely, and none of them presented as double masses.^{[1],[2],[3],[12]} Plain abdominal radiograph is less sensitive and specific than ultrasonography for diagnosing of double-site intussusception. The main feature of double-site intussusceptions on ultrasonography is a double concentric circular sign. This sign may not be seen in all cases; most being diagnosed during surgery.^[5],[6] The second intussusception usually remains concealed owing to abdominal gaseous distention and lack of suspicion.^[7] Computed tomography may help in the diagnosis. However, the majority of the cases including ours were diagnosed intraoperatively.^{[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[15]}

Early surgical intervention is recommended for small-bowel involvement in double-site intussusceptions. Conservative management for double-site intussusception has also been reported.^[12],[16]

Surgical methods may be laparotomy or laparoscopy. At the laparotomy, a high index of suspicion should be present for the second intussusception. The locations of double-site intussusceptions in earlier reported pediatric cases were (a) jejunojejunal and ileocolic, (b) double ileoileal, (c) ileocolic and ileocecocolic, (d) ileocolic and colocolic, (e) double colocolic, and (f) ileoileal and ileocolic.^{[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17]} In our case, the intussusceptions involved the ileum (double ileoileal).

While idiopathic types are easily reducible manually, as seen in the present case, the presence of a lead point may make the reduction difficult. As no lead point or etiology could be identified in our case, the reason for the evolution of this entity and the chronological sequence of the two intussusceptions could not be ascertained. Except a few cases, most of the cases reported in literature have had a favorable outcome, similar to our case.

To conclude, by reporting this case, we draw the attention of pediatricians and pediatric surgeons toward this rare entity. The dual sites are not always detected by imaging methods. A high index of suspicion and careful intraoperative inspection of the entire bowel is essential to identify an unsuspected second-site intussusception.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given consent for images and other clinical information to be reported in the journal. The guardian understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Acknowledgment

We would also like to acknowledge: the parents of the baby, Department of Pediatrics, Paediatric Surgery, and Department of Radiodiagnosis, SMS Medical College, Jaipur, Rajasthan, India, for helping in our endeavor.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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