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  • Open Access
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Infant progressive colonic stenosis caused by antibiotic-related Clostridium difficile colitis – a case report and literature review

BMC Pediatrics201818:320

https://doi.org/10.1186/s12887-018-1302-9

  • Received: 22 August 2017
  • Accepted: 3 October 2018
  • Published:
Open Peer Review reports

Abstract

Background

Colonic stenosis is a rare cause of pediatric intestinal obstruction. The root cause underlying colonic stenosis is unclear and there is no fixed operation.

Case presentation

We reported on a male infant with progressive colonic stenosis caused by antibiotic-related colitis. The infant was admitted to our hospital with pneumonia but developed progressive abdominal distension and diarrhea following antibiotic treatment with meropenem. Initial testing of stool culture showed a Clostridium difficile infection. Additional testing with barium enema imaging showed stenosis at the junction of the sigmoid and descending colon at first and another stenosis occurred at the right half of the transverse colon 3 weeks later. Staged surgical treatment was performed with primary resections of the two parts suffering stenosis, ileostomy, and secondary intestinal anastomosis. A pathological exam then confirmed the diagnosis of colonic stenosis and the patient had an uneventful recovery and has been recovering well as evidenced by the 1-year follow-up.

Conclusions

Based on a review of the literature and our case report, we found that progressive colonic stenosis caused by colitis due to antibiotic-related Clostridium difficile infection is rare in infants. Infants with colitis and repeated abdominal distention, vomiting, and constipation should be treated with the utmost caution and screened. Despite this, clinical manifestations depended on the severity of the stenosis. Barium enema, colonoscopy, laprascopy or laparotomy and colonic biopsy are helpful for diagnosis and differential diagnosis. While both one-stage and multiple-stage operations are feasible, a staged operation should be used for multiple colonic stenoses.

Keywords

  • Colonic stenosis
  • Clostridium difficile
  • Colonitis

Background

Colonic stenosis is a rare cause of pediatric intestinal obstruction. The root cause underlying colonic stenosis is unclear. Both congenital and acquired colonic stenoses (e.g post-necrotizing enterocolitis) has been reported on [129]. Here we present an infant case of progressive colonic stenosis after antibiotic-related Clostridium difficile colitis and a review of the related literature, which to the best of our knowledge had not been reported previously.

Case presentation

This male infant was the second child of a 39-year-old mother and was born via cesarean section during the 38th week of the pregnancy with a birth weight of 3300 g. The infant was admitted to our hospital 10 days after birth due to pneumonia and was treated with meropenem. He developed abdominal distension and diarrhea gradually from the 10th day of therapy on and stool culture revealed a Clostridium difficile infection. This was considered to be antibiotic-related and oral metronidazole and vancomycin were given. His symptoms were soon resolved but after discharge he gradually developed abdominal distension and constipation. A barium enema exam on the 42nd day after birth showed stenosis at the junction of the sigmoid and descending colon and a distended proximal bowel (Fig. 1a). Abdominal distension and constipation became more severe after 3 weeks of conservative treatment. A second barium enema exam then revealed another stenosis of the right transverse colon in addition to the previous stenosis (Fig. 1b).
Fig. 1
Fig. 1

a Stenosis at the junction of the sigmoid and descending colon at the first barium enema. c No stenosis at the hepatic flexure at the first barium enema. b and d Stenoses at the junction of the sigmoid and descending colon and right transverse colon at the second barium enema

Primary surgical exploration revealed two segments of stenoses. One was at the junction of the sigmoid and descending colon and was 3.5 cm in length, while the other one was at the right transverse colon and was 4 cm in length. The small intestine, however, was still intact. Both the two parts were resected and an ileostomy was conducted at the terminal ileum. A pathological exam showed fibrosis of lamina propria in the narrow segments. Ganglion cells were normal (Fig. 2a and b). Closure of ileostomy was performed 3 months later and he made uneventful recovery. At the 1-year follow-up, he exhibited a normal dietary intake and defecation. His state of growth and development was in the 70th percentile.
Fig. 2
Fig. 2

a Fibrosis of lamina propria of the narrow segment. b Normal ganglion cells

Discussion and conclusions

Intestinal obstruction caused by colonic stenosis is rare in children. We reviewed literature relating to pediatric colonic stenoses since 1961 (Tables 1 and 2) [129]. Pathogenesis of pediatric colonic stenoses varied among patients but congenital stenosis featured prominently among the literature. George Ekema and Reyes C introduced cases with congenital cytomegavirus (CMV) infection which involved the gastrointestinal tract and finally developed into colonic stenosis [30, 31]. Many researchers accepted the theory that fetal intestinal injury in the uterus due to disturbance in the blood supply was key. The causes of ischemia included emboli originating in the placenta, fetal herniation, kinks, intussusceptions, drugs (particularly cocaine) and placental causes [7, 10, 14, 16, 3234]. Some colonic stenoses were secondary to necrotizing enterocolitis (NEC) [1729], which were the most common type of non-congenital colon stenoses. The present case did not have complications during the pregnancy and perinatal period though. TORCH exam of the child and the mother was also negative. Colonic stenoses developed after antibiotic-related cololitis caused by Clostridium difficile progressively, which was confirmed by two barium exams. Sahara K and Kawaratani H reported that it is adult inflammatory bowel disease that causes colonic stenosis, and stool culture suggests Clostridium difficile [35, 36], but in our case the underlying disease wasn’t present. This patient did have a history of intestinal infection of Clostridium difficile prior to onset of symptoms though. The second barium exam showed a new site of stenosis compared to the first barium exam. This evidence shows that stenoses occurred secondary to infection rather than being congenital.
Table 1

Literature review of infant congenital colonic stenosis from 1961 to 2016

Authors

Year

Age

Localization

Surgical approach

Length of Stenosis

Zambaiti et al. [1]

2016

2 m

Ascending and transverse

II

Not described

Saha et al. [2]

2013

1.5Y

Descending

I

Not described

Galván-Montaño et al. [3]

2010

3Y

Ascending

I

5.0 cm

Ruggeri et al. [4]

2009

4 m

Ascending

Not described

Mizuno et al. [5]

2003

Newborn

Descending

I

Not described

García Vázquez et al. [6]

2002

2 m

Sigmoid

I

Not described

Abu-Judeh et al. [7]

2001

Ascending

I

4.0 cm

Dalla Vecchia et al. [8]

1998

Newborn

Not described

Not described

Newborn

Not described

Not described

Murphree et al. [9]

1992

Newborn

Sigmoid

I

Not described

Sax [10]

1991

Descending-sigmoid

II

Not described

Pai GK and Pai PK [11]

1990

4 m

Rectosigmoid junction

I

Not described

Rescorla and Grosfeld [12]

1985

Not described

II

Not described

Newborn

Sigmoid

II

9.5 cm

Schiller et al. [13]

1979

Newborn

Descending

II

3.0 cm

Newborn

Sigmoid

II

4.0 cm

Erskine [14]

1970

2 d

Descending-sigmoid

II

16.0 cm

Benson et al. [15]

1968

Sigmoid

I

Not described

SANTULLI and BLANC [16]

1961

Sigmoid

II

Not described

I: Resection of stenotic segment and primary anastomosis II: A staged approach

Table 2

Literature review of colonic stenosis secondary to necrotizing enterocolitis treated by surgery

Authors

Year

patient

Localization of colon

Surgical approach

Marseglia L et al. [17]

2015

1 case

sigmoid

I

28 cases with 46 stenoses

5 ascending

I

Gaudin A et al. [18]

2013

including 32 colonic stenosis

7 transverse

I

 

20 descending

I

 

1 whole colon

II

Pelizzo G et al. [19]

2013

3 cases

1 transverse

II

 

1 right

II

Martinez-Ferro M et al. [20]

2009

11 cases

I

 

10 right colon

I

Baudon JJ et al. [21]

1997

15 cases with 26 stenoses

5 transverse

I

 

11 left colon

I

Vilariño Mosquera A et al. [22]

1995

15 cases

I

Schimpl G et al. [23]

1994

21 cases

I

Gobet R et al. [24]

1994

22 cases

I

Radhakrishnan J et al. [25]

1991

9 cases

I

D’Agostino S et al. [26]

1988

1 case

1 sigmoid

I

Schwartz MZ et al. [27]

1982

7 cases

7 left colon

I

Kosloske AM et al. [28]

1980

6 cases

II

Bonte C et al. [29]

1977

3 cases

3 sigmoid

I

I: Resection of stenotic segment and primary anastomosis II: A staged approach

It has been suggested that infants who have had abdominal distension, vomiting, and constipation should be suspected to suffer from colon stenosis. The barium enema was important in the diagnosis colonic stenosis in this case as it could determine the site of obstruction and severity of stenosis. However, a colonoscopy would be an alternative method to help with diagnosis [6]. The major differential diagnosis was Hirschsprung’s disease confirmed by pathological exam. In humans and other mammals, both domestic and wild, Clostridium difficile takes hold of the large intestine. While toxigenic and nontoxigenic strains do exist, toxigenic forms are responsible for causing disease in humans. Toxin A (TcdA) and toxin B (TcdB), are two closely related diarrhea-causing toxins and their presence is seen is a cause of pathogenicity. TcdB is found in all toxigenic strains, regardless of whether TcdA occurs concurrently. In addition to this, inactivation of Rho GTPases through enzymatic glucosylation of a conserved threonine residue is a similar molecular mechanism of action found in both of these toxins. Most often actin depolymerization and cell death follow, and the mechanism leads to the stimulation of an inflammatory cascade, with the end result being tissue damage, diarrhea, and pseudomembranous colitis [37, 38]. Moreover, significantly correlated with this tissue damage, diarrhea, and pseudomembranous colitis was the occurrence of a progression to fibrosis at the lamina propria.

Surgery is the major treatment of colon stenosis (Tables 1 and 2). For stenoses in both the right and left half of the colon, resection and primary anastomosis or proximal diversion could be successfully performed [129]. Pelizzo G reported three cases of colonic stenoses with norovirus infection in preterm babies. All patients received primary ileostomy followed by an immediate or staged coloectomy. Proximal diversion of intestinal contents is recommended to help to preserve colon integrity [19]. In our patient, primary resections of strictures of the colon with proximal diversion had successfully preserved the rest of the colon. This was important as the colonic stenoses was proved to be progressive in this case. The main reason why we chose to perform ileostomy rather than colon anastomosis was due to the fact of the colon stenosis being progressive. We didn’t know whether new stenosis would occur. Barium enema imaging before enterostomy didn’t reveal another colonic stenosis and the patient had an uneventful recovery followed by a clean check of health at the 1-year follow-up.

Based on a review of the literature and our case report, we found that progressive colonic stenosis caused by colitis due to antibiotic-related Clostridium difficile infection is rare in infants. Infants with colitis and repeated abdominal distention, vomiting, and constipation should be suspected and screened. Clinical manifestations depended on the severity of the stenosis. Barium enema, colonoscopy, laprascopy or laparotomy and colonic biopsy are helpful for diagnosis and differential diagnosis. Both one-stage surgery and multiple-stage operations are feasible, however staged operation should be used for multiple colonic stenoses.

Abbreviations

CMV: 

Congenital cytomegavirus

NEC: 

Necrotizing enterocolitis

Declarations

Availability of data and materials

All data generated or analyzed during this study are included in this published article.

Authors’ contributions

Study conception and design: XJ. Data acquisition: XX, YW, YZ, QW. Analysis and data interpretation: BX, XJ. Drafting of the manuscript: XX. Critical revision: XJ. All authors read and approved the final manuscript.

Ethics approval and consent to participate

This study was approved by the Institutional Review Board and Ethical Committee at the West China Hospital of Sichuan University in China with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Consent for publication

Written informed consent was obtained from the patient’s parents to publish this case report and any accompanying images. This material is original research. It has not been previously published and has not been submitted for publication elsewhere while under consideration.

Competing interests

The authors declare that they have no competing interests.

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Authors’ Affiliations

(1)
Department of Pediatric Surgery, West China Hospital of Sichuan University, Chengdu, Sichuan Province, China

References

  1. Elisa Z, Cinzia C, Sergio S, Giuseppe LV, Fortunato S. Multiple congenital colonic stenosis:a rare gastrointestinal malformation. Case Rep Pediatr. 2016:6329793. Published online 2016 Mar 15. https://doi.org/10.1155/2016/6329793.View ArticleGoogle Scholar
  2. Saha N, Shezote T, Alam S. Congenital stenosis in the descending colon causing intestinal obstruction in a one and half years male child. Mymensingh Med J. 2013;22(3):574–7.PubMedGoogle Scholar
  3. Galván-Montaño A, Suárez-Roa Mde L, Carmona-Moreno E. Congenital stenosis of the colon with foreign bodies. Case report. Cir Cir. 2010;78(3):259–61.PubMedGoogle Scholar
  4. Ruggeri G, Libri M, Gargano T, Pavia S, Pasini L, Tani G, Lima M. Congenital colonic stenosis: a case of late-onset. Pediatr Med Chir. 2009;31(3):130–3.PubMedGoogle Scholar
  5. Mizuno M, Kato T, Hebiguchi T, Yoshino H. Congenital membranous colonic stenosis-case report of an extremely rare anomaly. J Pediatr Surg. 2003;38(8):E13–5.View ArticleGoogle Scholar
  6. García Vázquez A, Cano Novillo I, Portela Casalod E, Benavent Gordo MI, Berchi García FJ. Congenital colonic stenosis. An Esp Pediatr. 2002;56(3):258–60.View ArticleGoogle Scholar
  7. Abu-Judeh HH, Methratta S, Ybasco A, Garrow E, Ali S. Congenital colonic stenosis. South Med J. 2001;94(3):344–6.View ArticleGoogle Scholar
  8. Dalla Vecchia LK, Grosfeld JL, West KW, Rescorla FJ, Scherer LR, Engum SA. Intestinal atresia and stenosis: a 25-year experience with 277 cases. Arch Surg. 1998;133(5):490–6.View ArticleGoogle Scholar
  9. Murphree SM, Dunkley AS. Colon atresia and stenosis in Zimbabwe: case reports and a review of the literature. Cent Afr J Med. 1992;38(12):463–5.PubMedGoogle Scholar
  10. Sax EJ. Pediatric case of the day. Congenital colonic stenosis. Am J Roentgenol. 1991;156(6):1315–7.View ArticleGoogle Scholar
  11. Pai GK, Pai PK. A case of congenital colonic stenosis presenting as rectal prolapse. J Pediatr Surg. 1990;25(6):699–700.View ArticleGoogle Scholar
  12. Rescorla FJ, Grosfeld JL. Intestinal atresia and stenosis:analysis of survival in 120 cases. Surgery. 1985;98(4):668–76.PubMedGoogle Scholar
  13. Schiller M, Aviad l, Freund H. Congenital colonic atresia and stenosis. Am J Surg. 1979;138(5):721–4.View ArticleGoogle Scholar
  14. Erskine JM. Colonic stenosis in the newborn: the possible thromboembolic etiology of intestinal stenosis and atresia. J Pediatr Surg. 1970;5(3):321–33.View ArticleGoogle Scholar
  15. Benson CD, Lotfi MW, Brouch AJ. Congenital atresia and stenosis of the colon. J Pediatr Surg. 1968;3(2):253–7.View ArticleGoogle Scholar
  16. Santulli TV, Blanc WA. Congenital atresia of the intestine: pathogenesis and treatment. Ann Surg. 1961;154(6):939–48.PubMedPubMed CentralGoogle Scholar
  17. Marseglia L, Manti S, D’Angelo G, Lima M, Impellizzeri P, Romeo C, Gitto E. Colonic stenosis post-necrotizing enterocolitis in term newborn with acquired cytomegalovirus infection. Chirurgia (Bucur). 2015;110(2):175–8.Google Scholar
  18. Gaudin A, Farnoux C, Bonnard A, Alison M, Maury L, Biran V, Baud O. Necrotizing enterocolitis (NEC) and the risk of intestinal stricture: the value of C-reactive protein. PLoS One. 2013;8(10):e76858.View ArticleGoogle Scholar
  19. Pelizzo G, Nakib G, Goruppi I, Fusillo M, Scorletti F, Mencherini S, Parigi GB, Stronati M, Calcaterra V. Isolated colon ischemia with norovirus infection in preterm babies: a case series. J Med Case Rep. 2013;7:108.View ArticleGoogle Scholar
  20. Martinez-Ferro M, Rothenberg S, St. Peter S, Bignon H, Holcomb G. Laparoscopic treatment of postnecrotizing enterocolitis colonic strictures. J Laparoendosc Adv Surg Tech A. 2010;20(5):477–80.View ArticleGoogle Scholar
  21. Baudon JJ, Josset P, Audry G, Benlagha N, Fresco O. Intestinal stenosis during ulceronecrotizing enterocolitis. Arch Pediatr. 1997;4(4):305–10.View ArticleGoogle Scholar
  22. Vilariño Mosquera A, CanoNovillo I, Benavent Gordo I, Jiménez MA, Delga Manzanares M, Barrios C, Berchi García FJ. Retrospective analysis of 80 cases of neonatal necrotizing enterocolitis. Cir Pediatr. 1995;8(4):148–50.PubMedGoogle Scholar
  23. Schimpl G, Höllwarth ME, Fotter R, Becker H. Late intestinal strictures following successful treatment of necrotizing enterocolitis. Acta Paediatr Suppl. 1994;396:80–3.View ArticleGoogle Scholar
  24. Gobet R, Sacher P, Schwöbel MG. Surgical procedures in colonic strictures after necrotizing enterocolitis. Acta Paediatr Suppl. 1994;396:77–9.View ArticleGoogle Scholar
  25. Radhakrishnan J, Blechman G, Shrader C, Patel MK, Mangurten HH, McFadden JC. Colonic strictures following successful medical management of necrotizing enterocolitis: a prospective study evaluating early gastrointestinal contrast studies. J Pediatr Surg. 1991;26(9):1043–6.View ArticleGoogle Scholar
  26. D’Agostino S, Stracca-Pansa V, Drei F, Valli F, Colombo B, Guarise P. Post-necrotizing enterocolitis stenosis of the colon associated with cytomegalovirus infection. Description of a clinical case. Pediatr Med Chir. 1988;10(6):637–9.PubMedGoogle Scholar
  27. Schwartz MZ, Keith Hayden C, Joan Richardson C, Tyson KRT, Lobe TE. A prospective evaluation of intestinal stenosis following necrotizing enterocolitis. J Pediatr Surg. 1982;17(6):764–70.View ArticleGoogle Scholar
  28. Kosloske AM, Burstein J, Bartow SA. Intestinal obstruction due to colonic stricture following neonatal necrotizing enterocolitis. Ann Surg. 1980;192(2):202–7.View ArticleGoogle Scholar
  29. Bonte C, Barberis D, Maillard E, Rousselle B, Dervaux D. Colonic stenosis following necrotizing enterocolitis in newborn infants. Apropos of 3 cases. Arch Fr Pediatr. 1977;34(7):622–31.PubMedGoogle Scholar
  30. Ekema G, Pedersini P, Milianti S, Ubertazzi M, Minoli D, Manciana A. Colonic stricture mimicking Hirschsprung’s disease: a localized cytomegalovirus infection. J Pediatr Surg. 2006;41(4):850–2.View ArticleGoogle Scholar
  31. Reyes C, Pereira S, Warden MJ, Sills J. Cytomegalovirus enteritis in a premature infant. J Pediatr Surg. 1997;32(11):1545–7.View ArticleGoogle Scholar
  32. Powell RW, Raffensperger JG. Congenital colonic atresia. J Pediatr Surg. 1982;17(2):166–70.View ArticleGoogle Scholar
  33. Louw JH, Barnard CN. Congenital intestinal atresia:observation on its origin. Lancet. 1955;269(6899):1065–7.View ArticleGoogle Scholar
  34. Hall TR, Zaninovi A, Lewin D, Barrett C, Boechat MI. Neonatal intestinal ischemia with bowl perforation:an in utero complication of maternal cocaine abuse. AJR Am J Roentgenol. 1992;158(6):1303–4.View ArticleGoogle Scholar
  35. Sahara K, Yamada R, Fujiwara T, Koizumi K, Horiguchi S-i, Hishima T, Yamaguchi T. Idiopathic myointimal hyperplasia of mesenteric veins: rare case of ischemic colitis mimicking inflammatory bowel disease. Dig Endosc. 2015;27(7):767–70.View ArticleGoogle Scholar
  36. Kawaratani H, Tsujimoto T, Toyohara M, Kin K, Taniguchi T, Shirai Y, Ikenaka Y, Nakayama M, Fujii H, Fukui H. Pseudomembranous colitis complicating ulcerative colitis. Dig Endosc. 2010;22(4):373–5.View ArticleGoogle Scholar
  37. Vedantam G, Clark A, Chu M, McQuade R, Mallozzi M, Viswanathan VK. Clostridium difficile infection: toxins and non-toxin virulence factors, and their contributions to disease establishment and host response. Gut Microbes. 2012;3(2):121–34.View ArticleGoogle Scholar
  38. Pothoulakis C, Lamont JT. Microbes and microbial toxins: paradigms for microbial-mucosal interactions II. The integrated response of the intestine to Clostridium difficile toxins. Am J Physiol Gastrointest Liver Physiol. 2001;280(2):G178–83.View ArticleGoogle Scholar

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© The Author(s). 2018

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