Diverting Ileostomy for the Treatment of Severe, Refractory, Pediatric Inflammatory Bowel Disease

Maxwell, Elizabeth C.; Dawany, Noor; Baldassano, Robert N.; Mamula, Petar; Mattei, Peter; Albenberg, Lindsey; Kelsen, Judith R.

doi:10.1097/mpg.0000000000001498

Cited by 27 publications

(10 citation statements)

References 22 publications

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“…8,11 In addition, the extent and location of disease can change and progress over time, and patients who present with exclusive colonic disease can later develop perianal and small bowel disease. 12 Therefore, indeterminate colitis is diagnosed more often in patients with VEO-IBD (11%-22%) as compared to older onset IBD (4%-10%). 9,[13][14][15] Because of this colonic involvement, patients with VEO-IBD are also more commonly diagnosed with UC (35%-59%) as compared to older onset IBD (older children > 6 and adults) in which CD is more prevalent (55%-60%).…”

Section: Disease Classificationmentioning

confidence: 99%

Genomic and Immunologic Drivers of Very Early-Onset Inflammatory Bowel Disease

Conrad

Kelsen

2019

Pediatr Dev Pathol

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Purpose of Review: Inflammatory bowel disease (IBD) is a multifactorial disease caused by dysregulated immune responses to commensal or pathogenic intestinal microbes, resulting in chronic intestinal inflammation. However, a subset of patients with IBD diagnosed <6 years of age, known as very early-onset (VEO)-IBD, can be phenotypically and genetically distinct from older onset IBD. We aim to review the clinical presentation of children with VEO-IBD and recent discoveries that point to the underlying genomic and immunologic drivers of disease, and the significant impact on our therapeutic decisions. Recent Findings: VEO-IBD is increasing in incidence and is associated with more severe disease, aggressive progression, and poor response to most conventional therapies. This article will review some of the genetic findings in this population and the subsequent impact on therapy, with targeted approaches. Summary: Children with VEO-IBD may present with a different phenotype and more severe disease than older children and adults. An integrated approach combining genetics, immunology, and traditional IBD evaluations can lead to the identification of causal defects that directly impact management. These strategies can also be employed in older onset refractory IBD.

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Section: Disease Classificationmentioning

confidence: 99%

Genomic and Immunologic Drivers of Very Early-Onset Inflammatory Bowel Disease

Conrad

Kelsen

2019

Pediatr Dev Pathol

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“…Fahy et al reported that in paediatric patients with severe refractory Crohn's colitis, total colectomy offers an opportunity to improve nutrition and growth and reduce steroid use with a reasonable likelihood of restoring intestinal continuity [18]. Maxwell et al reported that in paediatric patients with refractory colonic IBD, a diverting ileostomy can be a successful intervention to induce clinical stability [19]. In our study, the surgical approach allowed patients to decrease the use of metronidazole, thalidomide and other drugs.…”

Section: Discussionmentioning

confidence: 61%

Complications of enterostomy and related risk factor analysis of very early onset inflammatory bowel disease with interleukin-10 signalling deficiency: a single-centre retrospective analysis

et al. 2020

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Background Interleukin-10 (IL10) signalling pathway deficiency results in severe very early onset inflammatory bowel disease (VEOIBD), and enterostomy is often inevitable. However, studies in these surgical populations are lacking. This study aims to determine the enterostomy characteristics, postoperative complications and related risk factors in enterostomy patients. Methods From March 1, 2015, to December 31, 2018, patients with IL10R-mutation who underwent enterostomy were recruited for analysis. We collected data on the patients’ clinical characteristics, enterostomy characteristics, postoperative complications and related risk factors. Results Twelve patients required emergency enterostomy, and 10 patients underwent elective enterostomy. Twelve patients experienced postoperative complications, including wound infection (27.3%), wound dehiscence (18.2%), reoperation (18.2%), etc. Compared with the pre-enterostomy values, there was a decrease in C-reactive protein (CRP) (P = 0.001), an increase in albumin (P = 0.001) and an improvement in the weight-for-age (P = 0.029) and body mass index (BMI) Z-scores (P = 0.004) after enterostomy. There was a significant difference between the pre-operation and postoperation medicine expenses (P = 0.002). Univariate binary logistic regression analysis revealed a statistically significant influence of CRP (OR: 1.43, 95% CI: 1.07–1.91, P = 0.016) and a tendency towards a significant influence of intestinal perforation, albumin level, BMI Z-score and weighted paediatric Crohn’s disease activity index (wPCDAI). Multivariate logistic regression analysis showed that CRP (OR: 1.40), wPCDAI (OR: 2.88) and perforation (OR: 1.72) showed a tendency to behave as independent risk factors for postoperative complications, but the results were not significant (all P > 0.05). Conclusions Surgery and enterostomy showed benefits for VEOIBD with IL-10 signalling deficiency. The timing of intervention, potential postoperative complications, economic burden and other related problems should be considered.

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“…Some studies have reported higher surgical rates in children with VEO-IBD, especially those with infantile IBD [56,57]. However, the Canadian cohort of patients with VEO-IBD showed a low frequency of surgical interventions in the younger group than in the older pediatric group [2].…”

Section: Surgerymentioning

confidence: 95%

Very Early-Onset Inflammatory Bowel Disease: A Challenging Field for Pediatric Gastroenterologists

Arai

2020

Pediatr Gastroenterol Hepatol Nutr

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With the increasing number of children with inflammatory bowel disease (IBD), very earlyonset IBD (VEO-IBD), defined as IBD that is diagnosed or that develops before 6 years of age, has become a field of innovation among pediatric gastroenterologists. Advances in genetic testing have enabled the diagnosis of IBD caused by gene mutations, also known as monogenic or Mendelian disorder-associated IBD (MD-IBD), with approximately 60 causative genes reported to date. The diagnosis of VEO-IBD requires endoscopic and histological evaluations. However, satisfactory small bowel imaging studies may not be feasible in this small population. Both genetic and immunological approaches are necessary for the diagnosis of MD-IBD, which can differ among countries according to the available resources. As a result of the use of targeted gene panels covered by the national health insurance and the nationwide research project investigating inborn errors of immunity, an efficient approach for the diagnosis of MD-IBD has been developed in Japan. Proper management of VEO-IBD by pediatric gastroenterologists constitutes a challenge. Some MD-IBDs can be curable by allogenic hematopoietic stem cell transplantation. With an understanding of the affected gene functions, targeted therapies are being developed. Social and psychological support systems for both children and their families should also be provided to improve their quality of life. Multidisciplinary team care would contribute to early diagnosis, proper therapeutic interventions, and improved quality of life in patients and their families.

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Diverting Ileostomy for the Treatment of Severe, Refractory, Pediatric Inflammatory Bowel Disease

Abstract: In pediatric patients with refractory colonic IBD, diverting ileostomy can be a successful intervention to induce clinical stability. Importantly, diversion is a steroid-sparing therapy and allows additional time to clarify the diagnosis.

Cited by 27 publications

References 22 publications

Genomic and Immunologic Drivers of Very Early-Onset Inflammatory Bowel Disease

Genomic and Immunologic Drivers of Very Early-Onset Inflammatory Bowel Disease

Complications of enterostomy and related risk factor analysis of very early onset inflammatory bowel disease with interleukin-10 signalling deficiency: a single-centre retrospective analysis

Very Early-Onset Inflammatory Bowel Disease: A Challenging Field for Pediatric Gastroenterologists

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