Infantile and Very Early Onset Inflammatory Bowel Disease: A Multicenter Study: A Multicenter Study
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| Titel: | Infantile and Very Early Onset Inflammatory Bowel Disease: A Multicenter Study: A Multicenter Study |
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| Autoren: | Guz-Mark A., Aloi M., Scarallo L., Bramuzzo M., Escher J. C., Alvisi P., Henderson P., Hojsak I., Lev-Tzion R., El-Matary W., Schwerd T., Granot M., Sladek M., Strisciuglio C., Muller K. E., Olbjorn C., Tzivinikos C., Yerushalmy-Feler A., Huysentruyt K., Norsa L., Viola I., de Ridder L., Shouval D. S., Lega S., Lionetti P., Catassi G., Assa A. |
| Weitere Verfasser: | Brussels Photonics, Public Health Sciences, Pediatrics, Vitality Research Group |
| Quelle: | Pediatrics. 154 |
| Verlagsinformationen: | American Academy of Pediatrics (AAP), 2024. |
| Publikationsjahr: | 2024 |
| Schlagwörter: | Inflammatory Bowel Diseases/genetics, Male, 0301 basic medicine, Child, preschool, Adolescent, Infant, Crohn Disease/diagnosis, Inflammatory Bowel Diseases, Colitis, Ulcerative/diagnosis, 03 medical and health sciences, 0302 clinical medicine, Crohn Disease, Child, Preschool, Humans, Female, Colitis, Ulcerative, Age of Onset, Child, Retrospective Studies, Follow-Up Studies |
| Beschreibung: | OBJECTIVES This study described disease characteristics and long-term outcomes in patients diagnosed with very early onset inflammatory bowel disease (VEOIBD) (diagnosed before 6 years of age) and infantile-IBD (before 2 years). METHODS Cases from 21 centers worldwide diagnosed with VEOIBD (2008–2018), with minimum 2 years of follow-up, were retrospectively reviewed. RESULTS The cohort included 243 patients (52% males, median follow-up of 5.8 [range 2−18] years, including 69 [28%]) with infantile-IBD. IBD subtypes included Crohn’s disease (CD), ulcerative colitis (UC), or IBD-unclassified (IBDU) in 30%, 59%, and 11%, respectively. Among patients with CD, 94% had colonic involvement, and among patients with UC/IBDU, 75% had pancolitis. Patients with infantile-IBD presented with higher rates of IBDU, lower hemoglobin and albumin levels, and higher C-reactive protein, and had lower response rates to first-induction therapy and corticosteroids therapy (P < .05 for all). Colectomy and diversion surgeries were performed in 11% and 4%, respectively, with no significant differences between age groups. Corticosteroid-free remission rates were 74% and 78% after 3 and 5 years, respectively, and 86% at end of follow-up. Genetic testing was performed in 96 (40%) patients. Among tested population, 15 (16%) were identified with monogenic disease. This group demonstrated lower response rates to induction therapies, higher rates of surgical intervention, and higher rates of major infections (P < .05 for all). CONCLUSIONS Patients with VEOIBD, including infantile-IBD, exhibit low rate of complications and surgical interventions at the long term. Patients with monogenic IBD are at risk for more severe disease course. |
| Publikationsart: | Article |
| Sprache: | English |
| ISSN: | 1098-4275 0031-4005 |
| DOI: | 10.1542/peds.2023-064546 |
| Zugangs-URL: | https://pubmed.ncbi.nlm.nih.gov/39015095 https://pure.eur.nl/en/publications/0c6ac83f-5659-454f-9a14-84a509711221 https://doi.org/10.1542/peds.2023-064546 https://ruj.uj.edu.pl/handle/item/449655 https://biblio.vub.ac.be/vubir/(9abf878d-8bc2-48c6-9457-2efd54359225).html https://hdl.handle.net/11591/545622 https://doi.org/10.1542/peds.2023-064546 |
| Rights: | taverne |
| Dokumentencode: | edsair.doi.dedup.....2f34935b78a61985ef56a05562456671 |
| Datenbank: | OpenAIRE |
Nájsť tento článok vo Web of Science | Abstract: | OBJECTIVES This study described disease characteristics and long-term outcomes in patients diagnosed with very early onset inflammatory bowel disease (VEOIBD) (diagnosed before 6 years of age) and infantile-IBD (before 2 years). METHODS Cases from 21 centers worldwide diagnosed with VEOIBD (2008–2018), with minimum 2 years of follow-up, were retrospectively reviewed. RESULTS The cohort included 243 patients (52% males, median follow-up of 5.8 [range 2−18] years, including 69 [28%]) with infantile-IBD. IBD subtypes included Crohn’s disease (CD), ulcerative colitis (UC), or IBD-unclassified (IBDU) in 30%, 59%, and 11%, respectively. Among patients with CD, 94% had colonic involvement, and among patients with UC/IBDU, 75% had pancolitis. Patients with infantile-IBD presented with higher rates of IBDU, lower hemoglobin and albumin levels, and higher C-reactive protein, and had lower response rates to first-induction therapy and corticosteroids therapy (P < .05 for all). Colectomy and diversion surgeries were performed in 11% and 4%, respectively, with no significant differences between age groups. Corticosteroid-free remission rates were 74% and 78% after 3 and 5 years, respectively, and 86% at end of follow-up. Genetic testing was performed in 96 (40%) patients. Among tested population, 15 (16%) were identified with monogenic disease. This group demonstrated lower response rates to induction therapies, higher rates of surgical intervention, and higher rates of major infections (P < .05 for all). CONCLUSIONS Patients with VEOIBD, including infantile-IBD, exhibit low rate of complications and surgical interventions at the long term. Patients with monogenic IBD are at risk for more severe disease course. |
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| ISSN: | 10984275 00314005 |
| DOI: | 10.1542/peds.2023-064546 |