Maria Oliva-Hemker1, Susan Hutfless2, Elie S Al Kazzi2, Trudy Lerer3, David Mack4, Neal LeLeiko5, Anne Griffiths6, Jose Cabrera7, Anthony Otley8, James Rick9, Athos Bousvaros10, Joel Rosh11, Andrew Grossman12, Shehzad Saeed13, Marsha Kay14, Ryan Carvalho15, David Keljo16, Marian Pfefferkorn17, William Faubion18, Michael Kappelman19, Boris Sudel20, Marc E Schaefer21, James Markowitz22, Jeffrey S Hyams3. 1. Division of Pediatric Gastroenterology and Nutrition, Johns Hopkins Children's Center, Baltimore, MD. Electronic address: moliva@jhmi.edu. 2. Johns Hopkins University, Baltimore, MD. 3. Connecticut Children's Medical Center, Hartford, CT. 4. Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada. 5. Hasbro Children's Hospital, Providence, RI. 6. Hospital for Sick Children, Toronto, Ontario, Canada. 7. Medical College of Wisconsin, Milwaukee, WI. 8. Izaak Walton Killam Health Center, Halifax, Nova Scotia, Canada. 9. Dayton Children's Hospital, Dayton, OH. 10. Children's Hospital, Boston, MA. 11. Goryeb Children's Hospital, Morristown, NJ. 12. Children's Hospital of Philadelphia, Philadelphia, PA. 13. Children's Hospital Medical Center, Cincinnati, OH. 14. Cleveland Clinic, Cleveland, OH. 15. Nationwide Children's Hospital, Columbus, OH. 16. Children's Hospital of Pittsburgh, Pittsburgh, PA. 17. Riley Hospital for Children, Indianapolis, IN. 18. Mayo Clinic, Rochester, MN. 19. University of North Carolina, Chapel Hill, NC. 20. University of Minnesota, Minneapolis, MN. 21. Penn State Hershey Children's Hospital, Hershey, PA. 22. Cohen Children's Medical Center of New York, New Hyde Park, NY.
Abstract
OBJECTIVE: To evaluate the presentation, therapeutic management, and long-term outcome of children with very early-onset (VEO) (≤ 5 years of age) inflammatory bowel disease (IBD). STUDY DESIGN: Data were obtained from an inception cohort of 1928 children with IBD enrolled in a prospective observational registry at multiple centers in North America. RESULTS: One hundred twelve children were ≤ 5 years of age with no child enrolled at <1 year of age. Of those, 42.9% had Crohn's disease (CD), 46.4% ulcerative colitis (UC), and 10.7% had IBD-unclassified. Among the children with CD, children 1-5 years of age had more isolated colonic disease (39.6%) compared with 6- to 10-year-olds (25.3%, P = .04), and 11- to 16-year-olds (22.3%, P < .01). The change from a presenting colon-only phenotype to ileocolonic began at 6-10 years. Children 1-5 years of age with CD had milder disease activity (45.8%) at diagnosis compared with the oldest group (28%, P = .01). Five years postdiagnosis, there was no difference in disease activity among the 3 groups. However, compared with the oldest group, a greater proportion of 1- to 5-year-olds with CD were receiving corticosteroids (P < .01) and methotrexate (P < .01), and a greater proportion of 1- to 5-year-olds with UC were receiving mesalamine (P < .0001) and thiopurine immunomodulators (P < .0002). CONCLUSIONS: Children with VEO-CD are more likely to have mild disease at diagnosis and present with a colonic phenotype with change to an ileocolonic phenotype noted at 6-10 years of age. Five years after diagnosis, children with VEO-CD and VEO-UC are more likely to have been administered corticosteroids and immunomodulators despite similar disease activity in all age groups. This may suggest development of a more aggressive disease phenotype over time.
OBJECTIVE: To evaluate the presentation, therapeutic management, and long-term outcome of children with very early-onset (VEO) (≤ 5 years of age) inflammatory bowel disease (IBD). STUDY DESIGN: Data were obtained from an inception cohort of 1928 children with IBD enrolled in a prospective observational registry at multiple centers in North America. RESULTS: One hundred twelve children were ≤ 5 years of age with no child enrolled at <1 year of age. Of those, 42.9% had Crohn's disease (CD), 46.4% ulcerative colitis (UC), and 10.7% had IBD-unclassified. Among the children with CD, children 1-5 years of age had more isolated colonic disease (39.6%) compared with 6- to 10-year-olds (25.3%, P = .04), and 11- to 16-year-olds (22.3%, P < .01). The change from a presenting colon-only phenotype to ileocolonic began at 6-10 years. Children 1-5 years of age with CD had milder disease activity (45.8%) at diagnosis compared with the oldest group (28%, P = .01). Five years postdiagnosis, there was no difference in disease activity among the 3 groups. However, compared with the oldest group, a greater proportion of 1- to 5-year-olds with CD were receiving corticosteroids (P < .01) and methotrexate (P < .01), and a greater proportion of 1- to 5-year-olds with UC were receiving mesalamine (P < .0001) and thiopurine immunomodulators (P < .0002). CONCLUSIONS:Children with VEO-CD are more likely to have mild disease at diagnosis and present with a colonic phenotype with change to an ileocolonic phenotype noted at 6-10 years of age. Five years after diagnosis, children with VEO-CD and VEO-UC are more likely to have been administered corticosteroids and immunomodulators despite similar disease activity in all age groups. This may suggest development of a more aggressive disease phenotype over time.
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Authors: Jodie Ouahed; Elizabeth Spencer; Daniel Kotlarz; Dror S Shouval; Matthew Kowalik; Kaiyue Peng; Michael Field; Leslie Grushkin-Lerner; Sung-Yun Pai; Athos Bousvaros; Judy Cho; Carmen Argmann; Eric Schadt; Dermot P B Mcgovern; Michal Mokry; Edward Nieuwenhuis; Hans Clevers; Fiona Powrie; Holm Uhlig; Christoph Klein; Aleixo Muise; Marla Dubinsky; Scott B Snapper Journal: Inflamm Bowel Dis Date: 2020-05-12 Impact factor: 5.325