Natural History of Very Early Onset Inflammatory Bowel Disease in North America: A Retrospective Cohort Study

被引：25

作者：

Kerur, Basavaraj ^{[1
]}

Benchimol, Eric, I ^{[2
]}

Fiedler, Karoline ^{[3
]}

Stahl, Marisa ^{[4
]}

Hyams, Jeffrey ^{[5
]}

Stephens, Michael ^{[6
]}

Lu, Ying ^{[7
]}

Pfefferkorn, Marian ^{[8
]}

Alkhouri, Raza ^{[9
]}

Strople, Jennifer ^{[10
]}

Kelsen, Judith ^{[11
]}

Siebold, Leah ^{[12
]}

Goyal, Alka ^{[13
]}

Rosh, Joel R. ^{[14
]}

LeLeiko, Neal ^{[15
]}

Van Limbergen, Johan ^{[16
]}

Guerrerio, Anthony L. ^{[17
]}

Maltz, Ross ^{[18
]}

Karam, Lina ^{[19
]}

Crowley, Eileen ^{[28
]}

Griffiths, Anne ^{[3
]}

Heyman, Melvin B. ^{[20
]}

Deneau, Mark ^{[21
]}

Benkov, Keith ^{[22
]}

Noe, Joshua ^{[23
]}

Mouton, Dedrick ^{[24
]}

Pappa, Helen ^{[25
]}

Galanko, Joseph A. ^{[26
]}

Snapper, Scott ^{[27
]}

Muise, Aleixo M. ^{[3
]}

Kappelman, Michael D. ^{[26
]}

机构：

[1] Univ Massachusetts, Med Sch UMMS, Worcester, MA 01655 USA

[2] Childrens Hosp Eastern Ontario, Ottawa, ON, Canada

[3] Hosp Sick Children, Toronto, ON, Canada

[4] Childrens Hosp Colorado, Denver, CO USA

[5] Connecticut Childrens Med Ctr, Hartford, CT USA

[6] Mayo Clin, Ctr Individualized Med, Rochester, MN USA

[7] Cohen Childrens Med Ctr New York, New York, NY USA

[8] Riley Childrens Hosp, Indianapolis, IN USA

[9] Univ Buffalo, Buffalo, NY USA

[10] Ann & Robert H Lurie Childrens Hosp Chicago, Chicago, IL 60611 USA

[11] Univ Penn, Childrens Hosp Philadelphia, Perelman Sch Med, Philadelphia, PA 19104 USA

[12] UPMC Childrens Hosp Pittsburgh, Pittsburgh, PA USA

[13] Childrens Mercy, Kansas City, MO USA

[14] Goryeb Childrens Hosp, Atlantic Hlth Syst, Morristown, NJ USA

[15] Hasbro Childrens Hosp, Providence, RI USA

[16] Univ Amsterdam, Emma Childrens Hosp, Med Ctr, Amsterdam, Netherlands

[17] Johns Hopkins Univ, Sch Med, Baltimore, MD USA

[18] Nationwide Childrens Med Ctr, Columbus, OH USA

[19] Texas Childrens Hosp, Houston, TX 77030 USA

[20] UCSF Benioff Childrens Hosp, San Francisco, CA USA

[21] Univ Utah, Intermt Primary Childrens Hosp, Salt Lake City, UT USA

[22] Icahn Sch Med Mt Sinai, New York, NY 10029 USA

[23] Med Coll Wisconsin, Milwaukee, WI 53226 USA

[24] Monroe Carell Jr Childrens Hosp Vanderbilt, Nashville, TN USA

[25] Cardinal Glennon Childrens Hosp, St Louis, MO USA

[26] Univ N Carolina, Chapel Hill, NC 27515 USA

[27] Childrens Hosp Boston, Boston, MA USA

[28] Univ Western Ontario, Childrens Hosp, London Hlth Sci Ctr, London, ON, Canada

来源：

INFLAMMATORY BOWEL DISEASES | 2021年 / 27卷 / 03期

关键词：

VEOIBD; surgery; epidemiology; CHILDREN; AGE; PHENOTYPE; IBD;

D O I：

10.1093/ibd/izaa080

中图分类号：

R57 [消化系及腹部疾病];

学科分类号：

摘要：

Background: The incidence of very early onset inflammatory bowel disease (VEOIBD) is increasing, yet the phenotype and natural history of VEOIBD are not well described. Methods: We performed a retrospective cohort study of patients diagnosed with VEOIBD (6 years of age and younger) between 2008 and 2013 at 25 North American centers. Eligible patients at each center were randomly selected for chart review. We abstracted data at diagnosis and at 1, 3, and 5 years after diagnosis. We compared the clinical features and outcomes with VEOIBD diagnosed younger than 3 years of age with children diagnosed with VEOIBD at age 3 to 6 years. Results: The study population included 269 children (105 [39%] Crohn's disease, 106 [39%] ulcerative colitis, and 58 [22%] IBD unclassified). The median age of diagnosis was 4.2 years (interquartile range 2.9-5.2). Most (94%) Crohn's disease patients had inflammatory disease behavior (B1). Isolated colitis (L2) was the most common disease location (70% of children diagnosed younger than 3 years vs 43% of children diagnosed 3 years and older; P = 0.10). By the end of follow-up, stricturing/penetrating occurred in 7 (6.6%) children. The risk of any bowel surgery in Crohn's disease was 3% by 1 year, 12% by 3 years, and 15% by 5 years and did not differ by age at diagnosis. Most ulcerative colitis patients had pancolitis (57% of children diagnosed younger than 3 years vs 45% of children diagnosed 3 years and older; P = 0.18). The risk of colectomy in ulcerative colitis/IBD unclassified was 0% by 1 year, 3% by 3 years, and 14% by 5 years and did not differ by age of diagnosis. Conclusions: Very early onset inflammatory bowel disease has a distinct phenotype with predominantly colonic involvement and infrequent stricturing/penetrating disease. The cumulative risk of bowel surgery in children with VEOIBD was approximately 14%-15% by 5 years. These data can be used to provide anticipatory guidance in this emerging patient population.

引用

页码：295 / 302

页数：8

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