Skip to main content

Advertisement

SpringerLink
Log in

Increasing rate of a positive family history of inflammatory bowel disease (IBD) in pediatric IBD patients

  • Original Article
  • Published:
European Journal of Pediatrics Aims and scope Submit manuscript

Abstract

The role of a positive family history in pediatric inflammatory bowel disease (IBD) in the era of biologic therapy has not been elucidated. We retrospectively reviewed the medical records of children with IBD and retrieved demographic and clinical characteristics, including the presence of a positive family history of IBD, IBD phenotype, disease course, and therapy. Overall, 325 children (age range at diagnosis 11–15 years) were included, of whom 82 (25.2%) had a positive family history. Children diagnosed during 2016–2020 had a higher frequency of positive family history compared to those diagnosed during 2010–2015 (31.8% versus 20.7%, respectively, p = 0.024). Children with a positive family history had a higher risk for a stricturing phenotype than those with a negative family history (11.3% versus 2.8%, respectively, p = 0.052). They more often received nutritional therapy (53.7% versus 36.6%, p = 0.007) and less often received corticosteroids (36.6% versus 52.7%, p = 0.012). More children with a negative family history needed intensification of biologic therapy (p = 0.041).

Conclusion: The rate of a positive family history of IBD in the pediatric IBD population is increasing. A positive family history may have some impact upon IBD phenotype but none on IBD outcome.

What is Known:

•Familial clustering of inflammatory bowel disease (IBD) has been reported in 5%-15% of IBD patients.

•The investigation of the impact of a positive family history upon IBD characteristics and severity revealed conflicting results.

What is New:

•In this cohort of 325 children with IBD, 25.2% had a positive family history.

•The rate of a positive family history of IBD in the pediatric IBD population is increasing.

•A positive family history may have some impact upon IBD phenotype but none on IBD outcome.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

Abbreviations

CD:

Crohn’s disease

EEN:

Exclusive enteral nutrition

ESPGHAN:

European Society for Pediatric Gastroenterology Hepatology and Nutrition

IBD:

Inflammatory bowel disease

IQR:

Interquartile range

PCDAI:

Pediatric Crohn’s Disease Activity Index

PUCAI:

Pediatric Ulcerative Colitis Activity Index

UC:

Ulcerative colitis

References

  1. Ordás I, Eckmann L, Talamini M, Baumgart DC, Sandborn WJ (2012) Ulcerative colitis. Lancet 380(9853):1606–1619

    Article  Google Scholar 

  2. Baumgart DC, Sandborn WJ (2012) Crohn’s disease. The Lancet 380(9853):1590–1605

    Article  Google Scholar 

  3. Halfvarson J, Bodin L, Tysk C, Lindberg EV, Järnerot G (2003) Inflammatory bowel disease in a Swedish twin cohort: a long-term follow-up of concordance and clinical characteristics. Gastroenterology 124(7):1767–1773

    Article  Google Scholar 

  4. Stittrich AB, Ashworth J, Shi M, Robinson M, Mauldin D, Brunkow ME, Biswas S, Kim JM, Kwon KS, Jung JU, Galas D (2016) Genomic architecture of inflammatory bowel disease in five families with multiple affected individuals. Hum Genome Var 3(1):1–9

    Article  Google Scholar 

  5. Halme L, Paavola-Sakki P, Turunen U, Lappalainen M, Farkkila M, Kontula K (2006) Family and twin studies in inflammatory bowel disease. World J Gastroenterol 12:3668–3672

    Article  Google Scholar 

  6. Bernstein CN, Rawsthorne P, Cheang M, Blanchard JF (2006) A population-based case control study of potential risk factors for IBD. Am J Gastroenterol 101(5):993–1002

    Article  Google Scholar 

  7. Orholm M, Munkholm P, Langholz E, Nielsen OH, Sørensen TI, Binder V (1991) Familial occurrence of inflammatory bowel disease. N Engl J Med 324:84–88

    Article  CAS  Google Scholar 

  8. Strisciuglio C, Giugliano F, Martinelli M, Cenni S, Greco L, Staiano A, Miele E (2017) Impact of environmental and familial factors in a cohort of pediatric patients with inflammatory bowel disease. J Pediatr Gastroenterol Nutr 64(4):569–574

    Article  Google Scholar 

  9. Brant SR (2011) Update on the heritability of inflammatory bowel disease: the importance of twin studies. Inflamm Bowel Dis 17:1–5

    Article  Google Scholar 

  10. Henriksen M, Jahnsen J, Lygren I, Vatn MH, Moum B; IBSEN Study Group (2007) Are there any differences in phenotype or disease course between familial and sporadic cases of inflammatory bowel disease? Results of a population-based follow-up study. Am J Gastroenterol 102:1955–1963

    Article  Google Scholar 

  11. Kuwahara E, Asakura K, Nishiwaki Y, Inoue N, Watanabe M, Hibi T, Takebayashi T (2012) Effects of family history on inflammatory bowel disease characteristics in Japanese patients. J Gastroenterol 47(9):961–968

    Article  CAS  Google Scholar 

  12. Weinstein TA, Levine M, Pettei MJ, Gold DM, Kessler BH, Levine JJ (2003) Age and family history at presentation of pediatric inflammatory bowel disease. J Pediatr Gastroenterol Nutr 37(5):609–613

    Article  Google Scholar 

  13. Roma ES, Panayiotou J, Pachoula J, Constantinidou C, Polyzos A, Zellos A, Lagona E, Mantzaris GJ, Syriopoulou VP (2010) Inflammatory bowel disease in children: the role of a positive family history. Eur J Gastroenterol Hepatol 22(6):710–715

    PubMed  Google Scholar 

  14. Hwang SW, Kwak MS, Kim WS, Lee JM, Park SH, Lee HS, Yang DH, Kim KJ, Ye BD, Byeon JS, Myung SJ (2016) Influence of a positive family history on the clinical course of inflammatory bowel disease. J Crohns Colitis 10(9):1024–1032

    Article  Google Scholar 

  15. Orlanski-Meyer E, Aardoom M, Ricciuto A, Navon D, Carman N, Aloi M, Bronsky J, Däbritz J, Dubinsky M, Hussey S, Lewindon P, Martin De Carpi J, Navas-López VM, Orsi M, Ruemmele FM, Russell RK, Veres G, Walters TD, Wilson DC, Kaiser T, de Ridder L, Griffiths A, Turner D (2021) Predicting outcomes in pediatric ulcerative colitis for management optimization: systematic review and consensus statements from the Pediatric Inflammatory Bowel Disease-Ahead Program. Gastroenterology 160(1):378–402

    Article  CAS  Google Scholar 

  16. Borren NZ, Conway G, Garber JJ, Khalili H, Budree S, Mallick H, Yajnik V, Xavier RJ, Ananthakrishnan AN (2018) Differences in clinical course, genetics, and the microbiome between familial and sporadic inflammatory bowel diseases. J Crohns Colitis 12(5):525–531

    Article  Google Scholar 

  17. Childers RE, Eluri S, Vazquez C, Weise RM, Bayless TM, Hutfless S (2014) Family history of inflammatory bowel disease among patients with ulcerative colitis: a systematic review and meta-analysis. J Crohns Colitis 8(11):1480–1497

    Article  Google Scholar 

  18. Levine A, Koletzko S, Turner D, Escher JC, Cucchiara S, de Ridder L, Kolho KL, Veres G, Russell RK, Paerregaard A, Buderus S, Greer ML, Dias JA, Veereman-Wauters G, Lionetti P, Sladek M, Martin de Carpi J, Staiano A, Ruemmele FM, Wilson DC (2014) ESPGHAN revised porto criteria for the diagnosis of inflammatory bowel disease in children and adolescents. J Pediatr Gastroenterol Nutr 58:795–806

    Article  CAS  Google Scholar 

  19. Levine A, Griffiths A, Markowitz J, Wilson DC, Turner D, Russell RK, Fell J, Ruemmele FM, Walters T, Sherlock M, Dubinsky M, Hyams JS (2011) Pediatric modification of the Montreal classification for inflammatory bowel disease: the Paris classification. Inflamm Bowel Dis 17:1314–1321

    Article  Google Scholar 

  20. Turner D, Griffiths AM, Walters TD, Seah T, Markowitz J, Pfefferkorn M, Keljo D, Otley A, Leleiko NS, Mack D, Hyams J, Levine A (2010) Appraisal of the pediatric Crohn’s disease activity index on four prospectively collected datasets: recommended cutoff values and clinimetric properties. Am J Gastroenterol 105:2085–2092

    Article  Google Scholar 

  21. Turner D, Otley AR, Mack D, Hyams J, de Bruijne J, Uusoue K, Walters TD, Zachos M, Mamula P, Beaton DE, Steinhart AH, Griffiths AM (2007) Development, validation, and evaluation of a pediatric ulcerative colitis activity index: a prospective multicenter study. Gastroenterology 133:423–432

    Article  Google Scholar 

  22. Schiff ER, Frampton M, Semplici F, Bloom SL, McCartney SA, Vega R, Lovat LB, Wood E, Hart AL, Crespi D, Furman MA, Mann S, Murray CD, Segal AW, Levine AP (2018) A new look at familial risk of inflammatory bowel disease in the Ashkenazi Jewish population. Dig Dis Sci 63(11):3049–3057

    Article  Google Scholar 

  23. Andreu M, Márquez L, Domènech E, Gisbert JP, García V, Marín-Jiménez I, Peñalva M, Gomollón F, Calvet X, Merino O, Garcia-Planella E, Vázquez-Romero N, Esteve M, Nos P, Gutiérrez A, Vera I, Cabriada JL, Martín MD, Cañas-Ventura A, Panés J (2014) Disease severity in familial cases of IBD. J Crohns Colitis 8(3):234–239

    Article  CAS  Google Scholar 

  24. Wu J, Lubman DM, Kugathasan S, Denson LA, Hyams JS, Dubinsky MC, Griffiths AM, Baldassano RN, Noe JD, Rabizadeh S, Gulati AS, Rosh JR, Crandall WV, Higgins PDR, Stidham RW (2019) Serum protein biomarkers of fibrosis aid in risk stratification of future stricturing complications in pediatric Crohn’s disease. Am J Gastroenterol 114(5):777–785

    Article  Google Scholar 

  25. Ben-Horin S, Avidan B, Yanai H, Lang A, Chowers Y, Bar-Meir S (2009) Familial clustering of Crohn’s disease in Israel: prevalence and association with disease severity. Inflamm Bowel Dis 15:171–175

    Article  Google Scholar 

  26. Cohen S, Padlipsky J, Yerushalmy-Feler A (2020) Risk factors associated with extraintestinal manifestations in children with inflammatory bowel disease. Eur J Clin Nutr 74(5):691–697

    Article  CAS  Google Scholar 

  27. Sahu P, Kedia S, Vuyyuru SK, Bajaj A, Markandey M, Singh N, Singh M, Kante B, Kumar P, Ranjan M, Sahni P, Panwar R, Sharma R, Das P, Makharia G, Travis SPL, Ahuja V (2021) Randomised clinical trial: exclusive enteral nutrition versus standard of care for acute severe ulcerative colitis. Aliment Pharm Ther 53:568–576

    CAS  Google Scholar 

  28. Shaoul R, Brown S, Day A (2018) Reasoning beyond the potential use of exclusive enteral nutrition and other specified diets in children with ulcerative colitis. J Pediatr Gastroenterol Nutr 66(3):378–382

    Article  Google Scholar 

  29. Trier Moller F, Andersen V, Andersson M, Jess T (2015) Hospital admissions, biological therapy, and surgery in familial and sporadic cases of inflammatory bowel disease: a population-based cohort study 1977–2011. Inflamm Bowel Dis 20:2825–2832

    Article  Google Scholar 

  30. Kelley-Quon LI, Jen HC, Ziring DA, Gupta N, Kirschner BS, Ferry GD, Cohen SA, Winter HS, Heyman MB, Gold BD, Shew SB (2012) Predictors of proctocolectomy in children with ulcerative colitis. J Pediatr Gastroenterol Nutr 55:534–540

    Article  Google Scholar 

  31. Carbonnel F, Macaigne G, Beaugerie L, Gendre JP, Cosnes J (1999) Crohn’s disease severity in familial and sporadic cases. Gut 44:91–95

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Pediatric Gastroenterology Unit, Dana-Dwek Children’s Hospital, Tel Aviv Sourasky Medical Center, affiliated to the Sackler Faculty of Medicine, Tel Aviv University, 6 Weizmann Street, 6423906, Tel Aviv, Israel

    Maya Ruban, Adam Slavick, Achiya Amir, Amir Ben-Tov, Hadar Moran-Lev, Yael Weintraub, Adi Anafy, Shlomi Cohen & Anat Yerushalmy-Feler

Authors
  1. Maya Ruban
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Adam Slavick
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Achiya Amir
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Amir Ben-Tov
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Hadar Moran-Lev
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Yael Weintraub
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Adi Anafy
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Shlomi Cohen
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Anat Yerushalmy-Feler
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

AYF, MR, AS, and SC designed the study, contributed to the data used in the study, searched the literature, interpreted the data, and wrote the manuscript. AA, HML, ABT, YW, and AA contributed to the data used in this article and to the discussion and reviewed and edited the manuscript. All of the authors approved the final version.

Corresponding author

Correspondence to Shlomi Cohen.

Ethics declarations

Ethics approval

This article does not contain any studies with human participants or animals performed by any of the authors.

Consent to participate

N/A

Consent for publication

N/A

Informed consent

N/A

Conflict of interest

The authors declare no competing interests.

Additional information

Communicated by Peter de Winter

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Ruban, M., Slavick, A., Amir, A. et al. Increasing rate of a positive family history of inflammatory bowel disease (IBD) in pediatric IBD patients. Eur J Pediatr 181, 745–751 (2022). https://doi.org/10.1007/s00431-021-04269-8

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00431-021-04269-8

Keywords

Search

Navigation