Skip to main content
Top
Published in: Pediatric Rheumatology 1/2022

Open Access 01-12-2022 | Disease Modifying Anti-Rheumatic Drug | Short Report

In a large Juvenile Idiopathic Arthritis (JIA) cohort, concomitant celiac disease is associated with family history of autoimmunity and a more severe JIA course: a retrospective study

Authors: Roberta Naddei, Simona Di Gennaro, Alfredo Guarino, Riccardo Troncone, Maria Alessio, Valentina Discepolo

Published in: Pediatric Rheumatology | Issue 1/2022

Login to get access

Abstract

Background

A higher prevalence of celiac disease (CD) has been reported in patients with juvenile idiopathic arthritis (JIA) compared to the general population. Factors related to the increased risk of co-occurrence and associated disease course have not been fully elucidated. Aims of this study were to determine the prevalence of CD in a large Southern Italian cohort of children with JIA, describe their clinical features and disease course and investigate risk factors associated with their co-occurrence.

Findings

Demographic, clinical and laboratory data of all patients with JIA admitted to our Pediatric Rheumatology Unit from January 2001 to June 2019, who underwent CD screening, were retrospectively extracted from clinical charts and analyzed. Eight of 329 JIA patients were diagnosed with CD, resulting in a prevalence higher than the general Italian population (2.4% vs 0.93%, p < 0.05). Familiarity for autoimmunity was reported by 87.5% patients with JIA and CD compared to 45.8% of those without CD (p < 0.05). 87.5% patients with JIA and CD required both a conventional Disease Modifying Anti-Rheumatic Drug (DMARD) and a biological DMARD over time compared to 36.4% of those without CD (p < 0.05).

Conclusion

A higher CD prevalence was found in a large JIA cohort, supporting the need for CD screening in all JIA children, especially those with a family history of autoimmunity, found to be associated with the co-occurrence of the two diseases. This is clinically relevant since patients with CD and JIA more often required a step-up therapy, suggesting a more severe JIA clinical course.
Appendix
Available only for authorised users
Literature
1.
go back to reference Sollid LM, Jabri B. Triggers and drivers of autoimmunity: lessons from coeliac disease. Nat Rev Immunol. 2013;13(4):294–302.CrossRef Sollid LM, Jabri B. Triggers and drivers of autoimmunity: lessons from coeliac disease. Nat Rev Immunol. 2013;13(4):294–302.CrossRef
2.
go back to reference Lepore L, Martelossi S, Pennesi M, Falcini F, Ermini ML, Ferrari R, et al. Prevalence of celiac disease in patients with juvenile chronic arthritis. J Pediatr. 1996;129(2):311–3.CrossRef Lepore L, Martelossi S, Pennesi M, Falcini F, Ermini ML, Ferrari R, et al. Prevalence of celiac disease in patients with juvenile chronic arthritis. J Pediatr. 1996;129(2):311–3.CrossRef
3.
go back to reference Stagi S, Giani T, Simonini G, Falcini F. Thyroid function, autoimmune thyroiditis and coeliac disease in juvenile idiopathic arthritis. Rheumatology (Oxford). 2005;44(4):517–20.CrossRef Stagi S, Giani T, Simonini G, Falcini F. Thyroid function, autoimmune thyroiditis and coeliac disease in juvenile idiopathic arthritis. Rheumatology (Oxford). 2005;44(4):517–20.CrossRef
4.
go back to reference Alpigiani MG, Haupt R, Parodi S, Calcagno A, Poggi E, Lorini R. Coeliac disease in 108 patients with juvenile idiopathic arthritis: a 13-year follow-up study. Clin Exp Rheumatol. 2008;26(1):162.PubMed Alpigiani MG, Haupt R, Parodi S, Calcagno A, Poggi E, Lorini R. Coeliac disease in 108 patients with juvenile idiopathic arthritis: a 13-year follow-up study. Clin Exp Rheumatol. 2008;26(1):162.PubMed
5.
go back to reference Pohjankoski H, Kautiainen H, Kotaniemi K, Korppi M, Savolainen A. Autoimmune diseases in children with juvenile idiopathic arthritis. Scand J Rheumatol. 2010;39(5):435–6.CrossRef Pohjankoski H, Kautiainen H, Kotaniemi K, Korppi M, Savolainen A. Autoimmune diseases in children with juvenile idiopathic arthritis. Scand J Rheumatol. 2010;39(5):435–6.CrossRef
6.
go back to reference Robazzi TC, Adan LF, Pimentel K, Guimarães I, Magalhães Filho J, Toralles MB, et al. Autoimmune endocrine disorders and coeliac disease in children and adolescents with juvenile idiopathic arthritis and rheumatic fever. Clin Exp Rheumatol. 2013;31(2):310–7.PubMed Robazzi TC, Adan LF, Pimentel K, Guimarães I, Magalhães Filho J, Toralles MB, et al. Autoimmune endocrine disorders and coeliac disease in children and adolescents with juvenile idiopathic arthritis and rheumatic fever. Clin Exp Rheumatol. 2013;31(2):310–7.PubMed
7.
go back to reference Moghtaderi M, Farjadian S, Aflaki E, Honar N, Alyasin S, Babaei M. Screening of patients with juvenile idiopathic arthritis and those with rheumatoid arthritis for celiac disease in southwestern Iran. Turk J Gastroenterol. 2016;27(6):521–4.CrossRef Moghtaderi M, Farjadian S, Aflaki E, Honar N, Alyasin S, Babaei M. Screening of patients with juvenile idiopathic arthritis and those with rheumatoid arthritis for celiac disease in southwestern Iran. Turk J Gastroenterol. 2016;27(6):521–4.CrossRef
8.
go back to reference Tronconi E, Miniaci A, Pession A. The autoimmune burden in juvenile idiopathic arthritis. Ital J Pediatr. 2017;43(1):56.CrossRef Tronconi E, Miniaci A, Pession A. The autoimmune burden in juvenile idiopathic arthritis. Ital J Pediatr. 2017;43(1):56.CrossRef
9.
go back to reference Skrabl-Baumgartner A, Christine Hauer A, Erwa W, Jahnel J. HLA genotyping as first-line screening tool for coeliac disease in children with juvenile idiopathic arthritis. Arch Dis Child. 2017;102(7):607–11.CrossRef Skrabl-Baumgartner A, Christine Hauer A, Erwa W, Jahnel J. HLA genotyping as first-line screening tool for coeliac disease in children with juvenile idiopathic arthritis. Arch Dis Child. 2017;102(7):607–11.CrossRef
10.
go back to reference Öman A, Hansson T, Carlsson M, Berntson L. Evaluation of screening for coeliac disease in children with juvenile idiopathic arthritis. Acta Paediatr. 2019;108(4):688–93.CrossRef Öman A, Hansson T, Carlsson M, Berntson L. Evaluation of screening for coeliac disease in children with juvenile idiopathic arthritis. Acta Paediatr. 2019;108(4):688–93.CrossRef
11.
go back to reference Sahin Y, Sahin S, Barut K, Cokugras FC, Erkan T, Adrovic A, et al. Serological screening for coeliac disease in patients with juvenile idiopathic arthritis. Arab J Gastroenterol. 2019;20(2):95–8.CrossRef Sahin Y, Sahin S, Barut K, Cokugras FC, Erkan T, Adrovic A, et al. Serological screening for coeliac disease in patients with juvenile idiopathic arthritis. Arab J Gastroenterol. 2019;20(2):95–8.CrossRef
12.
go back to reference Petty RE, Southwood TR, Manners P, Baum J, Glass DN, Goldenberg J, et al. International league of associations for rheumatology classification of juvenile idiopathic arthritis: second revision, Edmonton, 2001. J Rheumatol. 2004;31(2):390–2.PubMed Petty RE, Southwood TR, Manners P, Baum J, Glass DN, Goldenberg J, et al. International league of associations for rheumatology classification of juvenile idiopathic arthritis: second revision, Edmonton, 2001. J Rheumatol. 2004;31(2):390–2.PubMed
13.
go back to reference Husby S, Koletzko S, Korponay-Szabó IR, Mearin ML, Phillips A, Shamir R, et al. European society for pediatric gastroenterology, hepatology, and nutrition guidelines for the diagnosis of coeliac disease. J Pediatr Gastroenterol Nutr. 2012;54(1):136–60.CrossRef Husby S, Koletzko S, Korponay-Szabó IR, Mearin ML, Phillips A, Shamir R, et al. European society for pediatric gastroenterology, hepatology, and nutrition guidelines for the diagnosis of coeliac disease. J Pediatr Gastroenterol Nutr. 2012;54(1):136–60.CrossRef
14.
go back to reference Roberts SE, Morrison-Rees S, Thapar N, et al. Systematic review and meta-analysis: the incidence and prevalence of paediatric coeliac disease across Europe. Aliment Pharmacol Ther. 2021;00:1–20. Roberts SE, Morrison-Rees S, Thapar N, et al. Systematic review and meta-analysis: the incidence and prevalence of paediatric coeliac disease across Europe. Aliment Pharmacol Ther. 2021;00:1–20.
15.
go back to reference Al-Toma A, Volta U, Auricchio R, Castillejo G, Sanders DS, Cellier C, et al. European Society for the study of Coeliac Disease (ESsCD) guideline for coeliac disease and other gluten-related disorders. United European Gastroenterol J. 2019;7(5):583–613.CrossRef Al-Toma A, Volta U, Auricchio R, Castillejo G, Sanders DS, Cellier C, et al. European Society for the study of Coeliac Disease (ESsCD) guideline for coeliac disease and other gluten-related disorders. United European Gastroenterol J. 2019;7(5):583–613.CrossRef
16.
go back to reference Nisihara R, Skare T, Jardim AC, Utiyama SR. Celiac disease autoantibodies in juvenile idiopathic arthritis. Rheumatol Int. 2017;37(2):323–4.CrossRef Nisihara R, Skare T, Jardim AC, Utiyama SR. Celiac disease autoantibodies in juvenile idiopathic arthritis. Rheumatol Int. 2017;37(2):323–4.CrossRef
17.
go back to reference Lerner, et al. Rheumatoid arthritis–celiac disease relationship: Joints get that gut feeling. Autoimmun Rew. 2015;14(11):1038.CrossRef Lerner, et al. Rheumatoid arthritis–celiac disease relationship: Joints get that gut feeling. Autoimmun Rew. 2015;14(11):1038.CrossRef
18.
go back to reference Tighe MP, Beattie RM. Is HLA testing for coeliac disease helpful in associated autoimmune conditions? Arch Dis Child. 2017;102(7):595–6.CrossRef Tighe MP, Beattie RM. Is HLA testing for coeliac disease helpful in associated autoimmune conditions? Arch Dis Child. 2017;102(7):595–6.CrossRef
19.
go back to reference Hunt KA, Zhernakova A, Turner G, Heap GA, Franke L, Bruinenberg M, et al. Newly identified genetic risk variants for celiac disease related to the immune response. Nat Genet. 2008;40(4):395–402.CrossRef Hunt KA, Zhernakova A, Turner G, Heap GA, Franke L, Bruinenberg M, et al. Newly identified genetic risk variants for celiac disease related to the immune response. Nat Genet. 2008;40(4):395–402.CrossRef
20.
go back to reference Albers HM, Kurreeman FA, Stoeken-Rijsbergen G, Brinkman DM, Kamphuis SS, van Rossum MA, et al. Association of the autoimmunity locus 4q27 with juvenile idiopathic arthritis. Arthritis Rheum. 2009;60(3):901–4.CrossRef Albers HM, Kurreeman FA, Stoeken-Rijsbergen G, Brinkman DM, Kamphuis SS, van Rossum MA, et al. Association of the autoimmunity locus 4q27 with juvenile idiopathic arthritis. Arthritis Rheum. 2009;60(3):901–4.CrossRef
21.
go back to reference Ross SH, Cantrell DA. Signaling and function of interleukin-2 in T lymphocytes. Annu Rev Immunol. 2018;36:411–33.CrossRef Ross SH, Cantrell DA. Signaling and function of interleukin-2 in T lymphocytes. Annu Rev Immunol. 2018;36:411–33.CrossRef
22.
go back to reference Ciszewski C, Discepolo V, Pacis A, Doerr N, Tastet O, Mayassi T, et al. Identification of a γc receptor antagonist that prevents reprogramming of human tissue-resident cytotoxic T cells by IL15 and IL21. Gastroenterology. 2020;158(3):625-37.e13.CrossRef Ciszewski C, Discepolo V, Pacis A, Doerr N, Tastet O, Mayassi T, et al. Identification of a γc receptor antagonist that prevents reprogramming of human tissue-resident cytotoxic T cells by IL15 and IL21. Gastroenterology. 2020;158(3):625-37.e13.CrossRef
23.
go back to reference Thompson SD, Sudman M, Ramos PS, Marion MC, Ryan M, Tsoras M, et al. The susceptibility loci juvenile idiopathic arthritis shares with other autoimmune diseases extend to PTPN2, COG6, and ANGPT1. Arthritis Rheum. 2010;62(11):3265–76.CrossRef Thompson SD, Sudman M, Ramos PS, Marion MC, Ryan M, Tsoras M, et al. The susceptibility loci juvenile idiopathic arthritis shares with other autoimmune diseases extend to PTPN2, COG6, and ANGPT1. Arthritis Rheum. 2010;62(11):3265–76.CrossRef
24.
go back to reference Parkkola A, Laine AP, Karhunen M, Härkönen T, Ryhänen SJ, Ilonen J, et al. HLA and non-HLA genes and familial predisposition to autoimmune diseases in families with a child affected by type 1 diabetes. PLoS One. 2017;12(11):e0188402.CrossRef Parkkola A, Laine AP, Karhunen M, Härkönen T, Ryhänen SJ, Ilonen J, et al. HLA and non-HLA genes and familial predisposition to autoimmune diseases in families with a child affected by type 1 diabetes. PLoS One. 2017;12(11):e0188402.CrossRef
25.
go back to reference Li J, Yuan X, March ME, Yao X, Sun Y, Chang X, et al. Identification of target genes at Juvenile idiopathic arthritis GWAS loci in human neutrophils. Front Genet. 2019;10:181.CrossRef Li J, Yuan X, March ME, Yao X, Sun Y, Chang X, et al. Identification of target genes at Juvenile idiopathic arthritis GWAS loci in human neutrophils. Front Genet. 2019;10:181.CrossRef
26.
go back to reference Elli L, Discepolo V, Bardella MT, Guandalini S. Does gluten intake influence the development of celiac disease-associated complications? J Clin Gastroenterol. 2014;48(1):13–20.CrossRef Elli L, Discepolo V, Bardella MT, Guandalini S. Does gluten intake influence the development of celiac disease-associated complications? J Clin Gastroenterol. 2014;48(1):13–20.CrossRef
27.
go back to reference Nardecchia S, Auricchio R, Discepolo V, Troncone R. Extra-intestinal manifestations of coeliac disease in children: clinical features and mechanisms. Front Pediatr. 2019;7:56.CrossRef Nardecchia S, Auricchio R, Discepolo V, Troncone R. Extra-intestinal manifestations of coeliac disease in children: clinical features and mechanisms. Front Pediatr. 2019;7:56.CrossRef
28.
go back to reference Iagnocco A, Ceccarelli F, Mennini M, Rutigliano IM, Perricone C, Nenna R, et al. Subclinical synovitis detected by ultrasound in children affected by coeliac disease: a frequent manifestation improved by a gluten-free diet. Clin Exp Rheumatol. 2014;32(1):137-42.19.PubMed Iagnocco A, Ceccarelli F, Mennini M, Rutigliano IM, Perricone C, Nenna R, et al. Subclinical synovitis detected by ultrasound in children affected by coeliac disease: a frequent manifestation improved by a gluten-free diet. Clin Exp Rheumatol. 2014;32(1):137-42.19.PubMed
Metadata
Title
In a large Juvenile Idiopathic Arthritis (JIA) cohort, concomitant celiac disease is associated with family history of autoimmunity and a more severe JIA course: a retrospective study
Authors
Roberta Naddei
Simona Di Gennaro
Alfredo Guarino
Riccardo Troncone
Maria Alessio
Valentina Discepolo
Publication date
01-12-2022
Publisher
BioMed Central
Published in
Pediatric Rheumatology / Issue 1/2022
Electronic ISSN: 1546-0096
DOI
https://doi.org/10.1186/s12969-022-00689-4

Other articles of this Issue 1/2022

Pediatric Rheumatology 1/2022 Go to the issue