Background. The main aim of the study was to assess the association between joint hypermobility (JH) and gastrointestinal (GI) disorders in children. Methods. All children aged 4-17 years attending the clinics of the participating Pediatric Gastroenterology Centres for functional GI disorders (FGIDs) and inflammatory bowel disease (IBD) were screened for joint laxity. JH diagnosis was inferred using the Beighton Score. JHS diagnosis was inferred based on the Brighton Criteria. Rome III Diagnostic Criteria were used to diagnose possible FGIDs. Ulcerative colitis and Crohn’s disease diagnoses were made according to the Porto Criteria. Age and sex- matched healthy children were enrolled as controls. Results. One-hundred-seventy children with GI disorders (70 with FGIDs, 50 with Crohn’s disease, and 50 with ulcerative colitis) and 100 healthy controls were enrolled in the study. JH was reported in 7/70 (10%) children with FGIDs (p=0.26 compared to controls), 4/50 (8%) children with Crohn’s disease (p=0.21 compared to controls) and 15/50 (30%) children with ulcerative colitis (p=0.09 compared to controls; p=0.01 compared to FGIDs; p=0.01 compared to Crohn’s). Conclusions. JH is more prevalent in patients suffering from ulcerative colitis compared to the healthy general population, yet the difference did not reach statistical significance. Likely, a proportion of children with ulcerative colitis and JH may show connective tissue abnormalities. However, whether JH can be considered a possible feature of pediatric GI disorders deserves further investigation.
___
1. Klemp P, Williams SM, Stansfield SA. Articular mobility in Maori and European New Zealanders. Rheumatology (Oxford) 2002; 41: 554-557.
2. Hakim A, Grahame R. Joint hypermobility. Best Prac Res Clin Rheumatol 2003; 17: 989-1004.
3. Zarate N, Farmer AD, Grahame R, et al. Unexplained gastrointestinal symptoms and joint hypermobility: is connective tissue the missing link? Neurogastroenterol Motil 2010; 22: 252-262. e78.
4. Rasquin-Weber A, Hyman PE, Cucchiara S, et al. Childhood functional gastrointestinal disorders. Gut 1999; 45(Suppl II): II60-II68.
5. Starfield B, Hoekelman RA, McCormick M, et al. Who provides health care to children and adolescents in the United States? Pediatrics 1984; 74: 991-997.
6. Vounotrypidis P, Efremidou E, Zezos P, et al. Prevalence of joint hypermobility and patterns of articular manifestations in patients with inflammatory bowel disease. Gastroenterol Res Pract 2009; 2009: 924138.
7. Danese C, Castori M, Celletti C, et al. Screening for celiac disease in the joint hypermobility syndrome/ Ehlers-Danlos syndrome hypermobility type. Am J Med Genet A 2011; 155A: 2314-2316.
8. Beighton P, Solomon L, Soskolne CL. Articular mobility in an African population. Ann Rheum Dis 1973; 32: 413-418.
9. Grahame R, Bird HA, Child A. The revised (Brighton 1998) criteria for the diagnosis of benign joint hypermobility syndrome (BJHS). J Rheumatol 2000; 27: 1777-1779.
10. Varni JW, Seid M, Kurtin PS. PedsQL 4.0: reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Med Care 2001; 39: 800-812.
11. Varni JW, Burwinkle TM, Seid M, Skarr D. The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr 2003; 3: 329-341.
12. Levine A, Koletzko S, Turner D, et al; European Society of Pediatric Gastroenterology, Hepatology, and Nutrition. ESPGHAN revised Porto Criteria for the diagnosis of inflammatory bowel disease in children and adolescents. J Pediatr Gastroenterol Nutr 2014; 58: 795-806.
13. Hakim AJ, Grahame R. Non-musculoskeletal symptoms in joint hypermobility syndrome. Indirect evidence for autonomic dysfunction? Rheumatology (Oxford) 2004; 43: 1194-1195.
14. Castori M, Sperduti I, Celletti C, Camerota F, Grammatico P. Symptom and joint mobility progression in the joint hypermobility syndrome (Ehlers-Danlos syndrome, hypermobility type). Clin Exp Rheumatol 2011; 29: 998-1005.
15. Castori M, Camerota F, Celletti C, et al. Natural history and manifestations of the hypermobility type Ehlers-Danlos syndrome: a pilot study on 21 patients. Am J Med Genet A 2010; 152A: 556-564.
16. De Wandele I, Rombaut L, Malfait F, De Backer T, De Paepe A, Calders P. Clinical heterogeneity in patients with the hypermobility type of Ehlers- Danlos Syndrome. Res Dev Disabil 2013; 34: 873-881.
17. Zweig A, Schindler V, Becker AS, van Maren A, Pohl D. Higher prevalence of joint hypermobility in constipation predominant irritable bowel syndrome. Neurogastroenterol Motil 2018; 30: e13353.
18. Fikree A, Grahame R, Aktar R, et al. A prospective evaluation of undiagnosed joint hypermobility syndrome in patients with gastrointestinal symptoms. Clin Gastroenterol Hepatol 2014; 12: 1680-1687.e2.
19. Castori M, Colombi M. Generalized joint hypermobility, joint hypermobility syndrome and Ehlers-Danlos syndrome, hypermobility type. Am J Med Genet C Semin Med Genet 2015; 169C: 1-5.
20. Reilly DJ, Chase JW, Hutson JM, et al. Connective tissue disorder–a new subgroup of boys with slow transit constipation? J Pediatr Surg 2008; 43: 1111- 1114.
21. Kovacic K, Chelimsky TC, Sood MR, Simpson P, Nugent M, Chelimsky G. Joint hypermobility: a common association with complex functional gastrointestinal disorders. J Pediatr 2014; 165: 973- 978.
22. Gocentas A, Jascaniniene N, Pasek M, et al. Prevalence of generalized joint hypermobility in school-aged children from east-central European region. Folia Morphol (Warsz) 2016; 75: 48-52.
23. Warschburger P, Hänig J, Friedt M, Posovszky C, Schier M, Calvano C. Health-related quality of life in children with abdominal pain due to functional or organic gastrointestinal disorders. J Pediatr Psychol 2014; 39: 45-54.
24. Blagden S, Kingstone T, Soundy A, Lee R, Singh S, Roberts L. A comparative study of quality of life in persons with irritable bowel syndrome and inflammatory bowel disease. Gastroenterol Nurs 2015; 38: 268-278