Celiac disease (CD) diagnosis can be extremely challenging in the case of atypical patterns. In this context, oral signs seem to play a decisive role in arousing suspicion of these forms of the disease. At the same time, the different expressions of the HLA-DQB1*02 allele apparently seem to facilitate the interpretation of signs and highlighted symptoms. The aim of this work was to verify whether it is possible to identify a correlation between the development of oral signs and different DQ2 haplotypes in celiac pediatric patients. 44 celiac patients with a medium age of 9.9 were studied. Oral examinations were performed in order to identify recurrent aphthous stomatitis (RAS) and dental enamel defects (DED). The diagnosis of DED resulted as being related to allele expression ( value = 0.042) while it was impossible to find a similar correlation with RAS. When both oral signs were considered, there was an increase in correlation with HLA-DQB1*02 expression ( value = 0.018). The obtained results identified both the fundamental role that dentists can play in early diagnosis of CD, as well as the possible role of HLA haplotype analysis in arousing suspicion of atypical forms of the disease. 1. Introduction Celiac disease (CD) is a complex pathologic condition involving the small intestine mucosa resulting from intolerance to gluten assumption [1]. More specifically, it is considered a genetic and autoimmune condition that can affect patients of any age and gender with a great variability of symptoms and clinical signs. Although the final diagnosis of CD is always based on a biopsy with the detection of severe villous atrophy coupled with crypt hyperplasia, the diagnostic pathway leading to this conclusion can often be long and winding [1, 2]. This pathology, initially considered as typical of the European population, is nowadays distributed worldwide. Epidemiological analysis has reported a prevalence of celiac disease as varying greatly between the western and the eastern parts of the world, so that CD is today considered as the most common genetic disorder in the west with a prevalence of 1%, while it is relatively unknown in Asian countries [1, 3]. This irregular distribution in different countries has been related to genetic and alimentary factors. CD is actually an intestinal enteropathy whose symptoms are determined by the ingestion of gliadin in genetically predisposed patients [1]. Gliadin is a prolamin, a class of peptides highly resistant to gastrointestinal enzymes, which causes histological changes in the small intestine mucosa of the celiac
References
[1]
N. Gujral, H. J. Freeman, and A. B. Thomson, “Celiac disease: prevalence, diagnosis, pathogenesis and treatment,” World Journal of Gastroenterology, vol. 18, no. 42, pp. 6036–6059, 2012.
[2]
D. Schuppan, Y. Junker, and D. Barisani, “Celiac Disease: from pathogenesis to novel therapies,” Gastroenterology, vol. 137, no. 6, pp. 1912–1933, 2009.
[3]
P. H. R. Green and C. Cellier, “Medical progress: celiac disease,” The New England Journal of Medicine, vol. 357, no. 17, pp. 1731–1743, 2007.
[4]
C. Ruggeri, A. T. la Masa, S. Rudi et al., “Celiac disease and non-organ-specific autoantibodies in patients with chronic hepatitis C virus infection,” Digestive Diseases and Sciences, vol. 53, no. 8, pp. 2151–2155, 2008.
[5]
L. C. Stene, M. C. Honeyman, E. J. Hoffenberg et al., “Rotavirus infection frequency and risk of celiac disease autoimmunity in early childhood: a longitudinal study,” American Journal of Gastroenterology, vol. 101, no. 10, pp. 2333–2340, 2006.
[6]
M. Erriu, F. Canargiu, G. Orru, V. Garau, and C. Montaldo, “Idiopathic atrophic glossitis as the only clinical sign for celiac disease diagnosis: a case report,” Journal of Medical Case Reports, vol. 6, no. 1, article 185, 2012.
[7]
J. Cheng, T. Malahias, P. Brar, M. T. Minaya, and P. H. R. Green, “The association between celiac disease, dental enamel defects, and aphthous ulcers in a United States cohort,” Journal of Clinical Gastroenterology, vol. 44, no. 3, pp. 191–194, 2010.
[8]
P. C. D. Silva, P. D. V. de Almeida, M. ？. N. Machado et al., “Oral manifestations of celiac disease. A case report and review of the literature,” Medicina Oral, Patologia Oral y Cirugia Bucal, vol. 13, no. 9, pp. E559–E562, 2008.
[9]
L. Pastore, A. Carroccio, D. Compilato, V. Panzarella, R. Serpico, and L. L. Muzio, “Oral manifestations of celiac disease,” Journal of Clinical Gastroenterology, vol. 42, no. 3, pp. 224–232, 2008.
[10]
M. Erriu, S. Sanna, A. Nucaro, G. Orrù, V. Garau, and C. Montaldo, “HLA-DQB1 haplotypes and their relation to oral signs linked to celiac disease diagnosis,” Open Dentistry Journal, vol. 5, no. 1, pp. 174–178, 2011.
[11]
A. Majorana, E. Bardellini, A. Ravelli, A. Plebani, A. Pol, and G. Campus, “Implications of gluten exposure period, CD clinical forms, and HLA typing in the association between celiac disease and dental enamel defects in children. A case-control study,” International Journal of Paediatric Dentistry, vol. 20, no. 2, pp. 119–124, 2010.
[12]
J. Cheng, T. Malahias, P. Brar, M. T. Minaya, and P. H. R. Green, “The association between celiac disease, dental enamel defects, and aphthous ulcers in a United States cohort,” Journal of Clinical Gastroenterology, vol. 44, no. 3, pp. 191–194, 2010.
[13]
L. Pastore, G. Campisi, D. Compilato, and L. L. Muzio, “Orally based diagnosis of celiac disease: current perspectives,” Journal of Dental Research, vol. 87, no. 12, pp. 1100–1107, 2008.
[14]
A. Ballinger, C. Hughes, P. Kumar, I. Hutchinson, and M. Clark, “Dental enamel defects in coeliac disease,” The Lancet, vol. 343, no. 8891, pp. 230–231, 1994.
[15]
C. D. Wierink, D. E. van Diermen, I. H. A. Aartman, and H. S. A. Heymans, “Dental enamel defects in children with coeliac disease,” International Journal of Paediatric Dentistry, vol. 17, no. 3, pp. 163–168, 2007.
[16]
L. Aine, M. Maki, O. Keyrilainen, and P. Collin, “Dental enamel defects in celiac disease,” Journal of Oral Pathology and Medicine, vol. 19, no. 6, pp. 241–245, 1990.
[17]
M. Erriu, F. Boscarelli, C. Peluffo et al., “A rapid sample method for HLA haplotype typization. A preliminary study on celiac patients,” Minerva Stomatologica, vol. 59, no. 9, pp. 477–487, 2010.
[18]
B. Admou, L. Essaadouni, K. Krati, K. Zaher, M. Sbihi, L. Chabaa, et al., “Atypical celiac disease: from recognizing to managing,” Gastroenterology Research and Practice, vol. 2012, Article ID 637187, 9 pages, 2012.
[19]
L. Pastore, M. de Benedittis, M. Petruzzi et al., “Importance of oral signs in the diagnosis of atypical forms of celiac disease,” Recenti Progressi in Medicina, vol. 95, no. 10, pp. 482–490, 2004.
[20]
P. Bucci, F. Carile, A. Sangianantoni, F. D'Angiò, A. Santarelli, and L. L. Muzio, “Oral aphthous ulcers and dental enamel defects in children with coeliac disease,” Acta Paediatrica, vol. 95, no. 2, pp. 203–207, 2006.
[21]
M. Costacurta, P. Maturo, M. Bartolino, and R. Docimo, “Oral manifestations of coeliac disease: a clinical-statistic study,” Journal of Oral Implantology, vol. 3, no. 1, pp. 12–19, 2010.
[22]
M. Rashid, M. Zarkadas, A. Anca, and H. Limeback, “Oral manifestations of celiac disease: a clinical guide for dentists,” Journal of the Canadian Dental Association, vol. 77, article b39, 2011.
[23]
M. Rashid, A. Cranney, M. Zarkadas et al., “Celiac disease: evaluation of the diagnosis and dietary compliance in Canadian children,” Pediatrics, vol. 116, no. 6, pp. e754–e759, 2005.
[24]
A. Cranney, M. Zarkadas, I. D. Graham et al., “The Canadian Celiac Health Survey,” Digestive Diseases and Sciences, vol. 52, no. 4, pp. 1087–1095, 2007.
[25]
G. Campisi, C. di Liberto, A. Carroccio et al., “Coeliac disease: oral ulcer prevalence, assessment of risk and association with gluten-free diet in children,” Digestive and Liver Disease, vol. 40, no. 2, pp. 104–107, 2008.
[26]
？. Ya？ar, B. Ya？ar, E. Abut, and Z. A？iran Serdar, “Clinical importance of celiac disease in patients with recurrent aphthous stomatitis,” Turkish Journal of Gastroenterology, vol. 23, no. 1, pp. 14–18, 2012.
[27]
M. A. El-Hodhod, I. A. El-Agouza, H. Abdel-Al, N. S. Kabil, and K. A. Bayomi, “Screening for celiac disease in children with dental enamel defects,” ISRN Pediatrics, vol. 2012, Article ID 763783, 7 pages, 2012.
[28]
R.-D. Jores, F. Frau, F. Cucca et al., “HLA-DQB1*0201 homozygosis predisposes to severe intestinal damage in celiac disease,” Scandinavian Journal of Gastroenterology, vol. 42, no. 1, pp. 48–53, 2007.
