Oral health in patients with Bruton’s disease: An integrative review
DOI:
https://doi.org/10.33448/rsd-v14i12.50315Keywords:
Agammaglobulinemia, Bruton’s Tyrosine Kinase, Oral Health, Oral Manifestations.Abstract
Introduction: Bruton’s disease (BD) or X-linked agammaglobulinemia is characterized by the patient’s inability to produce antibodies. Immunological vulnerability combined with inadequate oral hygiene, makes the oral environment more conducive to bacterial proliferation. Objective: To conduct an integrative review of the oral manifestations affecting patients with BD. Methods: A search was carried out in the BVS, CAPES Journals, and PubMed databases using the descriptors: Agammaglobulinemia, Oral Health, Oral Manifestations, Dental Caries, and Periodontal Disease. The PICOT strategy guided the research and the selection process was performed in pairs. Results: A total of 180 articles were identified, of which five were included in the final sample. The described alterations included aphthous ulcers, herpes infections, candidiasis, tonsillitis, gingivitis, and dental calculus. Pseudomonas sepsis occurred in one case report, presenting with injected tonsils with exudates, ulcerated tongue, and edematous upper lip with erythematous induration and crusts that progressed to oral ulcers and ecthyma gangrenosum. Significantly higher levels of lactobacilli and yeasts were found in the oral cavity of these patients compared with individuals with selective IgA deficiency and with healthy controls. Alveolar bone resorption of the maxillary incisors, multiple carious lesions, and marginal gingivitis along the dental arch were observed in a patient with BD associated with Trisomy 21. Conclusion: Individuals with BD may present alterations that compromise not only oral health but also contribute to the worsening of systemic conditions. Early diagnosis, immunoglobulin replacement therapy, and the maintenance of adequate oral hygiene may result in better oral health outcomes.
References
Balkarlı, E., Özen, B S., Akçal, Ö., Taşkırdı. İ., Akay, H İ. & Gülez, N. (2023). Clinical, Molecular, Immunological Properties and Our Clinical Experiences in Patients Diagnosed with X-linked Agamaglobulinemia. J Behcet Uz Child Hosp., 13 (3), 185-191. doi: 10.4274/jbuch.galenos.2023.78545
Black, C., Zavod, M. B. & Gosselin, B. J. (2005). Haemophilus influenzae lymphadenopathy in a patient with agammaglobulinemia: clinical-histologic-microbiologic correlation and review of the literature. Rev. Arch Pathol Lab Med. 129 (1), 100-103. doi: 10.5858/2005-129-100-HILIAP
Cardoso, C. R., Passos, D. & Raimondi, J. V. (2017). Compreendendo a cárie dental. Rev. Salusvista, 36(4), 1153-1168. https://secure.unisagrado.edu.br/static/biblioteca/salusvita/salusvita_v36_n4_2017_art_12.pdf
Condé, K., Atakla, H., Barry, M., Condé, M. & Dore, M. (2020). Presentation of a case of Bruton type primary agammaglobulinemia in Guinea. Pan African Medical Journal, 36 (385), 1-4. doi: 10.11604/pamj.2020.36.385.24771
Hassunuma R.M., Garcia P. C., Ventura, T. M. O., Senada, A.L. & Messias, S. H. N. (2024). Revisão integrativa e redação de artigo científico: uma proposta metodológica em 10 passos. Revista Multidisciplinar de Educação e Meio Ambiente, 5 (3), 1-16. https://doi.org/10.51189/integrar/rema/4275
Kusumoto, Y., Imai, K., Ohyama, Y., Fukayama, H. & Shinozuka, O. (2020). Oral management of a patient with downsyndrome and agammaglobulinemia: acase report. BMC Oral Health, 20 (71), 1-5. https://doi.org/10.1186/s12903-020-1056-2
Kıykım, A., Topçuoğlu, N., Külekçi, G., Cokçaliş, H., Akçakaya, N. & Camcioglu, Y. (2013) Comparison of oral microflora in selective IgA deficiency and x linked agammaglobulinemia cases with control group. Turk Arch Pediatr., 48, 204-209. 10.4274/tpa.438
Lin, S., Chang, Y., Chiang, B., Fang, S. & Lee, Y. (2020). Pseudomonas aeruginosa sepsis presenting as oral ecthyma gangrenosum in identical twins with Bruton tyrosine kinase gene mutation: Two case reports and review of the literature. Journal of Microbiology, Immunology and Infection, 53(6), 1030-1034. https://doi.org/10.1016/j.jmii.2020.04.009
Medeiros e Paula, A B., Melo T R N., Ribeiro, A L P F., Pimentel, A L M L L., Gurgel, L O., Oliveira, MC., Oliveira V A., & Nóbrega, V M. (2024). Agamaglobulinemia ligada ao X em paciente pediátrico: relato de caso. Journal Health NPEPS, 9 (1), e12624. http://dx.doi.org/10.30681/2526101012624
Meighani, G., Aghamohammadi, A., Javanbakht, H., Abolhassani, H., Nikayin, S., Jafari, S., Motlagh, M., Shamshiri, A. & Rezaei. N. (2011). Estado de saúde bucal e odontológica em pacientes com deficiências primárias de anticorpos. Iranian Journal Alergia Asma Imunol., 10(4), 289-293. https://pubmed.ncbi.nlm.nih.gov/22184272/
Pereira, A. S. et al. (2018). Metodologia da pesquisa científica. [Free ebook]. Santa Maria. Editora da UFSM. https://repositorio.ufsm.br/bitstream/handle/1/15824/Lic_Computacao_Metodologia-Pesquisa-Cientifica.pdf?sequence=1
Riva, J. J., Malik, K. M., Burnie, S. J., Endicott, A. R., & Busse, J. W. (2012). What is your research question? An introduction to the PICOT format for clinicians. The Journal of the Canadian Chiropractic Association, 56 (3), 167–171. https://pmc.ncbi.nlm.nih.gov/articles/PMC3430448/
Robertson, P. B., Mackler, B. F., Wright, T. E., & Levy, B. M. (1980). Periodontal status of patients with abnormalities of the immune system. II. Observations over a 2-year period. Journal of periodontology, 51(2), 70–73. https://doi.org/10.1902/jop.1980.51.2.70
Snyder, H. (2019). Literature review as a research methodology: An overview and guidelines. Journal of Business Research, 104, 333-339. https://doi.org/10.1016/j.jbusres.2019.07.039
Yong, P. F K., Chee, R. & Grimbacher, B. (2008). Hypogammaglobulinemia. Rev. Immunol Allergy Clin North Am., 28, 691-701. https://doi.org/10.1016/j.iac.2008.06.003
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Copyright (c) 2025 Marcos José Pereira Silva Santos, Larissa Ribeiro de Faria, Joice Kelly de Paula Nascimento, Tiago Lima da Silva, Walter Barros Leonel, Patrícia da Silva Santos, Raquel Xavier Ligeiro Dias

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