Oral manifestations of Chediak-Higashi syndrome: A systematic review |
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| Affiliation: | 1. Department of Oral Surgery, Pathology and Clinical Dentistry, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Minas Gerais, Brazil;2. Department of Stomatology (Oral Pathology), School of Dentistry, Universidade Federal de Goiás, Goiânia, Goiás, Brazil;3. Department of Child''s and Adolescent''s Oral Health, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Minas Gerais, Brazil;4. Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas, Piracicaba, São Paulo, Brazil;5. Private Medical Clinic, Belo Horizonte, Brazil;6. Department of Oral and Maxillofacial Pathology, School of Dentistry, Universidade de Pernambuco, Recife, Pernambuco, Brazil;7. Department of Oral and Maxillofacial Surgery, Manipal College of Dental Sciences, Mangalore, Manipal Academy of Higher Education, A constituent of MAHE, Manipal, Karnataka, India;8. Department of Oral and Maxillofacial Surgery, School of Dentistry, Universidade de Pernambuco, Recife, Pernambuco, Brazil;9. Department of Clinics and Preventive Dentistry, School of Dentistry, Universidade Federal de Pernambuco, Recife, Pernambuco, Brazil;1. Department of Maxillofacial Surgery and Diagnostic Sciences, Division of Oral Pathology, College of Dentistry, Jazan University, Jazan, Saudi Arabia;2. Department of Periodontics, Saveetha Dental College and Hospitals, Saveetha University, Chennai 600 077, India;3. Oral Pathology Division, Department of Dental Sciences, College of Dentistry, King Khalid University, Abha, Saudi Arabia;4. Department of Oral and Maxillofacial Surgery and Diagnostic Sciences, College of Dentistry, Jouf University, Sakaka, Saudi Arabia;5. Department of Public Health, Texilla American University, Guyana;6. Dow International Dental College, Dow University of Health Sciences, Karachi, Pakistan;7. Department of Maxillofacial Surgery & Diagnostic Sciences, Division of Oral Pathology, College of Dentistry, Jazan University, Jazan 45142, Saudi Arabia;8. College of Dental Medicine, Roseman University of Health Sciences, South Jordan, Utah, United States |
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| Abstract: | Chediak-Higashi syndrome (CHS) is an autosomal recessive disorder characterized by leukocytes with giant secretory granules and a myriad of clinical features. However, it is unknown whether oral lesions are part of the syndrome or are refractory to systemic treatment. Herein, we integrated the available data published in the literature on the oral manifestations of individuals with CHS. Searches on PubMed, Web of Science, Embase, Scopus, and LILACS were conducted to identify studies published up to March/2022. The Joanna Briggs Institute tool was used for the critical appraisal of studies. Fourteen articles (21 cases) were detected. The mean age of individuals was 15.9±8.8 years. There was a slight predominance of males (52.4%). The major manifestation was periodontal disease (81%), although ulceration of the oral mucosa (14.3%), gingival/labial abscess (4.8%), and periodontal abscess (4.8%) were also reported. Oral rehabilitation including dental implants (9.5%) was performed after tooth losses due to the poor prognosis of periodontal therapy. CHS is usually diagnosed in an early stage due to its systemic manifestations such as classic oculocutaneous albinism, recurrent infections, and a propensity for bleeding. Oral health providers should be aware of the manifestations of individuals with CHS. Special care, including oral prophylaxis, is indispensable. |
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