TY - JOUR
T1 - Oral manifestations of Chediak-Higashi syndrome
T2 - A systematic review
AU - de Arruda, José Alcides Almeida
AU - Sousa-Neto, Sebastião Silvério
AU - Abreu, Lucas Guimarães
AU - Schuch, Lauren Frenzel
AU - Souza, Vinícius Gomes
AU - Alves, Thainara Vitória Lima
AU - Martins-Andrade, Bárbara
AU - Shetty, Sameep S.
AU - Monteiro, João Luiz Gomes Carneiro
AU - Mendonça, Elismauro Francisco
AU - Mesquita, Ricardo Alves
AU - Callou, Gerhilde
N1 - Funding Information:
This study was supported by Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES, Finance Code 001) , Brazil. J.A.A.A., S.S.S.N., L.F.S., and J.L.G.C.M. are recipients of fellowships. We also thank Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq) (#305493/2018-3; #435644/2018-1; #404710/2018-2; #310797/2019-5) for financial support. E.F.M, L.G.A. and R.A.M., are research fellows of CNPq. Mrs. E. Greene provided English editing of the manuscript.
Publisher Copyright:
© 2022 Elsevier Inc.
PY - 2023/1
Y1 - 2023/1
N2 - 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.
AB - 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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U2 - 10.1016/j.disamonth.2022.101356
DO - 10.1016/j.disamonth.2022.101356
M3 - Article
AN - SCOPUS:85127851345
SN - 0011-5029
VL - 69
JO - Disease-a-Month
JF - Disease-a-Month
IS - 1
M1 - 101356
ER -