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Background <p>Histiocytoses are rare diseases and represent a heterogeneous group of pathologies with variable clinical features, determined by the tissue accumulation of cells of presumed dendritic or macrophage origin. Oral manifestations may constitute an early clinical sign and sometimes the only initial evidence of the pathology. The aim of this systematic review with meta-analysis was to investigate the prevalence of oral manifestations in patients affected by LCH, describing their clinical and histopathological characteristics.</p> Methods <p>A systematic search was conducted on three major databases (PubMed, Scopus and ScienceDirect) and on Google Scholar grey literature. Relevant articles were selected based on inclusion and exclusion criteria, following the guidelines of the Cochrane handbook. The primary outcome was the prevalence of oral manifestations, calculated as the proportion of affected patients compared to the total cohort in each study. A fixed-effects meta-analysis was performed. A sensitivity analysis was also performed excluding studies with high heterogeneity and a correlation analysis between the mean age at diagnosis and the prevalence of oral lesions, weighted by sample size.</p> Results <p>16 studies were included for a total of 2,174 patients. The aggregate prevalence of oral manifestations was 14.86% (95% CI: 11.80–18.58%), reduced to 10.57% in sensitivity analysis. The most frequently involved sites were the mandible and the gums, with osteolytic, ulcerative or periodontal lesions. The weighted correlation analysis between age and prevalence showed a very weak and non-significant correlation (r = 0.007, p = 0.991; Spearman ρ = − 0.258, p = 0.734), confirmed even after the exclusion of outliers.</p> Conclusions <p>Oral manifestations represent a relevant clinical component in LCH and may constitute an early diagnostic sign. However, the prevalence varies widely between studies. No significant correlation was observed between age at diagnosis and the presence of oral manifestations. However, the interpretation of these findings is limited by the predominance of retrospective studies and the overall moderate methodological quality of the available evidence. Therefore, further standardized prospective studies are needed to better clarify the clinical impact of oral lesions in LCH.</p>

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Prevalence of oral lesions in Langerhans cell histiocytosis: a systematic review and meta-analysis

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年7月3日

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  • Mario Dioguardi,
  • Angela Pia Cazzolla,
  • Ciro Guerra,
  • Diego Sovereto,
  • Angelo Martella,
  • Lorenzo Lo Muzio,
  • Khrystyna Zhurakivska,
  • Angela Tisci,
  • Roberto Lovero,
  • Vincenzo Brescia,
  • Giuseppe Troiano,
  • Andrea Ballini

摘要

Background

Histiocytoses are rare diseases and represent a heterogeneous group of pathologies with variable clinical features, determined by the tissue accumulation of cells of presumed dendritic or macrophage origin. Oral manifestations may constitute an early clinical sign and sometimes the only initial evidence of the pathology. The aim of this systematic review with meta-analysis was to investigate the prevalence of oral manifestations in patients affected by LCH, describing their clinical and histopathological characteristics.

Methods

A systematic search was conducted on three major databases (PubMed, Scopus and ScienceDirect) and on Google Scholar grey literature. Relevant articles were selected based on inclusion and exclusion criteria, following the guidelines of the Cochrane handbook. The primary outcome was the prevalence of oral manifestations, calculated as the proportion of affected patients compared to the total cohort in each study. A fixed-effects meta-analysis was performed. A sensitivity analysis was also performed excluding studies with high heterogeneity and a correlation analysis between the mean age at diagnosis and the prevalence of oral lesions, weighted by sample size.

Results

16 studies were included for a total of 2,174 patients. The aggregate prevalence of oral manifestations was 14.86% (95% CI: 11.80–18.58%), reduced to 10.57% in sensitivity analysis. The most frequently involved sites were the mandible and the gums, with osteolytic, ulcerative or periodontal lesions. The weighted correlation analysis between age and prevalence showed a very weak and non-significant correlation (r = 0.007, p = 0.991; Spearman ρ = − 0.258, p = 0.734), confirmed even after the exclusion of outliers.

Conclusions

Oral manifestations represent a relevant clinical component in LCH and may constitute an early diagnostic sign. However, the prevalence varies widely between studies. No significant correlation was observed between age at diagnosis and the presence of oral manifestations. However, the interpretation of these findings is limited by the predominance of retrospective studies and the overall moderate methodological quality of the available evidence. Therefore, further standardized prospective studies are needed to better clarify the clinical impact of oral lesions in LCH.