Non-Cochrane · prognostic · PM42397633
Prevalence of oral lesions in Langerhans cell histiocytosis: a systematic review and meta-analysis.
Dioguardi Mario, Cazzolla Angela Pia, Guerra Ciro, et al. · Oral and maxillofacial surgery · 2026 · PubMed · doi:10.1007/s10006-026-01586-w
Findings
8 findings
| Population | ||||
|---|---|---|---|---|
| Patients with a diagnosis of Langerhans cell histiocytosis (LCH) | Oral manifestations of LCH (oral mucosa and jaw bone lesions) | Not applicable (single-arm prevalence) | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) At diagnosis (not specified) · Clinical cohorts of LCH patients · Sensitivity analysis excluding 5 studies with poor CI overlap; random-effects model | Proportion 0.097 [0.076, 0.119] GRADE not reported 11 studies · 1806 participants |
| Patients with a diagnosis of Langerhans cell histiocytosis (LCH) | Oral manifestations of LCH (oral mucosa and jaw bone lesions) | Not applicable (single-arm prevalence) | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) At diagnosis (not specified) · Clinical cohorts of LCH patients · All 16 studies; random-effects model (DerSimonian-Laird) | Proportion 0.205 [0.144, 0.267] Low Downgraded: risk of bias 16 studies · 2052 participants |
| Patients diagnosed with Langerhans cell histiocytosis (histiocytosis) | Oral manifestations (oral mucosa and jaw bone lesions) | Not applicable | Prevalence of oral manifestations At diagnosis · Overall (16 studies), aggregate proportion as stated in abstract | Prevalence (%) 14.86 [11.80, 18.58] Low Downgraded: risk of bias 16 studies · 2052 participants |
| Patients with a diagnosis of Langerhans cell histiocytosis (LCH) | Oral manifestations of LCH (oral mucosa and jaw bone lesions) | Not applicable (single-arm prevalence) | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) At diagnosis (not specified) · Clinical cohorts of LCH patients · Sensitivity analysis excluding 5 studies; as stated in the abstract | Proportion 0.1057 GRADE not reported 11 studies · 1806 participants |
| Patients with LCH across 16 included studies | Mean age at diagnosis (study level) | Prevalence of oral manifestations | Correlation between mean age at diagnosis and prevalence of oral manifestations Clinical cohorts of LCH patients · Weighted by study sample size | Spearman rho -0.258 GRADE not reported 16 studies |
| Patients with LCH across 16 included studies | Mean age at diagnosis (study level) | Prevalence of oral manifestations | Correlation between mean age at diagnosis and prevalence of oral manifestations Clinical cohorts of LCH patients · Unweighted | Spearman rho 0.218 GRADE not reported 16 studies |
| Patients with LCH across 16 included studies | Mean age at diagnosis (study level) | Prevalence of oral manifestations | Correlation between mean age at diagnosis and prevalence of oral manifestations Clinical cohorts of LCH patients · Unweighted | Pearson r 0.337 GRADE not reported 16 studies |
| Patients with LCH across 16 included studies | Mean age at diagnosis (study level) | Prevalence of oral manifestations | Correlation between mean age at diagnosis and prevalence of oral manifestations Clinical cohorts of LCH patients · Weighted by study sample size | Pearson r 0.007 GRADE not reported 16 studies |
Included primary studies 16
Open in Studies| Study | Year | Also included by | Outcomes it informs here | Trial data |
|---|---|---|---|---|
| al-Ammar 1999 Langerhans' cell histiocytosis: paediatric head and neck study. PMID:10579156 | 1999 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Annibali 2009 Langerhans cell histiocytosis: oral/periodontal involvement in adult patients DOI:10.1111/j.1601-0825.2009.01601.x | 2009 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Buchmann 2006 Primary head and neck Langerhans cell histiocytosis in children DOI:10.1016/j.otohns.2006.03.019 | 2006 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Cochrane 2003 Langerhans' Cell Histiocytosis in the Paediatric Population: Presentation and Treatment of Head and Neck Manifestationsy DOI:10.2310/7070.2003.35266 | 2003 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| DiNardo 1989 Head and neck manifestations of histiocytosis‐X in children DOI:10.1288/00005537-198907000-00011 | 1989 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Gadner 1994 Treatment strategy for disseminated langerhans cell histiocytosis DOI:10.1002/mpo.2950230203 | 1994 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Garcia 2022 Langerhans cell histiocytosis of the head and neck in the pediatric population in a tertiary center: Clinical presentation, classification… DOI:10.1016/j.ijporl.2022.111073 | 2022 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Hartman 1980 Histiocytosis X: A review of 114 cases with oral involvement DOI:10.1016/0030-4220(80)90030-4 | 1980 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Irving 1994 Langerhans' cell histiocytosis in childhood: Management of head and neck manifestations DOI:10.1288/00005537-199401000-00011 | 1994 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Lee 2014 Clinical Characteristics and Treatment Outcome of Langerhans Cell Histiocytosis: 22 Years’ Experience of 154 Patients at a Single Center DOI:10.3109/08880018.2013.865095 | 2014 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Lin 2023 Craniofacial involvement in Langerhans cell histiocytosis: A review of 44 cases at a single medical center DOI:10.1016/j.bjps.2023.03.006 | 2023 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Pavan 2023 Langerhans cell histiocytosis oral lesions in pediatric patients. PMID:38345277 | 2023 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Quraishi 1995 Langerhans' cell histiocytosis: Head and neck manifestations in children DOI:10.1002/hed.2880170310 | 1995 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Rapidis 1978 Histiocytosis X DOI:10.1016/s0300-9785(78)80051-9 | 1978 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Sedano 1969 Histiocytosis X DOI:10.1016/0030-4220(69)90146-7 | 1969 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
| Sigala 1972 Dental involvement in histiocytosis DOI:10.1016/0030-4220(72)90206-x | 1972 | this review only | Prevalence of oral manifestations (oral mucosa or jaw bone lesions) | not extracted yet |
Review question
| Element | Planned (methods) | Found (results) |
|---|---|---|
| Population | Patients with a diagnosis of histiocytosis (analysis focused on Langerhans cell histiocytosis) | Patients with LCH (n=2052 in pooled analysis; 16 studies) |
| Intervention | Oral manifestations of histiocytosis involving the mucosa and jaw bones | Oral manifestations (mandible, maxilla, gingiva, mucosa; osteolytic, ulcerative, periodontal lesions) |
| Comparison | Not applicable | Not applicable |
| Outcomes | Prevalence of oral lesions and manifestations in patients diagnosed with histiocytosis | Pooled prevalence of oral manifestations 20.5% (95% CI 14.4-26.7; 16 studies); 9.7% (7.6-11.9; 11 studies) after sensitivity analysis |
| Study designs | Clinical trials and epidemiological studies (case reports, case series, reviews excluded); at least 20 patients; English | Retrospective single or two center studies (n=14), one prospective observational study, one multicenter RCT |
| Timing | Not specified | At diagnosis |