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Published in: Clinical Oral Investigations 6/2021

Open Access 01-06-2021 | Langerhans Cell Histiocytosis | Review

Periodontal manifestations of Langerhans cell histiocytosis: a systematic review

Authors: Julia C. Difloe-Geisert, Selina A. Bernauer, Noémie Schneeberger, Michael M. Bornstein, Clemens Walter

Published in: Clinical Oral Investigations | Issue 6/2021

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Abstract

Objectives

To explore the evidence of periodontal manifestations and treatment modalities in patients with Langerhans cell histiocytosis (LCH).

Material and methods

A systematic literature search was performed and the criteria for PRISMA and risk of bias assessment were applied. Human clinical studies (≥10 patients) presenting patients with LCH and periodontal findings were considered for inclusion.

Results

From 298 titles identified, six case series with a total of 1278 patients suffering from LCH were included. In these studies, oral symptoms were reported in a frequency ranging from 10 to 100%. Overall, in 216 patients (17%), oral symptoms were observed. Out of these patients, 49–100% demonstrated periodontal symptoms. The most common oral findings were pain, swelling, tooth loss/mobility, and bone lesions. Specific periodontal findings comprised varying frequencies of gingival ulcerations, increased pocket depths, and gingival bleeding. Treatment measures constituted of surgical curettage of bone lesions, soft tissue excision and/or tooth extractions, radiotherapy, systemic chemotherapy, or a combination of these approaches. Healing without recurrence of oral lesions was reported in most of the cases.

Conclusions

The available evidence on periodontal manifestations in LCH patients is heterogeneous. Several oral and periodontal findings were reported and may occur as initial symptoms and/or at later stages of the disease.

Clinical relevance

The dentist should be aware of possible oral involvement of systemic diseases such as LCH, and these manifestations may mimic periodontal disease.
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Literature
1.
go back to reference Guyot-Goubin A, Donadieu J, Barkaoui M, Bellec S, Thomas C, Clavel J (2008) Descriptive epidemiology of childhood Langerhans cell histiocytosis in France, 2000-2004. Pediatr Blood Cancer 51:71–75CrossRef Guyot-Goubin A, Donadieu J, Barkaoui M, Bellec S, Thomas C, Clavel J (2008) Descriptive epidemiology of childhood Langerhans cell histiocytosis in France, 2000-2004. Pediatr Blood Cancer 51:71–75CrossRef
2.
go back to reference Salotti JA, Nanduri V, Pearce MS, Parker L, Lynn R, Windebank KP (2009) Incidence and clinical features of Langerhans cell histiocytosis in the UK and Ireland. Arch Dis Child 94:376–380CrossRef Salotti JA, Nanduri V, Pearce MS, Parker L, Lynn R, Windebank KP (2009) Incidence and clinical features of Langerhans cell histiocytosis in the UK and Ireland. Arch Dis Child 94:376–380CrossRef
3.
go back to reference Stalemark H, Laurencikas E, Karis J, Gavhed D, Fadeel B, Henter JI (2008) Incidence of Langerhans cell histiocytosis in children: a population-based study. Pediatr Blood Cancer 51:76–81CrossRef Stalemark H, Laurencikas E, Karis J, Gavhed D, Fadeel B, Henter JI (2008) Incidence of Langerhans cell histiocytosis in children: a population-based study. Pediatr Blood Cancer 51:76–81CrossRef
4.
go back to reference Baumgartner I, von Hochstetter A, Baumert B, Luetolf U, Follath F (1997) Langerhans’-cell histiocytosis in adults. Med Pediatr Oncol 28:9–14CrossRef Baumgartner I, von Hochstetter A, Baumert B, Luetolf U, Follath F (1997) Langerhans’-cell histiocytosis in adults. Med Pediatr Oncol 28:9–14CrossRef
5.
go back to reference Arico M, Girschikofsky M, Genereau T et al (2003) Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur J Cancer 39:2341–2348CrossRef Arico M, Girschikofsky M, Genereau T et al (2003) Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur J Cancer 39:2341–2348CrossRef
6.
go back to reference Allen CE, Ladisch S, McClain KL (2015) How I treat Langerhans cell histiocytosis. Blood 126:26–35CrossRef Allen CE, Ladisch S, McClain KL (2015) How I treat Langerhans cell histiocytosis. Blood 126:26–35CrossRef
7.
go back to reference Collin M, Bigley V, McClain KL, Allen CE (2015) Cell(s) of Origin of Langerhans Cell Histiocytosis. Hematol Oncol Clin North Am 29:825–838CrossRef Collin M, Bigley V, McClain KL, Allen CE (2015) Cell(s) of Origin of Langerhans Cell Histiocytosis. Hematol Oncol Clin North Am 29:825–838CrossRef
8.
go back to reference Abla O, Egeler RM, Weitzman S (2010) Langerhans cell histiocytosis: current concepts and treatments. Cancer Treat Rev 36:354–359CrossRef Abla O, Egeler RM, Weitzman S (2010) Langerhans cell histiocytosis: current concepts and treatments. Cancer Treat Rev 36:354–359CrossRef
9.
go back to reference Emile JF, Abla A, Fraitag S et al (2016) Revised classification of histiocytoses and neoplasms of the macrophage-dendritic cell lineages. Blood 127:2672–2681CrossRef Emile JF, Abla A, Fraitag S et al (2016) Revised classification of histiocytoses and neoplasms of the macrophage-dendritic cell lineages. Blood 127:2672–2681CrossRef
10.
go back to reference Albandar JM, Susin C, Hughes FJ (2018) Manifestations of systemic diseases and conditions that affect the periodontal attachment apparatus: case definitions and diagnostic considerations. J Clin Periodontol 45(Suppl 20):S171–S189CrossRef Albandar JM, Susin C, Hughes FJ (2018) Manifestations of systemic diseases and conditions that affect the periodontal attachment apparatus: case definitions and diagnostic considerations. J Clin Periodontol 45(Suppl 20):S171–S189CrossRef
11.
go back to reference Jepsen S, Caton JG, Albandar JM et al (2018) Periodontal manifestations of systemic diseases and developmental and acquired conditions: consensus report of workgroup 3 of the 2017 World Workshop on the Classification of Periodontal and Peri-Implant Diseases and Conditions. J Clin Periodontol 45(Suppl 20):S219–S229CrossRef Jepsen S, Caton JG, Albandar JM et al (2018) Periodontal manifestations of systemic diseases and developmental and acquired conditions: consensus report of workgroup 3 of the 2017 World Workshop on the Classification of Periodontal and Peri-Implant Diseases and Conditions. J Clin Periodontol 45(Suppl 20):S219–S229CrossRef
12.
go back to reference Moher D, Liberati A, Tetzlaff J, Altman DG, Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 6:e1000097CrossRef Moher D, Liberati A, Tetzlaff J, Altman DG, Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 6:e1000097CrossRef
13.
go back to reference Liberati A, Altman DG, Tetzlaff J et al (2009) The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation and elaboration. PLoS Med 6:e1000100CrossRef Liberati A, Altman DG, Tetzlaff J et al (2009) The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation and elaboration. PLoS Med 6:e1000100CrossRef
14.
go back to reference Walter C, Schmidt JC, Rinne CA, Mendes S, Dula K, Sculean A (2020) Cone beam computed tomography (CBCT) for diagnosis and treatmentt planning in periodontology: systematic review update. Clin Oral Investig 24:2943–2958CrossRef Walter C, Schmidt JC, Rinne CA, Mendes S, Dula K, Sculean A (2020) Cone beam computed tomography (CBCT) for diagnosis and treatmentt planning in periodontology: systematic review update. Clin Oral Investig 24:2943–2958CrossRef
15.
go back to reference Landis JR, Koch GG (1977) The measurement of observer agreement for categorical data. Biometrics 33:159–174CrossRef Landis JR, Koch GG (1977) The measurement of observer agreement for categorical data. Biometrics 33:159–174CrossRef
16.
go back to reference Sedano HO, Cernea P, Hosxe G, Gorlin RJ (1969) Histiocytosis X. Clinical, radiologic, and histologic findings with special attention to oral manifestations. Oral Surg Oral Med Oral Pathol 27:760–771CrossRef Sedano HO, Cernea P, Hosxe G, Gorlin RJ (1969) Histiocytosis X. Clinical, radiologic, and histologic findings with special attention to oral manifestations. Oral Surg Oral Med Oral Pathol 27:760–771CrossRef
17.
go back to reference Sigala JL, Silverman S Jr, Brody HA, Kushner JH (1972) Dental involvement in histiocytosis. Oral Surg Oral Med Oral Pathol 33:42–48CrossRef Sigala JL, Silverman S Jr, Brody HA, Kushner JH (1972) Dental involvement in histiocytosis. Oral Surg Oral Med Oral Pathol 33:42–48CrossRef
18.
go back to reference Hartman KS (1980) Histiocytosis X: a review of 114 cases with oral involvement. Oral Surg Oral Med Oral Pathol 49:38–54CrossRef Hartman KS (1980) Histiocytosis X: a review of 114 cases with oral involvement. Oral Surg Oral Med Oral Pathol 49:38–54CrossRef
19.
go back to reference Minguez I, Minguez JM, Bonet J, Penarrocha M, Sanchis JM (2004) Oral manifestations of chronic disseminated histiocytosis. A report of 10 cases. Med Oral 9:152–154PubMed Minguez I, Minguez JM, Bonet J, Penarrocha M, Sanchis JM (2004) Oral manifestations of chronic disseminated histiocytosis. A report of 10 cases. Med Oral 9:152–154PubMed
20.
go back to reference Annibali S, Cristalli MP, Solidani M et al (2009) Langerhans cell histiocytosis: oral/periodontal involvement in adult patients. Oral Dis 15:596–601CrossRef Annibali S, Cristalli MP, Solidani M et al (2009) Langerhans cell histiocytosis: oral/periodontal involvement in adult patients. Oral Dis 15:596–601CrossRef
21.
go back to reference Capodiferro S, Tempesta A, Limongelli L, Ingravallo G, Maiorano E, Sfasciotti GL, Bossu M, Polimeni A, Favia G (2020) Primary oro-facial manifestations of Langerhans cell histiocytosis in pediatric age: a bi-institutional retrospective study on 45 cases. Children 7:104CrossRef Capodiferro S, Tempesta A, Limongelli L, Ingravallo G, Maiorano E, Sfasciotti GL, Bossu M, Polimeni A, Favia G (2020) Primary oro-facial manifestations of Langerhans cell histiocytosis in pediatric age: a bi-institutional retrospective study on 45 cases. Children 7:104CrossRef
22.
go back to reference Schmidt JC, Walter C, Rischewski JR, Weiger R (2013) Treatment of periodontitis as a manifestation of neutropenia with or without systemic antibiotics: a systematic review. Pediatr Dent 35:E54–E63PubMed Schmidt JC, Walter C, Rischewski JR, Weiger R (2013) Treatment of periodontitis as a manifestation of neutropenia with or without systemic antibiotics: a systematic review. Pediatr Dent 35:E54–E63PubMed
23.
go back to reference Moga C, Guo B, Schopflocher D, Harstall C (2012) Development of a quality appraisal tool for case series studies using a modified Delphi technique. Institute of Health Economics (IHE), Edmonton Moga C, Guo B, Schopflocher D, Harstall C (2012) Development of a quality appraisal tool for case series studies using a modified Delphi technique. Institute of Health Economics (IHE), Edmonton
24.
go back to reference Mendes S, Rinne CA, Schmidt JC, Dagassan-Berndt WC (2020) Evaluation of magnetic resonance imaging for diagnostic purposes in operative dentistry – a systematic review. Clin Oral Investig 24:547–557CrossRef Mendes S, Rinne CA, Schmidt JC, Dagassan-Berndt WC (2020) Evaluation of magnetic resonance imaging for diagnostic purposes in operative dentistry – a systematic review. Clin Oral Investig 24:547–557CrossRef
25.
go back to reference Buset SL, Walter C, Friedmann A, Weiger R, Borgnakke WS, Zitzmann NU (2016) Are periodontal diseases really silent? A systematic review of their effect on quality of life. J Clin Periodontol 43:333–344CrossRef Buset SL, Walter C, Friedmann A, Weiger R, Borgnakke WS, Zitzmann NU (2016) Are periodontal diseases really silent? A systematic review of their effect on quality of life. J Clin Periodontol 43:333–344CrossRef
Metadata
Title
Periodontal manifestations of Langerhans cell histiocytosis: a systematic review
Authors
Julia C. Difloe-Geisert
Selina A. Bernauer
Noémie Schneeberger
Michael M. Bornstein
Clemens Walter
Publication date
01-06-2021
Publisher
Springer Berlin Heidelberg
Published in
Clinical Oral Investigations / Issue 6/2021
Print ISSN: 1432-6981
Electronic ISSN: 1436-3771
DOI
https://doi.org/10.1007/s00784-021-03873-0

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