Oral Manifestations of Histiocytosis of Langerhans Cells: a Pediatric Case Involving Mandible Bone

Authors

  • René Abdiel Montero-Sánchez Head of Departament of Oral and Maxillofacial Surgery, General Hospital Boca del Río. Veracruz, Mexico. Faculty of Dentistry, Universidad Veracruzana. Veracruz, México.ORCID ID https://orcid.org/0000-0002-6644-5251 Author
  • José Andrés Velázquez-Martínez Oral Pathologist and Medicine of Departament of Pathology Anatomy, High Specialty Hospital of Veracruz. Veracruz, México. Faculty of Dentistry, Universidad Veracruzana. Veracruz, México.ORCID ID https://orcid.org/0000-0002-9592-8199 Author
  • Mariana Gabriela Lecourtois-Amézquita Faculty of Dentistry, Universidad Veracruzana. Veracruz, México. ORCID ID https://orcid.org/0000-0002-4633-6023 Author

DOI:

https://doi.org/10.15517/ijds.2021.44504

Keywords:

Langerhans cells; Histiocytosis; Osteolytic; Biopsy; Oral pathology; Bone.

Abstract

Langerhans cells histiocytosis is a rare disease characterized by monoclonal proliferation and migration of special dendritic cells in a variety of organs, most commonly appears eosinophilic granuloma localized, often solitary, and bone lesions that occurs predominantly in pediatric patients. Although is most prevalent in children under the age of 15, this disorder presents in all ages and occurs at a rate of 2 to 5 cases per million per year. LCH is a complex entity; the clinic manifestations can mimic other common conditions and therefore a comprehensive evaluation is indicated. Since oral manifestations are frequent, the early diagnose of this pathology could be detected by dental professionals. The aim of this case report is to describe a case of LCH who initially was misdiagnosed and treated for a dental infection. This disease requires accurate histopathological diagnosis and timely treatment; hence it is necessary to raise awareness among dentists to avoid misdiagnose of oral manifestations of LCH.

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References

Margo C.E., Goldman D.R. Langerhans cell histiocytosis. Surv Ophthalmol. 2008; 53: 332-58. doi:10.1016/j.survophthal.2008.04.007 DOI: https://doi.org/10.1016/j.survophthal.2008.04.007

Satter E.K., High W.A. Langerhans cell histiocytosis: a review of the current recommendations of the Histiocyte Society. Pediatr Dermatol. 2008; 25: 291-5. doi:10.1111/j.1525-1470.2008.00669.x DOI: https://doi.org/10.1111/j.1525-1470.2008.00669.x

Allen C.E., Merad M., McClain K.L. Langerhans-cell histiocytosis. N Eng J Med. 2018, 379 (9), 856-868. doi: 10.1056/NEJMra1607548 DOI: https://doi.org/10.1056/NEJMra1607548

Krooks J., Minkov M., Weatherall A.G. Langerhans cell histiocytosis in children: History, classification, pathobiology, clinical manifestations, and prognosis. J Am Acad Dermatol. 2018 Jun; 78 (6): 1035-1044. doi: 10.1016/j.jaad.2017.05.059. PMID: 29754885 DOI: https://doi.org/10.1016/j.jaad.2017.05.059

Guyot-Goubin A., Donadieu J., Barkaoui M., Bellec S., Thomas C., Clavel J. Descriptive epidemiology of childhood Langerhans cell histiocytosis in France, 2000-2004. Pediatr Blood Cancer 2008; 51: 71-5. doi:10.1002/pbc.21498 DOI: https://doi.org/10.1002/pbc.21498

Stålemark H., Laurencikas E., Karis J., Gavhed D., Fadeel B., Henter J-I. Incidence of Langerhans cell histiocytosis in children: a population-based study. Pediatr Blood Cancer. 2008; 51: 76-81. doi:10.1002/pbc.21504 DOI: https://doi.org/10.1002/pbc.21504

Peckham-Gregory E.C., McClain K.L., Allen C.E., Scheurer M.E., Lupo P.J. The role of parental and perinatal characteristics on Langerhans cell histiocytosis: characterizing increased risk among Hispanics. Ann Epidemiol. 2018; 28: 521-8. https://doi.org/10.1016/j.annepidem.2018.04.005 DOI: https://doi.org/10.1016/j.annepidem.2018.04.005

Venkatramani R., Rosenberg S., Indramohan G., Jeng M., Jubran R. An exploratory epidemiological study of Langerhans cell histiocytosis. Pediatr Blood Cancer 2012; 59: 1324-6. doi:10.1002/pbc.24136 DOI: https://doi.org/10.1002/pbc.24136

Haupt R., Minkov M., Astigarraga I., Schäfer E., Nanduri V., Jubran R., et al. Langerhans cell histiocytosis (LCH): guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years. Pediatr Blood Cancer. 2013; 60: 175-184. doi:10.1002/pbc.24367 DOI: https://doi.org/10.1002/pbc.24367

Su M., Gao Y-J, Pan C., Chen J., Tang J-Y. Outcome of children with Langerhans cell histiocytosis and single-system involvement: A retrospective study at a single center in Shanghai, China. Pediatr Hematol Oncol. 2018; 35: 385-92. doi:10.1080/08880018.2018.1545814 DOI: https://doi.org/10.1080/08880018.2018.1545814

Eden P., Abeyasinghe, Mufeesc M.B.M., Jayasooriya P.R. A series of 13 new cases of langerhans cell histiocytosis of the oral cavity: a master of disguise. Oral Health Care 2. 2017. DOI: 10.15761/OHC.1000110 DOI: https://doi.org/10.15761/OHC.1000110

Li Z., Yanqiu L., Yan W., Xiaoying Q., Hamze F., Siyuan C., et al. Two case report studies of Langerhans cell histiocytosis with an analysis of 918 patients of Langerhans cell histiocytosis in literatures published in China. Int J Dermatol. 2010; 49: 1169-1174. doi:10.1111/j.1365-4632.2009.04360.x DOI: https://doi.org/10.1111/j.1365-4632.2009.04360.x

Varga E., Korom I., Polyánka H., Szabó K., Széll M., Baltás E., et al. BRAFV600E mutation in cutaneous lesions of patients with adult Langerhans cell histiocytosis. J Eur Acad Dermatol Venereol. 2015;29: 1205-1211. doi:10.1111/jdv.12792 DOI: https://doi.org/10.1111/jdv.12792

Rigaud C., Barkaoui M.A., Thomas C., Bertrand Y., Lambilliotte A., Miron J., et al. Langerhans cell histiocytosis: therapeutic strategy and outcome in a 30-year nationwide cohort of 1478 patients under 18 years of age. Br J Haematol. 2016; 174 (6): 887-898. doi:10.1111/bjh.14140 DOI: https://doi.org/10.1111/bjh.14140

Emile J.F., Abla O., Fraitag S., Horne A., Haroche J., Donadieu J., et al. Histiocyte Society. Revised classification of histiocytoses and neoplasms of the macrophage-dendritic cell lineages. Blood. 2016 Jun 2; 127 (22): 2672-81. doi: 10.1182/blood-2016-01-690636 DOI: https://doi.org/10.1182/blood-2016-01-690636

Leung A.K., Lam J.M., Leong K.F. Childhood Langerhans cell histiocytosis: a disease with many faces. World Journal of Pediatrics,2019; 1-10. doi: https://doi.org/10.1007/s12519-019-00304-9 DOI: https://doi.org/10.1007/s12519-019-00304-9

Thacker N.H., Abla O. Pediatric Langerhans cell histiocytosis: state of the science and future directions. Clin Adv Hematol Oncol. 2019;17: 122-31.

Rodriguez G.C., Allen C.E. Langerhans cell histiocytosis. Blood 2020; 135 (16): 1319-1331. doi: 10.1182/blood.2019CM0000 DOI: https://doi.org/10.1182/blood.2019000934

Neves-Silva R., Fernandes D.T., Fonseca F.P., Rebelo P.H.A., Ferreira B.B., Santos-Silva R.A., et al. Oral manifestations of Langerhans cell histiocytosis: A case series. Spec Care Dentist. 2018; 38: 426-433. https://doi.org/10.1111/scd.12330 DOI: https://doi.org/10.1111/scd.12330

Goyal G., Shah M.V., Hook C.C., et al. Adult disseminated Langerhans cell histiocytosis: incidence, racial disparities and long-term outcomes. Br J Haematol. 2018; 182 (4): 579-581. doi:10.1111/bjh.14818 DOI: https://doi.org/10.1111/bjh.14818

Ribeiro K.B., Degar B., Antoneli C.B.G., Rollins B., Rodriguez-Galindo C. Ethnicity, race, and socioeconomic status influence incidence of Langerhans cell histiocytosis. Pediatr Blood Cancer. 2015; 62 (6): 982-987. doi:10.1002/pbc.25404 DOI: https://doi.org/10.1002/pbc.25404

Chow T.W., Leung W.K., Cheng F.W.T., Kumta S.M., Chu W.C.W., Lee V., Li C.K., et al. Late outcomes in children with Langerhans cell histiocytosis. Arch Dis Child. 2017; 102 (9): 830-835. DOI: https://doi.org/10.1136/archdischild-2016-312185

Merglová V., Hrušák D., Boudova L., Mukenšnabl P., Valentová E., Hostička L. Langerhans cell histiocytosis in childhood–Review, symptoms in the oral cavity, differential diagnosis and report of two cases.J Craniomaxillofac Surg. 2014; 42 (2), 93-100. http://dx.doi.org/10.1016/j.jcms.2013.03.005 DOI: https://doi.org/10.1016/j.jcms.2013.03.005

Published

2026-05-27