Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

Samira Mostafazadeh; Masoud Fallahi Motlagh; Nesa Rasooli

doi:doi:10.11648/j.ijcoms.20251102.12

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Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

Samira Mostafazadeh

, Masoud Fallahi Motlagh

, Nesa Rasooli^*

Published in International Journal of Clinical Oral and Maxillofacial Surgery (Volume 11, Issue 2)

Received: 20 September 2025 Accepted: 4 October 2025 Published: 28 October 2025

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Abstract

Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.

Published in	International Journal of Clinical Oral and Maxillofacial Surgery (Volume 11, Issue 2)
DOI	10.11648/j.ijcoms.20251102.12
Page(s)	63-68
Creative Commons	This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.
Copyright	Copyright © The Author(s), 2025. Published by Science Publishing Group

Keywords

Langerhans Cell Histiocytosis, Mandible, Pediatric, Elderly, Jaw Lesions, Case Report

References

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[2]	van de Ven R, van den Hout MF, Lindenberg JJ, Sluijter BJ, van Leeuwen PA, Lougheed SM, et al. Characterization of four conventional dendritic cell subsets in human skin-draining lymph nodes in relation to T-cell activation. Blood, The Journal of the American Society of Hematology. 2011; 118(9): 2502-10. https://doi.org/10.1182/blood-2011-03-344838
[3]	Shirley J, Thornton JB. Oral manifestations of Langerhans' cell histiocytosis: review and report of case. ASDC Journal of Dentistry for Children. 2000; 67(4): 293-6.
[4]	Hernández-Juyol M, Boj-Quesada JR. Oral manifestations of Langerhans cell histiocytosis. Case study of a two-year-old boy. Medicina Oral: Organo Oficial de la Sociedad Espanola de Medicina Oral y de la Academia Iberoamericana de Patologia y Medicina Bucal. 2003; 8(1): 19-25.
[5]	Alston RD, Tatevossian R, McNally RJ, Kelsey A, Birch JM, Eden TO. Incidence and survival of childhood Langerhans cell histiocytosis in Northwest England from 1954 to 1998. Pediatric blood & cancer. 2007; 48(5): 555-60. https://doi.org/10.1002/pbc.20884
[6]	Guyot‐Goubin A, Donadieu J, Barkaoui M, Bellec S, Thomas C, Clavel J. Descriptive epidemiology of childhood Langerhans cell histiocytosis in France, 2000–2004. Pediatric blood & cancer. 2008; 51(1): 71-5. https://doi.org/10.1002/pbc.21498
[7]	Salotti JA, Nanduri V, Pearce MS, Parker L, Lynn R, Windebank KP. Incidence and clinical features of Langerhans cell histiocytosis in the UK and Ireland. Archives of disease in childhood. 2009; 94(5): 376-80. https://doi.org/10.1136/adc.2008.144527
[8]	Stålemark H, Laurencikas E, Karis J, Gavhed D, Fadeel B, Henter JI. Incidence of Langerhans cell histiocytosis in children: a population‐based study. Pediatric blood & cancer. 2008; 51(1): 76-81. https://doi.org/10.1002/pbc.21504
[9]	Baumgartner I, von Hochstetter A, Baumert B, Luetolf U, Follath F. Langerhans'‐cell histiocytosis in adults. Medical and Pediatric Oncology: The Official Journal of SIOP—International Society of Pediatric Oncology (Societé Internationale d'Oncologie Pédiatrique. 1997; 28(1): 9-14. https://doi.org/10.1002/(SICI)1096-911X(199701)28:1%3C9::AID-MPO3%3E3.0.CO;2-P
[10]	Goyal G, Shah MV, Hook CC, Wolanskyj AP, Call TG, Rech KL, et al. Adult disseminated Langerhans cell histiocytosis: incidence, racial disparities and long‐term outcomes. British journal of haematology. 2018; 182(4). https://doi.org/10.1111/bjh.14818
[11]	Badalian-Very G, Vergilio J-A, Degar BA, MacConaill LE, Brandner B, Calicchio ML, et al. Recurrent BRAF mutations in Langerhans cell histiocytosis. Blood, The Journal of the American Society of Hematology. 2010; 116(11): 1919-23. https://doi.org/10.1182/blood-2010-04-279083
[12]	Berres M-L, Lim KPH, Peters T, Price J, Takizawa H, Salmon H, et al. BRAF-V600E expression in precursor versus differentiated dendritic cells defines clinically distinct LCH risk groups. Journal of Experimental Medicine. 2014; 211(4): 669-83. https://doi.org/10.1084/jem.20130977
[13]	Hyman DM, Puzanov I, Subbiah V, Faris JE, Chau I, Blay J-Y, et al. Vemurafenib in multiple nonmelanoma cancers with BRAF V600 mutations. New England Journal of Medicine. 2015; 373(8): 726-36. https://doi.org/10.1056/nejmoa1502309
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[15]	Neckel N, Lissat A, von Stackelberg A, Thieme N, Doueiri M-S, Spors B, et al. Primary oral manifestation of Langerhans cell histiocytosis refractory to conventional therapy but susceptible to BRAF-specific treatment: a case report and review of the literature. Therapeutic Advances in Medical Oncology. 2019; 11: 1758835919878013. https://doi.org/10.1177/1758835919878013
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APA Style

Mostafazadeh, S., Motlagh, M. F., Rasooli, N. (2025). Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features. International Journal of Clinical Oral and Maxillofacial Surgery, 11(2), 63-68. https://doi.org/10.11648/j.ijcoms.20251102.12

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ACS Style

Mostafazadeh, S.; Motlagh, M. F.; Rasooli, N. Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features. Int. J. Clin. Oral Maxillofac. Surg. 2025, 11(2), 63-68. doi: 10.11648/j.ijcoms.20251102.12

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AMA Style

Mostafazadeh S, Motlagh MF, Rasooli N. Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features. Int J Clin Oral Maxillofac Surg. 2025;11(2):63-68. doi: 10.11648/j.ijcoms.20251102.12

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@article{10.11648/j.ijcoms.20251102.12,
  author = {Samira Mostafazadeh and Masoud Fallahi Motlagh and Nesa Rasooli},
  title = {Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features
},
  journal = {International Journal of Clinical Oral and Maxillofacial Surgery},
  volume = {11},
  number = {2},
  pages = {63-68},
  doi = {10.11648/j.ijcoms.20251102.12},
  url = {https://doi.org/10.11648/j.ijcoms.20251102.12},
  eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ijcoms.20251102.12},
  abstract = {Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.
},
 year = {2025}
}

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TY  - JOUR
T1  - Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

AU  - Samira Mostafazadeh
AU  - Masoud Fallahi Motlagh
AU  - Nesa Rasooli
Y1  - 2025/10/28
PY  - 2025
N1  - https://doi.org/10.11648/j.ijcoms.20251102.12
DO  - 10.11648/j.ijcoms.20251102.12
T2  - International Journal of Clinical Oral and Maxillofacial Surgery
JF  - International Journal of Clinical Oral and Maxillofacial Surgery
JO  - International Journal of Clinical Oral and Maxillofacial Surgery
SP  - 63
EP  - 68
PB  - Science Publishing Group
SN  - 2472-1344
UR  - https://doi.org/10.11648/j.ijcoms.20251102.12
AB  - Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.

VL  - 11
IS  - 2
ER  -

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Author Information

Samira Mostafazadeh

Department of Oral and Maxillofacial Pathology, Urmia University of Medical Sciences, Urmia, Iran

Contact Email

http://orcid.org/0000-0002-8620-6127
Masoud Fallahi Motlagh

Department of Oral and Maxillofacial Surgery, Azarbayjan Hospital, Urmia, Iran

Contact Email

http://orcid.org/0000-0002-0612-8852
Nesa Rasooli

Faculty of Dentistry, Urmia University of Medical Sciences, Urmia, Iran

Contact Email

http://orcid.org/0009-0002-4387-4827

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APA Style

Mostafazadeh, S., Motlagh, M. F., Rasooli, N. (2025). Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features. International Journal of Clinical Oral and Maxillofacial Surgery, 11(2), 63-68. https://doi.org/10.11648/j.ijcoms.20251102.12

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ACS Style

Mostafazadeh, S.; Motlagh, M. F.; Rasooli, N. Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features. Int. J. Clin. Oral Maxillofac. Surg. 2025, 11(2), 63-68. doi: 10.11648/j.ijcoms.20251102.12

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AMA Style

Mostafazadeh S, Motlagh MF, Rasooli N. Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features. Int J Clin Oral Maxillofac Surg. 2025;11(2):63-68. doi: 10.11648/j.ijcoms.20251102.12

Copy | Download

@article{10.11648/j.ijcoms.20251102.12,
  author = {Samira Mostafazadeh and Masoud Fallahi Motlagh and Nesa Rasooli},
  title = {Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features
},
  journal = {International Journal of Clinical Oral and Maxillofacial Surgery},
  volume = {11},
  number = {2},
  pages = {63-68},
  doi = {10.11648/j.ijcoms.20251102.12},
  url = {https://doi.org/10.11648/j.ijcoms.20251102.12},
  eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ijcoms.20251102.12},
  abstract = {Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.
},
 year = {2025}
}

Copy | Download

TY  - JOUR
T1  - Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

AU  - Samira Mostafazadeh
AU  - Masoud Fallahi Motlagh
AU  - Nesa Rasooli
Y1  - 2025/10/28
PY  - 2025
N1  - https://doi.org/10.11648/j.ijcoms.20251102.12
DO  - 10.11648/j.ijcoms.20251102.12
T2  - International Journal of Clinical Oral and Maxillofacial Surgery
JF  - International Journal of Clinical Oral and Maxillofacial Surgery
JO  - International Journal of Clinical Oral and Maxillofacial Surgery
SP  - 63
EP  - 68
PB  - Science Publishing Group
SN  - 2472-1344
UR  - https://doi.org/10.11648/j.ijcoms.20251102.12
AB  - Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.

VL  - 11
IS  - 2
ER  -

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