Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report
Abstract Background Langerhans cell histiocytosis is a rare disease of the reticuloendothelial system. This report presents a novel case of Langerhans cell histiocytosis with systemic involvement that started with a simple chief complaint. The reporting of this case raises awareness of the distincti...
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BMC
2025-04-01
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| Series: | Journal of Medical Case Reports |
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| Online Access: | https://doi.org/10.1186/s13256-025-05062-z |
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| author | Hassan Mirmohammad Sadeghi Sanaz Gholami Toghchi Fatemeh Mashhadiabbas Milad Baseri Maryam Mohammadalizadeh Chafjiri Parnian Nikraftar Ardeshir Khorsand |
| author_facet | Hassan Mirmohammad Sadeghi Sanaz Gholami Toghchi Fatemeh Mashhadiabbas Milad Baseri Maryam Mohammadalizadeh Chafjiri Parnian Nikraftar Ardeshir Khorsand |
| author_sort | Hassan Mirmohammad Sadeghi |
| collection | DOAJ |
| description | Abstract Background Langerhans cell histiocytosis is a rare disease of the reticuloendothelial system. This report presents a novel case of Langerhans cell histiocytosis with systemic involvement that started with a simple chief complaint. The reporting of this case raises awareness of the distinctive characteristics of this challenging disorder. Case presentation A 24-year-old male patient of Persian ethnicity presented with a chief complaint of halitosis following coronavirus disease 2019 recovery to his general dentist’s office. Intraoral and extraoral examinations revealed no specific problem. In the follow-up session after phase I periodontal treatment, teeth sensitivity to cold stimulus was evident, and radiographs revealed a large lytic intraosseous lesion in the mandible. An incisional biopsy revealed Langerhans cells and a positive reaction to Langerin and cluster of differentiation 1a, thus, he was diagnosed with Langerhans cell histiocytosis. After performing positron emission tomography with fluoro-2-deoxyglucose and computed tomography, magnetic resonance imaging, and cone beam computed tomography, owing to generalized disease involvement, the patient was referred to an oncologist. Ultimately, it was found that the patient’s childhood health issues, including endocrine problems, were likely caused by an undiagnosed Langerhans cell histiocytosis. The oncologist chose denosumab, vinblastine, etoposide, 6-mercaptopurine, methotrexate, and pegfilgrastim regimen. The follow-up was not possible as the patient died following an accident. Conclusion This reveals the vitality of the early diagnosis of Langerhans cell histiocytosis to prevent disease progression. Awareness of diverse and nonpathognomonic manifestations of Langerhans cell histiocytosis, proper medical interview and history taking, and the use of routine radiographs may aid clinicians in lowering morbidity and mortality rates associated with such conditions. |
| format | Article |
| id | doaj-art-02e3415d009841ecb97ff085a22fbdcd |
| institution | DOAJ |
| issn | 1752-1947 |
| language | English |
| publishDate | 2025-04-01 |
| publisher | BMC |
| record_format | Article |
| series | Journal of Medical Case Reports |
| spelling | doaj-art-02e3415d009841ecb97ff085a22fbdcd2025-08-20T03:07:41ZengBMCJournal of Medical Case Reports1752-19472025-04-0119111310.1186/s13256-025-05062-zPolyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case reportHassan Mirmohammad Sadeghi0Sanaz Gholami Toghchi1Fatemeh Mashhadiabbas2Milad Baseri3Maryam Mohammadalizadeh Chafjiri4Parnian Nikraftar5Ardeshir Khorsand6Department of Oral and Maxillofacial Surgery, Dentistry Faculty, Shahid Beheshti University of Medical SciencesDepartment of Oral and Maxillofacial Pathology, Dentistry Faculty, Shahid Beheshti University of Medical SciencesDepartment of Oral and Maxillofacial Pathology, Dentistry Faculty, Shahid Beheshti University of Medical SciencesDepartment of Oral and Maxillofacial Surgery, Dentistry Faculty, Shahid Beheshti University of Medical SciencesDepartment of Oral and Maxillofacial Pathology, Dentistry Faculty, Shahid Beheshti University of Medical SciencesDepartment of Radiology, Medicine Faculty, Shahid Beheshti University of Medical SciencesDepartment of Oral and Maxillofacial Surgery, Dentistry Faculty, Shahid Beheshti University of Medical SciencesAbstract Background Langerhans cell histiocytosis is a rare disease of the reticuloendothelial system. This report presents a novel case of Langerhans cell histiocytosis with systemic involvement that started with a simple chief complaint. The reporting of this case raises awareness of the distinctive characteristics of this challenging disorder. Case presentation A 24-year-old male patient of Persian ethnicity presented with a chief complaint of halitosis following coronavirus disease 2019 recovery to his general dentist’s office. Intraoral and extraoral examinations revealed no specific problem. In the follow-up session after phase I periodontal treatment, teeth sensitivity to cold stimulus was evident, and radiographs revealed a large lytic intraosseous lesion in the mandible. An incisional biopsy revealed Langerhans cells and a positive reaction to Langerin and cluster of differentiation 1a, thus, he was diagnosed with Langerhans cell histiocytosis. After performing positron emission tomography with fluoro-2-deoxyglucose and computed tomography, magnetic resonance imaging, and cone beam computed tomography, owing to generalized disease involvement, the patient was referred to an oncologist. Ultimately, it was found that the patient’s childhood health issues, including endocrine problems, were likely caused by an undiagnosed Langerhans cell histiocytosis. The oncologist chose denosumab, vinblastine, etoposide, 6-mercaptopurine, methotrexate, and pegfilgrastim regimen. The follow-up was not possible as the patient died following an accident. Conclusion This reveals the vitality of the early diagnosis of Langerhans cell histiocytosis to prevent disease progression. Awareness of diverse and nonpathognomonic manifestations of Langerhans cell histiocytosis, proper medical interview and history taking, and the use of routine radiographs may aid clinicians in lowering morbidity and mortality rates associated with such conditions.https://doi.org/10.1186/s13256-025-05062-zPolyostoticLangerhans cellHistiocytosisHalitosisCase report |
| spellingShingle | Hassan Mirmohammad Sadeghi Sanaz Gholami Toghchi Fatemeh Mashhadiabbas Milad Baseri Maryam Mohammadalizadeh Chafjiri Parnian Nikraftar Ardeshir Khorsand Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report Journal of Medical Case Reports Polyostotic Langerhans cell Histiocytosis Halitosis Case report |
| title | Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report |
| title_full | Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report |
| title_fullStr | Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report |
| title_full_unstemmed | Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report |
| title_short | Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report |
| title_sort | polyostotic langerhans cell histiocytosis presenting as halitosis in a 24 year old man a case report |
| topic | Polyostotic Langerhans cell Histiocytosis Halitosis Case report |
| url | https://doi.org/10.1186/s13256-025-05062-z |
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