Evidence map›Paper›PMID 40181439›Full record

ArticleJournal of medical case reports2025

Polyostotic Langerhans cell histiocytosis presenting as halitosis in a 24-year-old man: a case report.

Hassan Mirmohammad Sadeghi, Sanaz Gholami Toghchi, Fatemeh Mashhadiabbas, Milad Baseri, Maryam Mohammadalizadeh Chafjiri, Parnian Nikraftar, Ardeshir Khorsand

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Article in Journal of medical case reports, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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5 · Who and what money

Authors and funding

7 authors.

Hassan Mirmohammad SadeghiDepartment of Oral and Maxillofacial Surgery, Dentistry Faculty, Shahid Beheshti University of Medical Sciences, Velenjak St., Shahid Chamran Highway, Tehran, 1985717443, Iran.ORCID http://orcid.org/0000-0002-3132-2489
Sanaz Gholami ToghchiDepartment of Oral and Maxillofacial Pathology, Dentistry Faculty, Shahid Beheshti University of Medical Sciences, Tehran, Iran.ORCID http://orcid.org/0000-0002-6823-6509
Fatemeh MashhadiabbasDepartment of Oral and Maxillofacial Pathology, Dentistry Faculty, Shahid Beheshti University of Medical Sciences, Tehran, Iran.ORCID http://orcid.org/0000-0003-3789-1727
Milad BaseriDepartment of Oral and Maxillofacial Surgery, Dentistry Faculty, Shahid Beheshti University of Medical Sciences, Velenjak St., Shahid Chamran Highway, Tehran, 1985717443, Iran.ORCID http://orcid.org/0000-0001-8531-8699
Maryam Mohammadalizadeh ChafjiriDepartment of Oral and Maxillofacial Pathology, Dentistry Faculty, Shahid Beheshti University of Medical Sciences, Tehran, Iran.ORCID http://orcid.org/0000-0002-1889-0553
Parnian NikraftarDepartment of Radiology, Medicine Faculty, Shahid Beheshti University of Medical Sciences, Tehran, Iran.ORCID http://orcid.org/0009-0002-4525-1527
Ardeshir KhorsandDepartment of Oral and Maxillofacial Surgery, Dentistry Faculty, Shahid Beheshti University of Medical Sciences, Velenjak St., Shahid Chamran Highway, Tehran, 1985717443, Iran. Ardeshir.khorsand1374@gmail.com.ORCID http://orcid.org/0000-0002-0618-4387

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundLangerhans 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.

conclusionThis 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.

Indexed as

HalitosisHistiocytosis, Langerhans-CellCOVID-19Diagnosis, DifferentialHumansMaleYoung AdultCase reportHalitosisHistiocytosisLangerhans cellPolyostotic

Identifiers

PMID40181439
PMCPMC11969994

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