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Published on: April 22, 2019
Langerhans cell histiocytosis mimicking a residual cyst
John Lennon Silva Cunha1, Rejane Andrade de Carvalho2, Oslei Paes de Almeida1
1Department of Oral Diagnosis, Piracicaba Dental School, University of Campinas (UNICAMP), Piracicaba, Brazil.
Insights
Langerhans cell histiocytosis (LCH) rarely mimics jaw cysts. This case highlights the importance of thorough evaluation for LCH in bone lesions to ensure correct diagnosis and management.
Area of Science:
- Oncology
- Pathology
Background:
- Langerhans cell histiocytosis (LCH) is a rare myeloid neoplasm involving Langerhans-type dendritic cells.
- LCH mimicking periapical lesions is exceptionally uncommon in the literature.
Observation:
- A 45-year-old woman presented with a mandibular lesion radiographically and microscopically resembling a residual cyst.
- Histopathology revealed histiocytes with characteristic nuclear grooves, positive for S-100, CD1a, and CD207.
Findings:
- The diagnosis was solitary LCH of the mandible after excluding other sites.
- The patient remained recurrence-free for four years following conservative surgical management.
Implications:
- This case underscores the necessity of comprehensive clinical, radiographic, and microscopic assessment for mandibular bone lesions.
- Neoplasms like LCH can present as benign-appearing jaw lesions, necessitating differential diagnosis.
- Conservative treatment may be suitable for solitary LCH, but long-term follow-up is crucial.
Abstract:
Langerhans cell histiocytosis (LCH) is an uncommon myeloid neoplasm characterized by clonal neoplastic proliferation of Langerhans-type dendritic cells associated with a reactive inflammatory infiltrate composed predominantly of lymphocytes and eosinophils. Only three cases of LCH mimicking periapical lesions have been reported in the English-language literature to date. Herein, we report a rare case of LCH involving the mandible of a 45-years-old woman mimicking microscopically and radiographically a residual cyst. The patient underwent enucleation and curettage of the lesion. Microscopically, the lesion showed fibrous tissue with an intense inflammatory infiltrate and histiocytes with irregular to elongated nuclei with prominent nuclear grooves. The tumor cells were positive for S-100 protein, CD1a, and CD207. After careful evaluation through imaging tests to rule out lesions in other anatomical locations, the diagnosis was solitary LCH of the mandible. After four years of follow-up, the patient remained with no evidence of recurrence. This case emphasizes the importance of a carefully clinical, radiographic, and microscopical evaluation of bone lesions, including periapical or residual cysts, since some neoplasms can mimic common benign lesions of the jaws. Although conservative approaches to treating solitary mandibular bone lesions of LCH can be employed, long-term follow-up is strongly recommended.

