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A Case Of Langerhans Cell Histiocytosis Of The Mandible That Spontaneously Regressed After Biopsy In A Child

Ono, K. 2021Other/Unknown

Ono, K., Okui, T., Kunisada, Y., Obata, K., Masui, M., Ryumon, S., Ibaragi, S., Nakamura, T., & Sasaki, A. (2021). A case of langerhans cell histiocytosis of the mandible that spontaneously regressed after biopsy in a child. Clinical case reports, 9(6), e04321. https://doi.org/10.1002/ccr3.4321

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Case Details

Disease Location

Mandible

Personal Characteristics

4-year-old boy

Clinical Characteristics

Painful swelling of his left cheek. CT and MRI showed osteolysis and mass formation in the left angle of the mandible. At the patient's first visit, his left cheek showed painful swelling, and the submandibular lymph nodes were palpable, tender, and mobile. Incisional biopsy showed granulation-like tissue. Proliferation of langerhans cells with large and distinct cytoplasm and coffee bean-like nuclei was observed. Additionally, many eosinophilic infiltrates were involved, and some areas showed bleeding, hyperplasia of the capillaries, congestion, and necrotic tissue. Immunohistochemical (ihc) staining identified these clusters of cells as langerhans cells due to their intense immunoreactivity for s-100 protein and cd1a. This patient was diagnosed with single-system single-site (ss)-type langerhans cell histiocytosis (lch)

Remission Characteristics

CT scans taken two months after the biopsy showed no apparent exacerbation of the lesions, and a CT scan at six months showed a marked reduction in the size of the lesion, regeneration of the buccolingual cortex, and no buccolingual periosteal response

Treatment & Mechanisms

Clinical Treatment

Biopsy