Resolution of blood eosinophilia and limited mouth opening after short-term follow-up in a pediatric Langerhans cell histiocytosis case

Indian J Otolaryngol Head Neck Surg. 2023 Sep;75(3):2568-2571. doi: 10.1007/s12070-023-03741-1. Epub 2023 May 2.

Abstract

Introduction: Langerhans cell histiocytosis (LCH) is an inflammatory myeloid neoplasia that often affects children, presenting a broad clinical spectrum.

Methods: Here, we report a 13-year-old male Salvadorian patient who was referred presenting a nodular swelling at the mandibular angle region, mildly symptomatic, few weeks ago, which relevantly was associated with limited mouth opening. Intraoral examination was unremarkable. Imaginological exams revealed an osteolytic lesion affecting the vestibular cortex at the right mandibular angle. The blood test results were normal, except for eosinophilia (21%; absolute eosinophil count 4 × 109/L). After an incisional biopsy, microscopical and immunohistochemical analyses were consistent with LCH diagnosis, which corresponded to a single system-single site category. After a few weeks, the mandibular movements were re-established, and complete resolution of blood eosinophilia was observed.

Conclusion: LCH with blood eosinophilia is rarely reported. To our knowledge, 3 cases have been previously published.

Keywords: Blood eosinophilia; Immunohistochemistry; Langerhans cell histiocytosis; Mandible; Restriction mouth opening.