Advances in Oral and Maxillofacial Surgery (Jan 2022)
Recurrent eosinophilic granuloma of the maxilla in an adult female: About a rare case report
Abstract
Langerhans cell histiocytosis (LCH) is a rare idiopathic disorder of the reticulo-endothelial system with abnormal proliferation of the bone marrow derived from Langerhans cells.LCH mainly affects children and young adults, with a slight male predilection. It is clinically divided into three groups namely Letter-Siwe disease (multiple multi organ affecting LCH at very young age), Hand-Schuler-Christian disease (LCH of bone involvement exophthalmos and diabetes insipidus), and Eosinophilic Granuloma (LCH of bone, solitary or multiple).Eosinophilic Granuloma (EG) is the most benign and common form of the Langerhans cell histiocytosis, it is a rare proliferative disease that can affect single or multiple organs.It is an intraosseous destructive lesion characterized by the presence of vast numbers of eosinophils and histiocytes. It can be localized or multifocal.Most frequent signs and symptoms are pain, swelling, ulceration, gingival necrosis, alveolar bone destruction with mobility and loss of teeth.The diagnosis is confirmed by immunohistochemistry using either the anti-CD1a or anti-S100 protein antibody.The severity and prognosis depend on the type and extension of the organ involved. The prognosis of patients with a single EG lesion is generally good.The literature reveals a lack of consensus about treatment, it varies depending on the size, number, localization of the lesions, and patient's general conditions.This article describes an unusual case of eosinophilic granuloma with maxillary involvement diagnosed in a young woman of 29 years old, treated in the oral surgery department of the dental consultation and treatment center in Casablanca.Although lesions underwent clinical remission after surgery and corticosteroid, a new lesion appeared 18 months after initial therapy, indicating recurrence of eosinophilic granuloma.
