Eosinophilic granuloma: resolution of maxillofacial bony lesions following minimal intervention Report of three cases and a review of the literature

• Published in: Journal of cranio-maxillo-facial surgery 2004-06, Vol.32 (3), p.170-175
• Main Authors: Key, Steven J, O’Brien, Ciaran J, Silvester, Keith C, Crean, St-John
• Format: Article
• Language: English
• Subjects: Biological and medical sciences; Biopsy; Bone Diseases - pathology; Child; Child, Preschool; Curettage; Eosinophilic Granuloma - pathology; Facial Bones - pathology; Female; Follow-Up Studies; Fundamental and applied biological sciences. Psychology; Hematologic and hematopoietic diseases; Histiocytosis, Langerhans-Cell - pathology; Humans; Male; Mandibular Diseases - pathology; Medical sciences; Middle Aged; Minimally Invasive Surgical Procedures; Neurology; Orbital Diseases - pathology; Other diseases. Hematologic involvement in other diseases; Otorhinolaryngology. Stomatology; Remission Induction; Temporomandibular Joint Disorders - pathology; Vertebrates: nervous system and sense organs
• ISSN: 1010-5182; 1878-4119
• DOI: 10.1016/j.jcms.2004.01.004

Introduction: Langerhans’ cell histiocytosis is a collective term used to describe a group of enigmatic proliferative disorders. The natural history of the disease varies from a slow, benign, localized symptomatic bony or soft tissue lesion, to a rapidly progressive widespread multiple organ disorder which is often fatal. Eosinophilic granuloma accounts for 60–70% of all cases of Langerhans’ cell histiocytosis and can present as solitary (50–75%) or multifocal defects in bone. It occasionally presents as a localized soft tissue lesion. There are multiple treatment options but the response is unpredictable. Aims: We present three separate cases, of the maxillofacial skeleton where the lesions of eosinophilic granuloma resolved following incisional biopsy only. Patients and results: Three patients presented with solitary lesions of the maxillofacial skeleton. All were diagnosed as Langerhans’ cell histiocytosis following open curettage, which also resulted in resolution of the lesions. Follow-up has thus far been disease free. Conclusion: For some solitary Langerhans’ cell histiocytosis lesions, simple curettage is the only treatment required. The paper discusses the need to confirm the solitary nature of the disease and the need for follow-up. Reviewing the literature on the disease, the authors suggest that perhaps cellular immaturity holds the cells of the lesion in a disease state until pushed to maturity by the trauma of open curettage surgery, resulting in a complete resolution of the disease.