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Cranial unifocal Langerhans cell histiocytosis in children
George A Alexiou1, Evriviadis Mpairamidis, George Sfakianos
1Department of Neurosurgery, Children's Hospital "Agia Sofia", Athens, Greece.
Insights
Eosinophilic granuloma (EG) of the skull is a benign pediatric condition. Surgical excision is the primary treatment, but antibiotic therapy is an effective alternative for cosmetically sensitive areas.
Area of Science:
- Pediatric Oncology
- Skeletal Pathology
- Histopathology
Background:
- Eosinophilic granuloma (EG) is a rare, benign proliferative disorder of histiocytes.
- Cranial involvement is common in pediatric EG, necessitating accurate diagnosis and management.
- Understanding the clinical presentation and treatment outcomes is crucial for pediatric neurosurgery and oncology.
Purpose of the Study:
- To present a series of 22 pediatric cases with eosinophilic granuloma (EG) of the skull.
- To evaluate the diagnostic methods, surgical approaches, and treatment outcomes for cranial EG.
- To determine the efficacy of alternative treatments for specific EG presentations.
Main Methods:
- Retrospective review of 22 pediatric patients diagnosed with cranial EG.
- Preoperative evaluation included skull X-ray, CT, MRI (in 10 cases), and bone scintigraphy.
- Surgical excision was the primary intervention, with histopathological confirmation of EG.
Main Results:
- A male predominance was observed; the frontal bone was the most frequently affected site.
- Complete surgical excision was achieved in 19 patients; 3 cases with periorbital EG underwent biopsy and antibiotic treatment.
- No recurrence was noted during a mean follow-up of 6.2 years; antibiotic therapy resolved lesions in the biopsy-only group.
Conclusions:
- Eosinophilic granuloma (EG) of the skull is a benign entity in pediatric patients.
- Bone scintigraphy is recommended to exclude multifocal disease.
- Surgical resection is the preferred treatment, with antibiotic therapy (sulfamethoxazole and trimethoprim) as a viable alternative for specific cases.
Purpose:
The present study presents 22 cases of pediatric patients harboring an eosinophilic granuloma (EG) of the skull.
Methods:
Twenty-two patients (13 males, 9 females; mean age, 7.5 years; range, 3-14 years) with a suspected diagnosis of a cranial EG were enrolled in the study. They all had been preoperatively evaluated by skull x-ray and computed tomography, whereas 10 patients additionally underwent magnetic resonance imaging. To rule out a multifocal disease, scintigraphy was performed in all cases preoperatively. Surgical excision of the lesions was performed, and EG was proven histopathologically.
Results:
There was a male predominance. Frontal bone was the most common affected bone. One patient had a multifocal disease. Total excision of the lesion was performed in 19 of 22 patients. No patient received postoperative radiotherapy. In the remaining 3 cases because of the periorbital localization of the EG and the subsequent risk of disfigurement, only a biopsy was performed. These patients were treated with sulfamethoxazole and trimethoprim for 6 months. All 3 lesions were resolved. The follow-up examinations ranged from 6 months to 17 years, with a mean follow-up of 6.2 years, and no tumor recurrence was noted.
Conclusions:
We conclude that EG is a benign disease. At the time of diagnosis, a bone scan should be performed to rule out a multifocal disease. Surgical resection is the treatment of choice. Nevertheless, for lesions in which excision can cause cosmetic defects, administration of sulfamethoxazole and trimethoprim after biopsy appeared to be an effective alternative treatment.