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Fibrous dysplasia is a benign developmental disorder of the bone. Ten percent of patients with monostotic fibrous dysplasia have craniofacial bone involvement. Twenty-six patients were followed up at the Neurosurgery and Otolaryngology, Head and Neck Surgery Departments between 1990 and 2004. The surgery was performed in 14 patients with considerable cosmetic and functional disturbance. Twelve patients who had mild pain and mass at the site of the lesion were followed up. Cosmetic and functional results, symptom analysis, and frequency of site of involvement were reviewed and compared with previous series. Cranial involvement including frontal, parietal, sphenoidal, and occipital bones was found in 13 (52%) patients. The next common site was the maxillary bone in 9 (33%) patients. Four patients had orbital decompression caused by fronto-orbital and sphenoid bone involvement. None of the patients had any major complication of surgery. Because of its benign nature and decreased growth rate after adolescence, surgical treatment should be reserved for those with functional impairment or severe cosmetic disturbance.
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A 72-year-old woman sought treatment with a rare monostotic fibrous dysplasia occurring in the mandible and involving the mandibular canal. Her chief complaint was swelling, which had appeared about 2 years previously, had enlarged gradually, and was associated with spontaneous pain. X-ray film examination revealed a ground-glass opaque image of blurred demarcation, and 99mTc-methylene diphosphonate bone scintigraphy disclosed an area of marked radioisotope uptake. Histopathologic examination revealed the area of bone marrow and spongy bone to be replaced by fibrous tissue, irregular beam-shaped woven bone, and lamellar bone. Contouring of the expanded portion of the bone and surgical decompression of the mandibular canal were performed with good results.
A swelling in the left maxilla of a 46-year-old man, which had been enlarging for several years, was diagnosed as fibrous dysplasia. The disease usually affects only one bone and the lesion is benign; however, it can involve multiple bones and systemic complications may develop. A diagnosis of fibrous dysplasia, indicated by radiographic evaluation, should be confirmed by biopsy examination.
Seventeen patients with benign cystic osseous lesions were treated by curettage and grafting using allogenic decalcified bone. Human bones were partially decalcified using 0.6 N HCl and preserved in 90% ethanol in a deep freezer at -16 degrees C. The cystic lesions were: 5 cases of fibrous dysplasia, 4 aneurysmal bone cysts, 3 simple bone cysts, 2 giant-cell tumours, 1 chondromyxoid fibroma, 1 non-ossifying fibroma and 1 fibrous cortical defect. The bones involved were: femur, tibia, humerus, fibula and calcaneum. Infection was a complication in three patients. In two of these it did not interfere with healing, but in one it persisted for more than 1 year with partial resorption of the graft. The time to adequate incorporation of the graft varied from 6 to 9 months in children and 9 to 15 months in adults. The overall response compares favourably with that to allograft from more sophisticated bone banks.
Children with monostotic and polyostotic bone dysplasias often exhibit localized bone overgrowth. We investigated the presence of nuclear estrogen and nuclear progesterone receptors by solid-phase radioimmunoassay, immunocytochemistry, and radioligand binding in osteoblast cell cultures derived from the areas of overgrowth of membranous bone, noninvolved membranous bone, and normal membranous bone from children undergoing elective craniotomy. Membranous bone of normal children had demonstrable levels of nuclear estrogen and progesterone receptors identified by radioimmunoassay and immunocytochemical assay. Two- to threefold increased levels of these receptors (p less than 0.001 versus normals) were found in cultures derived from the involved bone of two children with monostotic fibrous dysplasia and in one patient with polyostotic dysplasia (McCune-Albright syndrome). The noninvolved bone in our patients with fibrous dysplasia exhibited nuclear sex steroid hormone receptor levels similar to those in the normal children. Radioligand binding studies demonstrated increased sex steroid hormone receptors in cultures derived from involved osteoblasts. The presence of an increased level of sex steroid hormone receptor was accompanied by increased alkaline phosphatase activity and increased production of osteocalcin in vitro compared to normal or noninvolved bone. The mechanisms by which sex steroid hormone receptor levels are increased in the ostotic dysplasias remain to be established.
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Two cases are reported with osteosarcomatous transformation in fibrous dysplasia, one in the skull and one in the iliac bone. In both cases the patient was known to have longstanding polyostotic fibrous dysplasia; no radiation therapy was ever given. The incidence of sarcomatous transformation in fibrous dysplasia in the files of the Netherlands Committee on Bone Tumours is 0.5%. When strict criteria for spontaneous malignant transformation are used, the actual incidence is probably lower than is suggested.
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We describe an unusual case of monostotic fibrous dysplasia involving the right sphenoid bone with gradual loss of vision occurring in a sexually precocious young woman. Vision remained stable after a four-year follow-up surgical decompression of the right optic canal.
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There have been no reports on choromosomal aberrations of benign bone tumors revealed by comparative genomic hybridization (CGH). CGH analysis of benign tumors may be useful in understanding the mechanism of tumorigenesis with comparisons to malignant tumors. There were 4 tumors (2 enchondromas, one chondromyxoid fibroma, and one osteoid osteoma) and 8 tumor-like conditions (4 aneurysmal bone cysts (ABCs), one eosinophilic granuloma, one fibrous dysplasia, one solitary bone cyst, and one Rosai-Dorfman disease) available for analysis. One of 2 enchondromas and one of 4 ABCs exhibited rapid growth. Six lesions showed chromosomal aberrations, while 6 others did not. The most frequent aberrations were the loss of a whole chromosome-19 in 6 cases, the loss of chromosome-arm 22q in 4 cases, and the loss of chromosome-arm 17p in 3 cases. Gains were seen in 13q21 in 2 cartilaginous tumors and at 12q15-q21 in eosinophilic granulomas. Therefore, in benign bone tumors or tumor-like lesions, chromosomal aberrations are not frequent; however, some clear tendencies of clustering of aberrations can be observed.