Cases reported • Hyperostosis; Bone Hypertrophy

11/24. Cortical hyperostosis: a complication of prostaglandin E1 treatment in congenital heart disease.

Prostaglandin E1 (PGE1) is an essential drug for infants with ductal-dependent congenital heart disease. Cortical hyperostosis of the long bones is one of the complications during and after PGE1 therapy and should be considered in the differential diagnosis of periostitis in the infant.

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12/24. osteogenesis of the mandible associated with implant reconstruction: a patient report.

A patient report is presented illustrating bone growth of the posterior mandible associated with a fixed implant reconstruction of the Branemark type. In this instance, the mandible had grown vertically approximately 3 mm and was causing discomfort beneath the cantilever sections of the prosthesis because of gingival impingement between the restoration and the bony mandible. The literature relative to osteogenesis associated with osseointegrated implants is reviewed.

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13/24. Primary intraosseous meningioma. Case report.

Primary intraosseous meningioma of the skull is an uncommon lesion often confused preoperatively with a primary bone tumor of the skull. The case of an intraosseous meningioma without dural connection or association with a cranial suture is reported that was radiographically diagnosed as Paget's disease and initially treated conservatively. Persistent headache prompted a biopsy, yielding a benign, heavily ossified, and psammomatous meningioma. The lesion was treated with wide surgical excision and acrylic cranioplasty. Subsequent review of the literature has revealed 35 additional cases of purely intraosseous meningioma. An analysis of these 36 cases demonstrates a mean patient age at diagnosis of 45 years and a 2:1 female preponderance, with the majority of lesions associated with cranial sutures. Sixty-four percent of the lesions were hyperostotic on plain skull x-ray films; the rest were osteolytic or a mixture of both. There was no relationship to prior trauma. The treatment of choice is wide surgical resection followed by cranial reconstruction.

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14/24. Is diffuse sclerosing osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome? Analysis of seven cases.

Diffuse sclerosing osteomyelitis of the mandible has been described as a localized disease of unknown origin. We offer evidence that mandibular involvement in the recently described synovitis acne pustulosis hyperostosis osteitis syndrome exactly fits the accepted description for diffuse sclerosing osteomyelitis of the mandible. The clinical, radiologic, and pathologic findings in seven such cases are presented and discussed.

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15/24. Sclerosteosis in a Spanish male: first report in a person of Mediterranean origin.

We report the first observation of sclerosteosis in spain. To the best of our knowledge, this is the first case of sclerosteosis in a person of Mediterranean origin with no known Dutch ancestors. He has the characteristic phenotype of the disease with right facial nerve palsy and syndactyly and the typical radiological features, including generalised bone sclerosis and cortical widening of the tubular bones.

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16/24. hyperostosis and osteoarthritis in patients surviving after tetanus.

Twenty patients (12 men and 8 women) who had survived tetanus were examined for bone and joint abnormalities, 1-9 years after the disease. The radiographic findings in the elbows were abnormal in 13 out of 20 patients, as were the shoulders of all 3 patients examined. Insertion hyperostoses were found at the elbow and/or in the shoulder region in 5 (25%) of the patients. The hyperostoses were bilateral in 3 patients. One of the patients with hyperostoses also had calcifications in the soft tissue of the elbow. The abnormalities were associated with mild clinical symptoms. Trauma to the periosteum caused by repeated traction of muscular contractions is possibly the causative mechanism. Twelve patients (60%) had osteoarthritis of the elbow joint presumably secondary to the violent stress to the joint.

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17/24. An aseptic inflammation of the clavicle in a patient with Crohn's disease. A potential manifestation of the SAPHO syndrome.

A female patient with Crohn's disease developed an septic osteoarticular involvement of the left clavicle and sterno- and acromioclavicular regions. Repeated surgical revisions combined with a broad-spectrum antimicrobial treatment could not prevent the progress of the disease. However, the patient started to improve after the diagnosis of the SAPHO (synovitis, acne, pustulosis, hyperostosis and osteomyelitis) syndrome was made and the dose of her immunosuppressive therapy increased. This patient reminds of the existence of extraintestinal aseptic infections in association with inflammatory bowel disease (IBD). Moreover, it may provide further evidence on the significant association between SAPHO and IBD.

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18/24. Intracranial and extracranial reduction osteoplasty for craniodiaphyseal dysplasia.

Craniodiaphyseal dysplasia is a rare, sporadic form of craniotubular bone dysplasia, characterized by massive generalized hyperostosis and sclerosis, particularly of the skull and facial bones, leading to severe deformity. The clinical course is typically characterized by progressive encroachment of the craniofacial foramina and brain by the relentless deposition of bone. Compression of cranial nerves, the foramen magnum, and intracranial contents commonly leads to blindness, loss of hearing, and death. This report describes a unique case of craniodiaphyseal dysplasia manifesting with asymmetric craniofacial and axial hyperostosis. The tubular bones demonstrated the characteristic diaphyseal endostosis, undertubulation, and relative overgrowth on the involved side. Significant brain compression with signs and symptoms of increased intracranial pressure was managed successfully with decompressing craniectomy at age 12 years, enlarging the anterior and middle fossae. Calvarial thickness measured nearly 4 cm. Further calvarial, midfacial, and mandibular recontouring were performed 6 and 22 months later. Follow-up in our case indicates that close observation is mandatory to manage further progression of the disease.

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19/24. SAPHO syndrome and pyoderma gangrenosum: is it fortuitous?

pyoderma gangrenosum is well known as an associated feature of inflammatory bowel disease (IBD). Recently, higher than normal prevalence of IBD in patients with the SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome has been reported. However, the association of pyoderma gangrenosum with SAPHO syndrome without definitely excluded IBD has not been reported. We describe a case that suggests a possible connection between these 2 entities.

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20/24. Hyperostotic and destructive osteoarthritis in a patient with vitamin a intoxication syndrome: a case report.

The authors present a case report of a 59-year-old female suffering from hyperlipidemia who developed chronic vitamin a intoxication syndrome after ingestion of 30,000 IU retinol/daily over a period of six years. The patient's main complaints included severe headaches, morning nausea, myalgias and disability around the hip, knee, and ankle joints. Radiologically, hyperostosis of the acetabular circumference and the spine was demonstrated. Because of rapidly increasing pain, total hip replacement was performed. histology of cross sections from the femoral head revealed destructive osteoarthritis. Since no other causative reason was found, retinol may not only be responsible for hyperostotic bone and soft tissue formations but may perhaps also account for rapid progressing of degenerative joint disease. Despite the cessation of vitamin a intake the clinical symptoms persisted due to hyperlipidemia. The enlarged number of chylomicrons and the higher fraction of very low density lipoproteins may represent a second retinyl ester pool in case of overloaded fat storing Ito-cells in the liver. Therefore, rheumatological treatment reducing risk factors such as hyperlipidemia is mandatory.

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