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21/55. Infantile cortical hyperostosis Caffey's disease.

    A 2 1/2-month-old girl with cortical hyperostosis (Caffey's disease) is described with a brief review of the literature. This is the first case reported from iran.
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22/55. Calvarial hyperostosis: a benign X-linked recessive disorder.

    We report a family with what appears to be a unique X-linked recessive disorder of isolated hyperostosis of the calvarium. Although irregularity of the calvarium and exophytic prominences of the frontoparietal bones were apparent in infancy, premature cranial suture closure did not occur and there was no evidence of increased intracranial pressure despite a Luckenshadel appearance of the skull. Other membranous bones and the tubular bones were not involved. Calvarial bone biopsy from one patient showed vacuolated histiocytes suggesting a storage disease; however, neurologic deterioration, hepatosplenomegaly, and dysostosis multiplex did not occur. The affected family members had normal stature, normal occipitofrontal circumference, and no other medical problems. The biochemical basis of this disorder is not known. Although storage of abnormal material is possible, the long-term prognosis seems favorable.
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23/55. Infantile cortical hyperostosis with raised immunoglobulins.

    Two cases of infantile cortical hyperostosis are reported. Both had raised immunoglobulins. Particularly remarkable were the IgA and IgM levels, a finding infrequently reported.
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24/55. Soft-tissue swelling in two neonates during prostaglandin E1 therapy.

    Two small neonates, one with hypoplastic right heart syndrome, and the other with tetralogy of fallot and pulmonary atresia, were treated with intravenous prostaglandin E1 (Prostyn VR, Upjohn, Kalamazoo, michigan) for 96 and 33 days, respectively. They developed limited cortical hyperostosis, but marked soft tissue swellings in all extremities. The changes were noted after PGE1 treatment for four weeks and one week, respectively. A skin biopsy was performed in one patient and revealed edematous changes and arteriolar wall abnormalities. We add this peripheral hard edema to the list of complications of PGE1 therapy, and speculate as to its pathophysiology.
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25/55. Caffey's disease in Cape Town.

    Six cases of infantile cortical hyperostosis or Caffey's disease are presented, with opinions as to its prevalence, incidence and origin.
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26/55. Infantile cortical hyperostosis with osteomyelitis of the humerus.

    A case is presented in which a delay in diagnosis and treatment of osteomyelitis occurred because of diagnostic confusion between the patient's osteomyelitis and his coincident infantile cortical hyperostosis.
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27/55. Infantile cortical hyperostosis with intramedullary lesions.

    A case report of an infant with the clinical, radiological, and histological presentations of infantile cortical hyperostosis is presented. Besides the classically described subperiosteal thickening, the present patient radiographically also had well-circumscribed intramedullary lytic lesions in both proximal tibial metaphyses, which subsequently disappeared in 3 months. These lesions have not been previously described in the literature. Resolution of these lesions suggests they may be part of the radiological picture of infantile cortical hyperostosis and probably may be managed by observation.
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28/55. Infantile cortical hyperostosis with unusual clinical manifestations.

    Infantile cortical hyperostosis may mimic a variety of serious conditions in early infancy, such as trauma, vitamin deficiencies, infection, or tumors, In a patient in good general condition there are typical roentgenological features often with involvement of the mandible that are diagnostic of the condition. In 3 cases of infantile cortical hyperostosis (Caffey's disease), one had the clinical picture of Erb's palsy and subsequently developed hyperostosis of the scapula. The second, had a "pulled elbow" and radial nerve palsy and later developed hyperostotic changes in the humerus and radius on the same side. The third was referred for treatment of congenital torticollis and found to develop hyperostosis of the clavicle on the same side. The recognition of this condition obviates unnecessary prolonged investigation and treatment.
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29/55. Caffey's disease: a case originating in the first metatarsal and review of a 12 year experience.

    This paper describes an infant with Caffey's disease who presented with swelling of the first metatarsal shown as hyperostosis histologically. He then developed scapular sclerosis and new bone formation. The mandible became involved 26 days after onset. A review of a 12 year experience with Caffey's disease (33 patients) disclosed a definite decrease in the number and percentage of patients seen since 1968.
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30/55. Infantile cortical hyperostosis of the ribs (Caffey's disease) without mandibular involvement.

    Infantile cortical hyperostosis (ICH), or Caffey's disease, first reported by Caffey and Silverman in 1945, is a benign condition characterized radiographically by corticoperiosteal thickening of bone with subperiosteal new bone formation. Sites of occurrence vary, with the mandible being involved in 75%-80% of cases. The following is a case report of ICH limited to four contiguous ribs with no evidence of mandibular involvement.
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