Cases reported "Bone Resorption"

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1/13. Severe osteoporosis in familial hajdu-cheney syndrome: progression of acro-osteolysis and osteoporosis during long-term follow-up.

    hajdu-cheney syndrome is an autosomal dominant inherited osteodysplastic bone disease with the hallmarks of acro-osteolysis, skull deformations, and generalized osteoporosis. Very few patients have been followed long-term with respect to the prognosis of acro-osteolysis and osteoporosis. Here we describe a 39-year-old woman and her 19-year-old daughter who are both affected with the hajdu-cheney syndrome. Skeletal lesions were followed in the mother between the ages of 22 and 39 years. The acro-osteolytic lesions progressed markedly and caused shortening of several fingers; some end phalanges had completely disappeared. Severe spinal osteoporosis with serial vertebral fractures was found at the age of 22 years. New vertebral fractures developed until the age of 33 years, but did not progress afterward. High turnover osteoporosis was found in the bone histology of iliac crest biopsies performed at the ages of 22 and 34 years. Bone mineral content (BMC) was strikingly decreased at the age of 34 years (T score -5.1 SD) and did not significantly change during further follow-up. In the daughter, BMC failed to increase between the ages of 12 and 19 years and was also markedly decreased (T score -4.4 SD). This suggests that osteoporosis in hajdu-cheney syndrome is related to a low peak bone mass and a high bone turnover, leading to insufficient bone formation compared with the increased bone resorption.
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2/13. Rapidly progressive destructive arthropathy of the shoulder--a viewpoint on pathogenesis.

    Two cases of rapidly progressive destructive shoulder arthropathy are described. Both patients were elderly women with generalized osteoarthropathy who had used forearm crutches for walking. Their shoulder disease was characterized by large, relatively painless effusions, low numbers of infiltrating leukocytes, biopsy evidence of mild inflammation, and radiological evidence of severe joint damage. Case 1 developed persistent drainage from a joint sinus, eventually necessitating arthrodesis. This complication has not previously been described in this condition. Hydroxyapatite crystals were demonstrated in the joint fluid in both cases but their pathological role is uncertain. We suggest that bearing weight through the upper limbs may hasten the development of degenerative arthritis in the shoulder, possibly by contributing to mechanical disruption of the rotator cuff.
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3/13. Idiopathic familial acroosteolysis: histomorphometric study of bone and literature review of the hajdu-cheney syndrome.

    We describe a 27-year-old man with familial acroosteolysis involving 9 fingers. Bone biopsy of an affected digit showed osteolysis with no tetracycline deposition, rare osteoclasts, increased vascularity, and numerous mast cells. In contrast, the iliac crest bone showed active bone remodeling and normal double-tetracycline labeling. We believe mast cells deserve further study as possible factors in this form of localized osteolysis.
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4/13. Radiologic case study. Neuropathic osteoarthropathy.

    Diabetic osteoarthropathy is the most common neuropathic condition seen clinically in the foot. It can develop rapidly with a time course of weeks to months. Soft tissue swelling and erythema with deformity are noted clinically, usually with decreased pain sensation. However, it is important to realize that these patients frequently do have pain; it may only be disproportionate to the degree of injury. Radiographic diagnosis can predate clinical suspicion and is based on recognizing the resorptive and the productive patterns of the osteoarthropathy. Often, the first clue is recognizing the radiographic features of joint instability. Remembering that overt trauma may precipitate neuropathic osteoarthropathy will also help its recognition in the follow up of fractures.
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ranking = 3341.8773777893
keywords = osteoarthropathy
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5/13. Osseous changes due to a false aneurysm of the proper digital artery: a case report.

    A 24-year-old black man had a laceration and subsequent pseudoaneurysm of the radial digital artery of the small finger. Two months after the injury, the vessel, a lacerated radial digital nerve, and a flexor digitorum profundus were all repaired. A depression was noted in the middle phalanx at surgery. Osseous reconstruction of the defect in the middle phalanx was shown on follow-up x-ray films.
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6/13. acro-osteolysis associated with hypertrophic pulmonary osteoarthropathy and pachydermoperiostosis.

    The authors report two cases of marked acro-osteolysis. One patient, a 16-year-old boy, had hypertrophic pulmonary osteoarthropathy associated with bronchiectasis, clubbing of the digits, synovitis, and osseous changes. The other patient, a 55-year-old man, had pachydermoperiostosis as well as synovial hypertrophy, clubbing of the digits, blepharitis, and a cataract. While the association of acro-osteolysis and pachydermoperiostosis has been reported before, this appears to be the first verified case of acro-osteolysis combined with hypertrophic pulmonary osteoarthropathy.
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ranking = 2864.4663238194
keywords = osteoarthropathy
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7/13. Self-limited autosomal recessive syndrome of skin ulceration, arthroosteolysis with pseudoacromegaly, keratitis, and oligodontia in a Kirghizian family.

    We report on a previously apparently undescribed Puretic syndrome-like self-limited autosomal recessive disorder with onset in infancy and running its course by 11 yr in five sibs in a Kirghizian family with recurrent skin ulceration, arthralgias, fever, fistulous osteolysis around joints, oligodontia, nail dystrophy and keratitis with visual impairment or blindess in three of the five affected sibs. Affected hands and feet acquire a pseudoacromegalic appearance; fingers become clawed; involvement of growth plates around knees may lead to asymmetrical shortness with secondary scoliosis. Slow but normal healing during and after the course of the disease leaves severe cicatrization and chronic arthroses. No other organ system is involved, intellect is normal, and health and reproductive capacity are otherwise unimpaired. We propose the name Kirghizian dermatoosteolysis for this condition.
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8/13. Occupational acro-osteolysis.

    Occupational acro-osteolysis is a serious disease definitely related to the inhalation of agents involved in the polymerization of vinyl chloride. After variable periods of latency, the more or less characteristic clinical picture appears. A Raynaud's phenomenon is usually a premonitory sign indicating that alterations in bone will probably ensue. Scleroderma-like skin lesions and systemic involvement of variable degree occur frequently. The pathophysiology of the disease is unknown, but obstructive lesions of small peripheral arteries are thought to cause osteolysis of the distal phalanges of the fingers.
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9/13. Hadju-Cheney syndrome. Report of a non-familial case.

    The case of a 24-year-old man with Hadju-Cheney syndrome is reported. No similar disease occurred in his family. Inverted and bullous tips of fingers were noted at age 9 and protruded occipital bone at age 10. He complained on left facial spasm and facial tics for 1 year. Examination revealed a man of short stature, with a brachycephalic skull and hypertelorism. Radiologically there was an extreme degree of basilar impression with the basal angle of 180 degrees, demineralized spinal vertebrae, and acro-osteolysis. Both bone and CT scans demonstrated the abnormalities of the skull clearly. The basilar artery ran almost horizontally on angiography. He gradually deteriorated neurologically with bulbar, pyramidal and cerebellar signs and symptoms. review of the literature revealed at least ten non-familial and nine familial cases of this disorder. It is considered that this syndrome is a genetically determined generalized dysplastic bone disorder.
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10/13. Acrolysis in pachydermoperiostosis. Primary or idiopathic hypertrophic osteoarthropathy.

    A 45-year-old black woman was admitted for correction of an equinus deformity of the right lower extremity that resulted from shortening of the achilles tendon. She exhibited classic findings of pachydermoperiostosis, including clubbing of the digits, periosteal new bone formation, and hypertrophy of skin and its adnexa, associated with several facial acne. In addition, she showed excessive resorption of distal phalanges of the hands and feet, a finding heretofore unreported in pachydermoperiostosis, to our knowledge. Reduced peripheral blood flow has been observed in patients with pachydermoperiostosis; the resorption of distal phalanges observed in our patient may have resulted from this mechanism.
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ranking = 1909.6442158796
keywords = osteoarthropathy
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