Cases reported "Fibrosis"

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1/58. Intraosseous fibrous lesions of the jaws: a manifestation of tuberous sclerosis.

    Four patients previously diagnosed with tuberous sclerosis are reported with intraosseous fibrous lesions of the jaws. review of the literature revealed comparable pathosis occurring in extragnathic bones and several previous reports of similar lesions within the jaws. Therefore, these intraosseous fibrous proliferations are thought to represent an intraoral manifestation of tuberous sclerosis and not coincidental findings. In all 4 cases, the tumors demonstrated significant collagenization with numerous interspersed plump fibroblasts. Although histopathologically similar, the features of the lesions are not specific and also can be found in desmoplastic fibromas and simple odontogenic fibromas. The definitive diagnosis requires appropriate clinicopathologic correlation.
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2/58. Pulmonary hypertension associated with pulmonary occlusive vasculopathy after allogeneic bone marrow transplantation.

    BACKGROUND: Pulmonary vasculature abnormalities, including pulmonary veno-occlusive disease, have been demonstrated in marrow allograft recipients. However, it is often difficult to make a correct diagnosis of pulmonary lesions. methods: An open lung biopsy was performed on a patient who developed severe pulmonary hypertension after bone marrow transplantation for T-cell lymphoma. RESULTS: An open lung biopsy specimen demonstrated pulmonary arterial occlusion due to intimal fibrosis and veno-occlusion. The most striking alteration was partial to complete occlusion of the small arteries by fibrous proliferation of the intima. CONCLUSION: High-dose preparative chemotherapy and radiation before transplantation are thought to have contributed to the development of vasculopathy in this patient, because arterial occlusion by intimal fibrosis and atypical veno-occlusion are often associated with lung injury due to chemoradiation. An open lung biopsy is essential for diagnosing pulmonary vascular disease presenting signs compatible with posttransplantation pulmonary hypertension.
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3/58. "Coated aorta": a new sign of erdheim-chester disease.

    erdheim-chester disease is a rare, non-Langerhans cell form of histiocytosis characterized by osteosclerosis of the metaphyseal regions of long bones, diabetes insipidus, proptosis, and retroperitoneal fibrosis. The latter usually involves the perirenal area and leads to hydronephrosis. Periaortic fibrosis is less frequent. We describe 3 unusual cases of erdheim-chester disease with periaortic fibrosis involving the whole aorta and leading to a "coated aorta" appearance on computed tomography scans. Faced with such a singular "coated aorta," bone scintigraphy can be very helpful when searching for Erdheim-Chester disease.
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4/58. Can migraine damage the inner ear?

    BACKGROUND: Auditory and vestibular symptoms and signs are common in patients with migraine, yet little is known about the pathogenesis of these symptoms and signs. OBJECTIVE: To perform clinicopathological correlation in a patient with migraine, sudden deafness, and delayed endolymphatic hydrops. methods: A patient with long-standing migraine with aura developed sudden hearing loss in the left ear at the age of 50 years and meniere disease on the right side at age 73. At age 76, he had a flurry of sudden drop attacks typical of otolithic crisis. He died of unrelated causes at age 81. The brain and temporal bones were removed approximately 24 hours after death. The cochlea and vestibular end organs were dissected after the surrounding bone was carefully removed. RESULTS: The brain and cerebrovasculature were normal. The left cochlea showed prominent fibrosis consistent with an old infarction. The right inner ear showed hydrops, with relatively good preservation of the hair cells in the cochlea, saccular macule, and cristae of the semicircular canals. However, the utricular macule was denuded of hair cells. CONCLUSIONS: The sudden left-sided deafness likely resulted from ischemia, possibly due to migraine-associated vasospasm. Presumably, the right ear suffered only minimal damage when the patient was 50 years old, but this damage later led to the development of delayed endolymphatic hydrops on the right. Otolithic crises are thought to result from pressure changes across the utricular macule. We speculate that loss of hair cells in the utricular macule resulted from a collapse of the utricular membrane onto the macule.
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5/58. Percutaneous release of abductor pollicis brevis muscle fibrosis in a bowler--a case report.

    The authors reported a patient with abductor pollicis brevis muscle fibrosis of the right thumb, stemming from a bowling injury that had occurred 6 years previously. At that time in the acute stage, a Chinese bonesetter treated the injury using manipulation, massage and herbal drugs. Abduction contracture of the patient's right thumb developed. She began to experience chronic pain at dorsal side of her right thumb and discovered that she could not move her thumb into a retro position. When she came into our hospital, physical examination revealed an abduction contracture of patient's right thumb, the angle of separation was 60 degrees, and the angle of circumduction was fixed at 90 degrees. In addition, a fibrotic band was palpable in abductor pollicis brevis muscle. The patient responded well to percutaneous release and physical therapy. As far as we know, this is an unusual case, which has not been reported before.
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6/58. Release of extra-articular ankylosis by coronoidectomy and insertion of a free abdominal flap: case report.

    INTRODUCTION: It is generally agreed that an effective treatment for extra-articular ankylosis may be coronoidectomy and excision of scar tissue. But these conventional procedures have shown a high rate of recurrence of ankylosis due to heterotopic bone and fibrous tissue formation. OBJECTIVE AND PATIENT: We report a case in whom a coronoid osteotomy and insertion of a free abdominal flap was used to treat ankylosis of the mandible following radiotherapy for maxillary cancer. RESULTS: This procedure prevented recurrence of ankylosis by heterotopic bone and fibrous tissue formation. In addition, this flap reduced the risk of postoperative infection and promoted primary healing. CONCLUSION: The procedure, coronoidectomy and insertion of a free flap, was successful because the well-vascularized musculocutaneous flap occupied the dead space, and replaced the shortage of oral mucosa consequently inhibiting the recurrence of extra-articular ankylosis.
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7/58. Constrictive bronchiolitis obliterans. Characterisation of fibrogenesis and lysyl oxidase expression patterns.

    The process leading to irreversible fibrotic constriction of the bronchioles was studied in two cases of bronchiolitis obliterans (BO) after bone marrow transplantation. Because lysyl oxidase (LOX) is the main collagen cross-linking enzyme that might account for irreversible fibrosis, its expression was studied together with expression of extracellular matrix (ECM) proteins. Characteristic types of lesions could be distinguished on the basis of histological and immunohistological criteria. An inflammatory stage was characterised by infiltration restricted to the bronchioles by lymphocytes and dendritic cells. A fibro-inflammatory stage was characterised by the coexistence of a persistent immune cellular lesion pattern with further focal modelling of a sub-epithelial neo-synthesised connective matrix. LOX expression was observed at the tips of intra-luminal fibrotic protrusions, together with tenascin and cellular fibronectin. A fibrotic stage was characterised by dense ECM deposits spreading throughout the peri-bronchiolar connective tissue, resulting in bronchiole obliteration and final disappearance. In contrast to reversible cases of fibrosis, persistence of long-term LOX expression reflecting continuing fibrosing activity might account for the irreversible status of BO. Our two cases illustrated that, at inflammatory and fibro-inflammatory stages, BO may be stabilised by immunosuppressive treatment, while the persistence of LOX expression in the fibrotic stage might correspond to a disease that becomes irreversible and fatal.
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8/58. Vascular occlusion in the endolymphatic sac in Meniere's disease.

    In 2 patients with severe Meniere's disease (MD), there was histologic evidence of occlusion of the vein of the vestibular aqueduct (VVA). This finding coincided with total or partial occlusion of numerous small vessels around the endolymphatic sac (ES), flattening of epithelium, extensive perisaccular fibrosis, and signs of new bone formation. Ultrastructural analysis of the occluding material showed foci with dense connective tissue, calcification, lipid deposits, and layers of basement membrane, sometimes concentrically arranged. The exact nature of the occluding material was unknown. In another 2 MD patients, the VVA was not visualized, and the ES vessels showed no signs of occlusion. Seven controls with acoustic schwannoma or meningioma had normal vasculature. The presence of vascular impairment in the ES in MD patients indicated that altered hemodynamics may contribute to the pathogenesis of endolymphatic hydrops and MD.
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9/58. Two-stage exchange of infected knee arthroplasty with an prosthesis-like interim cement spacer.

    Two-stage revision in infected knee arthroplasty is standard practice. One problem during the interim period is soft tissue fibrosis. Attempts have been made to preserve leg length and ligament length by introducing spacers, usually made out of antibiotic-loaded bone cement. We present a new interim prosthesis, which is made intra-operatively out of polymethylmethacrylate (PMMA). Antibiotic-loaded cement provides a therapeutic level of antibiotics in the periarticular soft tissue. We report the results in ten patients, who were treated with this prosthesis-like spacer and were prospectively studied. After an average follow-up of 13.5 months, there was no recurrent infection.
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10/58. Exostosis following a free gingival graft.

    BACKGROUND: There have been few cases reported of exostoses following a free gingival graft. In 1980, a free gingival graft was placed on the facial level of 33-34, developing over the years a significant enlargement. In 1999, since the patient felt progressively uncomfortable with the enlarged area, its surgical reduction was proposed. METHOD: Under local anesthesia, the hard tissue developed under the previously-grafted area, was significantly reduced. The specimen, together with a fragment of the covering soft tissue, was sent for histological analysis. RESULTS: The surgical wound healed uneventfully, and the patient was satisfied with the results. The histology showed the presence of mature bone surrounded by a dense connective tissue, whereas the gingival tissue showed acanthosis and fibrosis. CONCLUSION: The development of exostoses following a free gingival graft can be considered an unpredictable, albeit infrequent side-effect of this procedure. The fact that most of these exostoses appear in the cuspid-premolar area, deserves further consideration.
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