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Results for “Polychondritis, Relapsing”

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PubMed1978

Relapsing polychondritis with crescentic glomerulonephritis.

Relapsing polychondritis is rare and its cause is unknown. The tissues affected are those with a high glycosaminoglycan content, such as cartilage, the aorta, the sclera and cornea, and parts of the ear. Symptoms can usually be controlled with oral steroids, but when there is coexistent progressive crescentic glomerulonephritis quadruple chemotherapy may be used. Three cases of the clinical syndrome of relapsing polychondritis were studied in which rapidly progressive cresentic glomerulonephritis developed. In two the patients appeared to respond to aggressive treatment with immunosuppressive agents and anticoagulants. The multisystemic nature of the disease, the renal lesions, and the response to treatment all suggested that the condition might be related to periarteritis nodosa.

Adult↗
PubMed1994

Ocular manifestations of relapsing polychondritis. Three case histories.

Relapsing polychondritis is a rare, presumably inflammatory autoimmune disorder affecting cartilagenous structures throughout the body. The ears, nose, joints, eyes and the respiratory tract are most frequently involved. The main ocular manifestations are episcleritis and scleritis, conjunctivitis, iridocyclitis and chorioretinitis, cataract and corneal infiltrates and melting. Extraocular signs and symptoms often are indicative of the diagnosis. Therapy includes systemic steroids, immunosuppressive drugs and dapsone. Three case histories are reported and the literature of ocular manifestations of relapsing polychondritis is reviewed.

Aged↗
PubMed1976

Relapsing polychondritis treated with dapsone.

Relapsing polychondritis is a rare and sometimes fatal disease of unknown cause and pathogenesis. Results of recent studies suggest that it is an immune disorder probably caused by the production of autoantibodies to possibly the mucopolysaccharides. Perpetuation of the cartilage inflammation may be maintained by a proteolytic process. Treatment primarily has been with orally given corticosteroids. Dapsone has been shown to possibly inhibit lysosomal enzyme activity. We present a patient who, to our knowledge, is the first with relapsing polychondritis to be treated successfully with dapsone. The rapid response of the patient's condition to treatment with dapsone suggests an alternative, if not preferred, method of controlling this disease.

Dapsone↗
PubMed1992

Relapsing polychondritis: new pulmonary manifestations.

Relapsing polychondritis is purported to be an autoimmune disease characterized by inflammation of cartilaginous structures including the nose, ears, glottis, trachea, and mainstem bronchi. Arthropathy, aortopathy, scleritis, conjunctivitis, iritis, vertigo, otitis media, glomerulonephritis, and skin lesions are common manifestations. We have recently cared for an elderly nonsmoker with a history of abdominal aortic aneurysm repair and recurrent bouts of painful ear swelling who presented with a pulmonary infiltrate and pulmonary function studies suggestive of small airways disease. Subsequent transbronchial biopsy revealed no pathogens but was associated with excessive bleeding, perhaps suggestive of a vasculitic process in the lung. The pulmonary infiltrate cleared over a 2-month interval with the use of corticosteroids. Unusual in this otherwise classical case of relapsing polychondritis was that the pulmonary infiltrate and small airways disease were the prominent pulmonary manifestations and no tracheobronchial abnormalities were visualized.

Adrenal Cortex Hormones↗
PubMed1989

Relapsing polychondritis in an adolescent.

Relapsing polychondritis (RP) is an uncommon systemic disorder with a highly variable course. A 17-year-old woman recently presented with a 1-month history of depression, weight loss, chest wall tenderness, hoarseness, and dysphagia. Physical examination revealed cachexia, low-grade fever, pharyngeal erythema, and tenderness of the right auricle, anterior chest, cricothyroid cartilage, and both knees. Laboratory studies included a hematocrit of 34% and a sedimentation rate of 50 mm/hr. Initial improvement on oral corticosteroids was followed by respiratory distress. At that time calcified tracheal cartilage, subglottic stricture, and a saddle nose deformity were present. Despite therapy with steroids, dapsone, and pulse cyclophosphamide, the respiratory distress reoccurred, eventually necessitating tracheostomy. Tracheal cartilage biopsy confirmed the presumptive diagnosis of RP. Bilateral auricular chondritis developed after initial presentation, as did acute vertigo. Although seen in all age groups, less than 10% of cases of RP are seen in children and adolescents. Auricular chondritis (89% of all cases), inflammatory asymetric arthritis (81% of all cases), nasal chondritis (72% of all cases), respiratory tract chondritis (56% of all cases), and audiovestibular abnormalities (46% of all cases) were present in our patient. Relapsing polychondritis may follow a slowly evolving or rapidly progressive course. Appropriate diagnosis and aggressive therapy are recommended to lessen the morbidity and mortality.

Adolescent↗
PubMed1988

[Relapsing polychondritis: a diversified disease picture].

Relapsing polychondritis is a rare, at times fatal systemic disease involving inflammatory destruction mainly of cartilaginous structures. The sensory organs, respiratory tract, cardiovascular system and kidneys are most frequently involved. Based on the current state of knowledge it seems probable that relapsing polychondritis is an autoimmune disease. Early involvement of the respiratory tract and kidneys, anemia, arthritis and saddle-nose deformity are associated with poor prognosis. 2 cases of relapsing polychondritis (one with erosive symmetrical polyarthritis and the second with massive involvement of the respiratory tract) are presented.

Adult↗
PubMed1987

Relapsing polychondritis syndrome associated with pustular psoriasis, spondylitis and arthritis mutilans.

We describe the first case of pustular psoriasis, HLA-B27 positive spondylitis, arthritis mutilans, and relapsing polychondritis. The relapsing polychondritis was mild and long undetected. Relapsing polychondritis is seen not uncommonly with a variety of inflammatory skin lesions. Because of this, and because many rheumatic diseases have been observed with relapsing polychondritis, we stress that relapsing polychondritis is a syndrome and not a unique disease. Relapsing polychondritis in mild form may be fairly common; a search for signs and symptoms of chondritis should be a routine part of the workup of patients with inflammatory rheumatic diseases and dermatoses.

Arthritis↗
PubMed1993

Relapsing polychondritis presenting as cutaneous polyarteritis nodosa.

Relapsing polychondritis is an infrequently diagnosed, though not necessarily uncommon, systemic disorder characterized by recurrent and potentially destructive inflammation of cartilaginous structures, the eye, and the audiovestibular and cardiovascular systems. Although dermal involvement occurs in approximately 25% of patients with relapsing polychondritis, in only few cases has a skin biopsy been obtained revealing lesions such as leukocytoclastic vasculitis, livedo reticularis, erythema nodosum or keratodermia blenorrhagicum. We describe a patient with relapsing polychondritis in whom a cutaneous polyarteritis nodosa preceded cartilage inflammation by 6 months. Cutaneous polyarteritis nodosa is a rare form of vasculitis that appears to be limited primarily to the skin, muscles, and joints. In contrast to the systemic form of the disease it is characterized by the absence of visceral lesions and a relapsing but benign course. The present case and the fact that vasculitis is a concomitant feature in approximately 30% of patients with relapsing polychondritis [21] demonstrates that this condition may not represent a distinct clinical entity.

Aged↗
PubMed1981

Aortic cusp involvement causing severe aortic regurgitation in a case of relapsing polychondritis.

A case with relapsing polychondritis is described where primary involvement of the aortic valve cusps produced severe aortic regurgitation requiring valve replacement. An aneurysmal dilatation of the ascending aorta developing later led to disruption of the prosthesis requiring re-operation. Superior vena caval obstruction, an abdominal aortic aneurysm which ruptured and required resection, and obstructive lesions in common iliac arteries, presumably the result of the same process that involved the aorta and the cartilaginous structures, were also seen.

Adult↗
PubMed1981

Immune complex-mediated renal involvement in relapsing polychondritis.

A patient with relapsing polychondritis who presented with rapidly progressive glomerulonephritis is described. This is the fourth case in which this association is reported. Crescentic glomerulonephritis was found on renal biopsy. Evidence for immunologic participation in the pathogenesis of this condition is suggested by the demonstration of circulating immune complexes, immunoglobulin and complement deposition by immunofluorescence, and electron dense deposits by electron microscopy of the kidney. Favorable response followed therapy with prednisone and dapsone.

Aged↗
PubMed1989

The radiological manifestations of relapsing polychondritis.

Four patients with relapsing polychondritis are described. The radiological appearances of the arthropathy and the tracheobronchial involvement are illustrated, and the value of these findings in early diagnosis and management are emphasised.

Bronchography↗
PubMedDate not supplied

CT manifestations of tracheobronchial involvement in relapsing polychondritis.

Two patients with relapsing polychondritis presented with tracheobronchial stenosis without clinical manifestation of nasal septum or ear lobe cartilage involvement. The CT findings included diffuse, smooth tracheobronchial wall thickening with narrowing and deformity of the lumen. Areas of dense calcium deposition within the thickened tracheal cartilage were seen. On follow-up after steroid medication, tracheobronchial wall thickening was decreased and the configuration of the tracheal lumen returned to normal.

Bronchi↗
PubMed1999

Endobronchial stenting for severe airway obstruction in relapsing polychondritis.

Airway complications of relapsing polychondritis (RP), including tracheobronchial stenosis, can be fatal. This paper describes a life-saving technique (placement of multiple metallic endobronchial stents under conscious sedation) to prevent life-threatening airway closure in a 50-year-old woman with RP. Using fluoroscopic and bronchoscopic guidance, a tracheal stent and three endobronchial metallic stents were deployed in the central airways, with good functional outcome. There were no complications. In critical airway compromise caused by RP, the insertion of endobronchial stents can result in improved symptoms, pulmonary function, and a return to daily activities, without the use of tracheotomy and mechanical ventilation.

Airway Obstruction↗
PubMed2003

Endobronchial ultrasonography in the diagnosis and treatment of relapsing polychondritis with tracheobronchial malacia.

Relapsing polychondritis (RP) with tracheobronchial involvement has a poor prognosis, and a delay in diagnosis increases morbidity and mortality; however, the diagnosis is difficult to make. Endobronchial ultrasonography (EBUS) revealed changes in the tracheobronchial cartilage in two patients who met the criteria for RP, and facilitated the diagnosis. In these cases, EBUS revealed a poorly defined bronchial wall structure with two patterns of cartilaginous damage: fragmentation and edema. These cases were successfully treated by the implantation of nitinol stents, the sizes of which were determined by EBUS. EBUS was found to be useful in the diagnosis and treatment of RP.

Aged↗
PubMed1984

Cyclosporin A treatment in a case of relapsing polychondritis.

A patient with relapsing polychondritis and high titres of anti-collagen II antibodies developed a progressive destruction of tracheal cartilage in spite of treatment with corticosteroids, Dapsone, Azathioprine and cyclophosphamide. Assumptions regarding the role of anti-collagen immunity, together with immunohistochemical studies on affected cartilage, suggested macrophage-T cell interactions to be important in the pathogenesis of the disease. Cyclosporin A treatment was instituted and has been continued for 2 years, concomitant with a steady improvement and remission of the disease.

Autoantibodies↗
PubMed2001

Ear, nose and throat manifestations of relapsing polychondritis in a child.

Relapsing polychondritis is a rare disorder, that can present initially to Otolaryngologists and can easily be overlooked due to its relative rarity and atypical initial symptoms and signs. Here we report on a 12-year-old schoolboy who presented initially with ear, nose and throat manifestations, including stridor, cough, hoarseness, sore throat and fever. The other clinical signs such as nasal tip, depression and softening of right auricle, developed subsequently. The clinical features, laboratory investigations, diagnostic criteria and treatment options were discussed with a brief review of literature.

Antirheumatic Agents↗