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Results for “Polychondritis, Relapsing”

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PubMed1992

Relapsing polychondritis--two cases with tracheal stenosis and inner ear involvement.

Relapsing polychondritis is a rare disease which often presents firstly to ear, nose and throat (ENT) departments. Its complications, respiratory, cardiovascular, renal and neurological are life-threatening; thus it is important to recognize the disease and its complications early. Treatment for relapsing polychondritis may have serious side-effects which should be taken into account when managing these patients. We report two young patients with relapsing polychondritis and their treatments; both had severe tracheal stenosis responding in one case to pharmacological and in the other to surgical intervention.

Adolescent↗
PubMed2005

[Bilateral uveitis in relapsing polychondritis. A case report].

We report a rare ocular manifestation of relapsing polychondritis. A 28-year-old woman presented with a 5-year history of relapsing polychondritis with chondritis of the nose, ears and tracheobronchial system. The ocular symptoms were bilateral uveitis with macular involvement and papillaedema. Ocular manifestations of relapsing polychondritis occur in 60% of patients. Uveitis is a rare symptom, sometimes severe, which can lead to blindness. This case report with a literature review sheds light on the features of this disease.

Adult↗
PubMed1997

Cutaneous manifestations of relapsing polychondritis in a patient receiving goserelin for carcinoma of the prostate.

Relapsing polychondritis is a chronic rheumatologic disorder of unknown etiology. Cutaneous manifestations occur in nearly half of the patients and often precede cartilaginous involvement. We present the case of a man with a history of prostatic adenocarcinoma who underwent monthly injections of goserelin (Zoladex), an LH-RH analogue. Five months after the beginning of the treatment, he presented cutaneous manifestations, which then recurred monthly, after each goserelin injection. After goserelin interruption and replacement with another treatment (cyproterone acetate), the patient was asymptomatic for 2 months. A cutaneous relapse then occurred followed by a typical cartilaginous involvement. In our observation, goserelin seems to have triggered the cutaneous manifestations of relapsing polychondritis. An hormonal precipitating factor in relapsing polychondritis has already been suggested by reports of patients whose disease worsened under chorionic gonadotropin treatment or during pregnancy.

Adenocarcinoma↗
PubMed1992

Cerebral aneurysm in relapsing polychondritis.

A 51-year-old woman with relapsing polychondritis involving auricular and nasal cartilage, hearing loss, vasculitic skin lesions and a cerebral aneurysm of the posterior cerebral artery is described. Cerebral aneurysm is a rare manifestation of relapsing polychondritis which to our knowledge has not yet been described.

Cerebral Angiography↗
PubMed2002

Amelioration of relapsing polychondritis in a child treated with oral collagen.

Relapsing polychondritis (RP) is a disease characterized by inflammation and the destruction of cartilage. The detection of antibodies to native type II collagen (CII) in the sera of some patients with relapsing polychondritis suggests that autoimmunity to this cartilage specific protein plays a role in the pathogenesis of the disease. RP is so rare that controlled therapeutic trials have not been carried out. We describe herein a child with RP who had amelioration of symptoms and a deviation in the cellular immune response to CII after being treated with daily oral CII as a toleragen.

Administration, Oral↗
PubMed1991

Complete heart block and severe aortic incompetence in relapsing polychondritis: clinicopathologic findings.

We describe a patient with relapsing polychondritis who developed fatal cardiac involvement comprising complete heart block, acute aortic incompetence, and cardiovascular collapse. Pathologic studies showed fibrosis of the cardiac conducting system and necrotizing inflammation of the aortic valve, features not previously described in relapsing polychondritis, as well as evidence of coronary artery vasculitis.

Adult↗
PubMed1977

Respiratory obstruction due to relapsing polychondritis in a Chinese male.

A case of relapsing polychondritis in a 30-year-old Chinese male is reported. The patient showed the following features: dyspnoea; hoarseness of voice; cauliflower deformity of the right ear; upper tracheal stenosis; swelling of the lower part of the sternum; pain in the costal cartilages. Airway obstruction as a presenting feature (as in this case) is unusual. The aetiology, clinical features and management of relapsing polychondritis are briefly discussed.

Adult↗
PubMed1996

[Isolated ocular recurrence of relapsing polychondritis. Apropos of a case].

Ocular manifestations of relapsing polychondritis occur in 60% of patients, most often in association with other systemic manifestations of the disease. Episcleritis is the most common manifestation, but scleral perforation, retinal vasculitis, optic neuritis and necrotizing scleritis can lead to blindness and require the use of immunosuppressive agents. We report the case of a 72-year-old woman with diffuse bilateral scleritis as the single manifestation of relapsing polychondritis. High dose steroids were used with success.

Aged↗
PubMed2004

Relapsing polychondritis: a rare disease with varying symptoms.

PURPOSE: Relapsing polychondritis (RPC) is a rare systemic disease affecting primarily cartilaginous and proteoglycan-rich structures. It is a potentially fatal disease with unknown aetiology. There are no specific tests for RPC. The diagnosis is dependent on clinical criteria, which include chondritis of both auricles, non-erosive inflammatory polyarthritis, nasal chondritis, ocular inflammation, respiratory tract chondritis and cochlear and/or vestibular damage. Ocular symptoms will occur in approximately 60% of RPC patients. As an example, a patient with signs of RPC is described. METHODS/RESULT: A 30-year-old woman was referred to our department for evaluation of a central corneal ulcer in the left eye. She had a history of recurrent pain in both her auricles and was also found to have a nasal septum perforation. Relapsing polychondritis was suspected. CONCLUSION: Non-healing corneal ulcers should alert the ophthalmologist to look for unusual reasons for this condition. RPC is one possible cause.

Adult↗
PubMed1984

Relapsing polychondritis with segmental necrotizing glomerulonephritis.

Glomerulonephritis is a very unusual feature of relapsing polychondritis. A 16-year-old woman presented with microscopic hematuria and severe renal insufficiency during the course of relapsing polychondritis. A renal biopsy specimen disclosed focal segmental proliferative glomerulonephritis with necrotizing lesions and crescent formation. Immunofluorescent studies suggested immune complex mediated glomerular injury. The renal involvement appeared during high-dose steroid therapy. Subsequently, she was treated by adding plasmapheresis and cyclophosphamide during 6 weeks, with sustained improvement. A regimen of plasmapheresis and immunosuppression warrants consideration for use in cases of relapsing polychondritis with glomerulonephritis.

Adolescent↗
PubMed2001

A case of multisymptomatic relapsing polychondritis in a 22-year-old woman.

A case of multisymptomatic relapsing polychondritis in a 22-year-old woman. We report a case of a 22-year-old woman with relapsing polychondritis (RP)--a rare and little known systemic autoimmune disease characterised by episodic inflammation of cartilaginous structures (ear, nose, bronchi, trachea, larynx, ribs, cardiovascular system). This patient presents with a seven-year history, initiated by the saddle nose. The patient developed a multitude of symptoms: auricular chondritis, ocular symptoms, recurrent arthritis, respiratory complications (laryngotracheomalacia, bilateral vocal cord palsy), sensorineural hearing loss and enchondroma of the humeral bone. The examination of an auricle biopsy by an immunofluorescent method and a positive serum reaction from the patient to normal cartilage supported the immunological nature of relapsing polychondritis. Treatment consisted of orally administered prednisone and diaminodiphenylsulfone (Dapsone).

Adult↗
PubMed2001

Relapsing polychondritis: a report of eight cases.

OBJECTIVE: To investigate clinical manifestations of relapsing polychondritis and to clarify the significance of type II collagen antibody in the disease. METHODS: Clinical manifestations and antibody titers were examined in eight cases of relapsing polychondritis which had been treated at Otolaryngology Department, Hokkaido University during the eight years from 1991 to 1998. Anti-type II collagen antibody titer was measured by ELISA method. RESULTS: The most frequent symptom was auricular chondritis; it was seen in 88% (7/8) of the cases. Ocular symptom, nasal chondritis, arthritis, respiratory tract chondritis, and audio-vestibular symptom were also common in the cases. Compared with previous reports, no difference was recognized in the manifestation frequency. Considering none of the samples from the controls was positive for anti-type II collagen antibody, two samples from the disease group were positive. The antibody positive rate was 25% (2/8). CONCLUSION: Though the measurement of type II collagen antibody titer is not a decisive factor for detection of relapsing polychondritis, it is useful as one of the complementary factors for the diagnosis, since there is no specific test for this disease.

Adolescent↗
PubMed1995

Cardiac valve replacement in relapsing polychondritis. A review.

Cardiac valve replacement is a rare but not exceptional eventuality in patients with relapsing polychondritis. One case requiring aortic and mitral valve replacement and its follow up is described. From the review of the literature an additional twenty patients who required cardiac valve replacement are analyzed. The mean delay between the first onset of relapsing polychondritis and operation was 6.51 years and the mean age at operation was 38.8 years. There was a preponderance of male patients (73.7%). Aortic and mitral valves were replaced in 100% and 28.5% of patients, respectively. During the four first postoperative years 23.8% of them were reoperated for periprosthetic leak or aortic aneurysm, and during the same period 52.6% died of a cardiovascular cause. Immunosuppressive agents should be employed in patients with relapsing polychondritis and cardiovascular involvement because they seem to be more effective than steroids in severe forms of the disease. Therefore, we recommend close and prolonged follow up: firstly because there can be early paravalvular prosthetic leakage due to the friability of the tissue to which it has been anchored; secondly because aortic aneurysms occur frequently in relapsing polychondritis, may be multiple, may involve all parts of the aorta and result in fatal rupture even in asymptomatic patients; and thirdly because there can be a fatal outcome due to other organ involvement, like airway obstruction, acute glomerulonephritis, or systemic vasculitis. Prophylactic composite graft replacement of the ascending aorta associated with replacement of the aortic valve and re-implantation of the coronary arteries could avoid the need for reoperation in these high risk patients.

Adult↗
PubMed1988

Relapsing polychondritis mimicking rheumatoid arthritis.

A woman with relapsing polychondritis presented with progressive and deforming polyarthritis (but always negative for rheumatoid factor) 14 years before the appearance of typical clinical and histological changes of nasal and auricular cartilage destruction.

Aged↗
PubMed1998

[A refractory case of relapsing polychondritis].

We report here a case of relapsing polychondritis (RPC), who responded to salazosulfapyridine (SASP) after 1.5 years of active disease. A 15-year-old girl having chondritis of bilateral auricles, nose and respiratory tract, ocular inflammation, cochlear and vestibular dysfunction and seronegative polyarthritis was diagnosed as RPC, and oral corticosteroid was initially effective. She was admitted to our hospital with obstructive tract chondritis and was refractory to any of the treatment such as intravenous (i.v.) pulse steroid therapy, i.v. pulse cyclophosphamide (CPA), oral intermittent methotrexate (MTX) and high dose i.v. gamma globulin. Although she was partly responsive to oral cyclosporine A (CsA), but clinical symptoms did not completely subside. SASP, initiated in combination with oral corticostreroid and CsA, however, was successful not only to show steroid sparing effect but also to induce complete remission. SASP might be useful to refractory case of RPC.

Adolescent↗
PubMedDate not supplied

Joint symptoms in relapsing polychondritis.

OBJECTIVE: To study articular symptoms in relapsing polychondritis (RP) and their relationship to other clinical manifestations and prognosis. METHODS: Fourteen patients who met the diagnostic criteria proposed by Damiani and Levine for RP were studied. Clinical symptoms were recorded and laboratory and radiologic examinations were carried out. In one patient a synovial histology was obtained. RESULTS: Twelve patients had joint symptoms affecting the peripheral joints (9), the chondrocostal junctions (5), or both sites together (2). Peripheral arthritis was the most frequent finding, affecting 6 patients, while peripheral arthralgia was the only articular manifestation in 3 patients. The development of joint symptoms (arthralgia, arthritis and costochondritis) was unrelated to the appearance of chondritis at other sites and no correlation was found between articular involvement and age at onset, duration of the disease, number of flares, or severity of the disease, either in terms of the number of organs involved or fatal outcome (p > 0.05). However, when articular symptoms were analysed separately arthritis was associated with a longer duration (50 months vs 30), more affected organs (4.5 vs 3.3) and a poorer prognosis compared with patients with arthralgias alone or chondrocostal symptoms. CONCLUSION: Articular symptoms are common in RP but the presence of peripheral arthritis is associated with widespread disease and a poorer prognosis.

Adolescent↗
PubMed1997

[Relapsing polychondritis associated with myelodysplasia and acute eosinophilic leukemia].

Authors report a case in which relapsing polychondritis had been diagnosed two years before myelodysplastic syndrome developed and terminated in eosinophilic leukemia. The observation that relapsing polychondritis may precede myelodysplasia is not in concordance with some of the previous reports regarding relapsing polychondritis as a paraneoplastic phenomenon of myelodysplastic syndrome. The terminally developed eosinophilic leukemia is not supposed to be a blastic phase of the underlying myelodysplasia, much rather a second malignant process. This opinion may be confirmed by the early presence of blast cells in the myelodysplastic process without eosinophilia. It seems interesting to note that both our patient and his daughter suffered from diseases of autoimmune origin: acquired vitiligo and subacute cutan lupus erythematodes, respectively.

Aged↗