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Results for “Polychondritis, Relapsing”

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PubMed2001

Otolaryngological aspects of relapsing polychondritis: course and outcome.

Relapsing Polychondritis (RP) is a rare disorder characterised by episodic inflammation of the cartilaginous structures. Differential diagnosis of this pathology is a challenging enigma since it appears only intermittently and none of the clinical features are pathognomonic, although most of the patients are usually referred to an ENT physician initially. The natural history of the disease is unpredictable. Initiating effective treatment sooner considerably reduces the rate of mortality. Patients with RP should be seen on a regular basis even if the patient is non-symptomatic. This study reports a retrospective analysis of the otolaryngological manifestations of 7 patients with RP as well as their long-term progress and reviews the symptomatology, histopathology, immunology and management.

Adult↗

PubMed1989

[Deafness in juvenile relapsing polychondritis. A case report].

Relapsing polychondritis is an autoimmune disease that affects all body cartilages. The disease may also involve the audio-vestibular system, usually bilaterally. Most of the reported cases were adults 20 years old or more. This is a case report of a 12-year-old girl with unilateral sensorineural hearing loss, and a review of the literature.

Child↗

PubMed2026

The utility of 18F-fluorodeoxyglucose PET/computed tomography in relapsing polychondritis: a systematic review and meta-analysis.

Relapsing polychondritis is a rare chronic autoimmune inflammation of the cartilage associated with life-threatening respiratory complications. Currently, no clear role of imaging modalities such as 18F-fluorodeoxyglucose (FDG) PET/computed tomography (CT) is defined in the literature. This systematic review and meta-analysis provide current evidence on the PET-positivity rate and utility in relapsing polychondritis. Prospective or retrospective studies with more than five patients of suspected relapsing polychondritis who underwent 18F-FDG PET/CT during their management and reported a PET-positivity rate were included. Low-sample-size studies describing chondritis due to other aetiologies or utilizing PET-based radiopharmaceuticals other than FDG were excluded. A systematic search using relevant keywords was conducted across four databases (PubMed, Embase, Scopus and Web of Science) to include studies up to 25 April 2025. The Joanna Briggs Institute critical appraisal tools were used for risk-of-bias analysis. Data were analysed using the R software package (v4.3.1; 2023). Out of 962 articles, three with a total of 97 patients were included. With a pooled PET-positivity rate of 94% [95% confidence interval (CI): 73-99%, I2 = 0%, P = 0.76] and a pooled baseline SUVmax of 4.0 (95% CI: 3.5-4.6, I2 = 32%, P = 0.23), 18F-FDG PET identified asymptomatic cartilage involvement in more than 25% patients and PET parameters correlated well with inflammatory markers. It had a higher positivity rate for inaccessible sites, such as peripheral airways, and was crucial in treatment monitoring. The pooled PET-positivity rate of 18F-FDG PET in relapsing polychondritis is high but requires prospective large-sample-size studies to explore the diagnostic accuracy and prognostic implications of 18F-FDG PET in relapsing polychondritis.

Polychondritis, Relapsing↗

PubMed1989

[Chronic relapsing polychondritis. Clinical characteristics].

Some imperfectly known clinical aspects of polychondritis chronica atrophicans (relapsing polychondritis), as extracted from a series of 12 cases, are presented. The osteoarticular lesions are sometimes unusual, involving the temporomandibular or cervical articulations, and the renal lesions may be severe. Pericarditis seems to be more frequent than usually mentioned in the literature. Pulmonary complications are not always those which are classically associated with lesions of the tracheo-bronchial cartilages. Haematological manifestations are not uncommon and must be taken into account when evaluating the prognosis of the disease.

Aged↗

PubMed2004

A case of relapsing polychondritis preceded by inner ear involvement.

Relapsing polychondritis is a rare disease of unknown etiology causing systematic inflammation of the whole cartilaginous tissues. We report a patient with relapsing polychondritis preceded by inner ear involvement. A 68-year-old female visited our hospital for a chief complaint of sudden bilateral hearing loss. On pure-tone audiometry, sensorineural hearing loss, 47dB on the right and 51dB on the left, was observed. Later, bilateral auricular chondritis and uveitis developed. On biopsy of the auricular cartilage, perichondritis was observed. The patient was diagnosed with relapsing polychondritis in the early stage, 2 weeks after the onset. Oral predonine administration was initiated, and the auricular inflammation was improved within 10 days. The hearing loss disorder was gradually improved. The steroid dose was reduced, and no decrease in hearing has been observed so far.

Aged↗

PubMed1992

Early aortic valve cusp rupture in relapsing polychondritis.

Aortic regurgitation associated with relapsing polychondritis usually occurs late in the disease as a result of aortic root dilatation. A case where aortic regurgitation occurred early and was due to cusp rupture with a normal aortic root is reported. The patient required urgent aortic valve replacement within six weeks of developing a murmur despite apparent control of inflammation with immunosuppressive treatment. The possibility of cusp rupture with sudden haemodynamic deterioration should be considered in patients with relapsing polychondritis who develop aortic regurgitation.

Aortic Valve↗

PubMed1990

[Relapsing polychondritis in a patient with myelodysplastic syndrome].

Relapsing polychondritis is a rare disorder of uncertain origin characterized by recurrent inflammation of cartilage. A case of myelodysplastic syndrome (MDS) associated with relapsing polychondritis is reported. A 60-year-old man who had been diagnosed as MDS was admitted because of pain and swelling in the bilateral preauricular regions and cheek. A diagnosis of relapsing polychondritis was made by coexistence of auricular chondritis, arthropathy, ocular inflammation and audio-vestibular disturbance. He also developed ocular palsies and optic neuritis. He was treated with prednisolone, azathioprine, dapsone, and then with steroid pulse therapy. Moreover, plasmapheresis and high dose gamma-globulin therapy were undertaken. However, all these treatments were unsuccessful and he died of respiratory failure.

Azathioprine↗

PubMed2003

A case of relapsing polychondritis involving placement of an expandable metallic stent.

Relapsing polychondritis is a relatively uncommon disease characterized by recurrence of progressive inflammation of cartilaginous structures. Laryngotracheal involvement occurs in 50-70% of cases, and occasionally results in acute airway obstruction. We reported a case of relapsing polychondritis with severe tracheal stenosis that was treated by the placement of two expandable metallic stents. A 58-year-old man was hospitalized with fever and breathing difficulties. A tracheostomy was performed and a silicone T-tube was placed. A diagnosis of relapsing polychondritis was made based on the biopsy from auricular and tracheal cartilages. Two expandable metallic stents were placed in the trachea to bilateral main bronchus. This dilated the airway lumen and resulted in the dyspnea disappearing completely. However, 18 months later a sudden massive hemorrhage occurred through the tracheostoma, and he died of respiratory failure. Autopsy showed a tracheoinnominate artery fistula. We discuss the management of relapsing polychondritis with severe tracheal stenosis.

Airway Obstruction↗

PubMed1976

Treatment of relapsing polychondritis with dapsone.

Three patients with relapsing polychondritis were treated successfully with dapsone. Mounting evidence suggests an immune-related pathogenesis for relapsing polychondritis. Dapsone may be effective in certain immune-related diseases because of its inhibition of lysosomal enzymes.

Aged↗

PubMed1994

[Relapsing polychondritis. Apropos of a case].

The relapsing polychondritis is a rare autoimmune disease. The pathologist finds a fibrous degeneration of the cartilage of the body. A case relapsing polychondritis is presented and the disorders of the disease are reviewed. The larynx and trachea location is a fact of a poor prognosis.

Adult↗

PubMed1992

Progressive aortic valve inflammation occurring despite apparent remission of relapsing polychondritis.

We describe a patient with relapsing polychondritis in whom aortic valve inflammation developed 3 years after diagnosis, when the polychondritis had been in apparent remission for an extended period of time. Infection and cardiac involvement can be significant complications of relapsing polychondritis. Recommendations for monitoring and treatment of patients with this disease are discussed.

Adult↗

PubMed1985

Relapsing polychondritis with severe aortic insufficiency.

Relapsing polychondritis (RP) is a rare disease characterized by auricular, nasal and respiratory tract chondritis, ocular inflammation, inflammatory polyarthritis and cardiovascular abnormalities. Here we describe a patient with a five-year history of mild nasal and auricular chondritis, which suddenly developed into a severe aortic insufficiency with rest dyspnea. The pathogenesis and the management of this rare connective tissue disease are discussed.

Adult↗

PubMed1994

Association between relapsing polychondritis and systemic lupus erythematosus.

Relapsing polychondritis (RP) is an autoimmune disorder with chondritis as a prominent clinical feature. This disease has been found to coexist in some patients with systemic lupus erythematosus (SLE) or vice versa. We report a patient with clinical criteria fulfilling the diagnosis of RP. She had elevated levels of anti-dsDNA and decreased levels of CH50 during the 6-month observation, without clinical manifestations of SLE except for arthritis. We also reviewed all 16 cases of previously described patients with RP and SLE. From our study, we conclude that chondritis is rare in patients with SLE. If immunologic abnormalities usually found in SLE are detected in patients with RP, clinicians should observe patients for other manifestations of SLE.

Adult↗

PubMedDate not supplied

A case of relapsing polychondritis associated with hemolytic anemia.

Relapsing Polychondritis (RPC) is a rare disorder of unknown etiology which affects mucopolysaccharide-rich tissues such as cartilage. A 64-year-old man developed auricular and nasal chondritis with complaints of arthralgia of the hands and cervical pain. The auricular biopsy established the diagnosis of RPC. The hematological data revealed normocytic, slightly hypochromic anemia, a persistently elevated reticulocyte count, slightly increased bilirubin, and decreased haptoglobin. The presence of hemolytic anemia was confirmed by the shortened half-life of erythrocytes and erythroid hyperplasia of the bone marrow. This case illustrates the coexistence of RPC and hemolytic anemia which has been only rarely reported. The pathogenesis of RPC is also discussed may elucidate the pathogenesis of this disease.

Anemia, Hemolytic, Autoimmune↗

PubMedDate not supplied

[Relapsing polychondritis. Apropos of a case].

Relapsing polychondritis is not a very frequent systemic disease. Nevertheless, it has to be borne in mind since very often the first symptoms appear in the sphere of otorhinolaryngology. We are faced with the problem of differential diagnosis in a case which in the beginning seemed to be a neurological pattern. The definitive diagnosis was not established until 15 months after the appearance of the symptoms when, after several outbreaks, the clinical pattern was clearer. As we have to deal with a recurrent and systemic pathology whose diagnosis is eminently clinical, long-term pursuit and valuation are necessary to allow us to know the natural history of the disease as well as the reaction to the treatment.

Diagnosis, Differential↗

PubMed1979

Relapsing polychondritis--report of ten cases.

Relapsing polychondritis is a rare disease of unknown etiology. There are approximately 211 reported cases in the world literature. This is a report of ten cases from the Cleveland Clinic Foundation. McAdam's diagnostic criteria for R.P. were reviewed and modified. For diagnosis, all patients had to have 1. at least three or more diagnostic criteria, histologic confirmation not necessary; 2. one or more of McAdam's signs with positive histologic confirmation; or 3. chondritis in two or more separate anatomic locations with response to steroids and/or Dapsone. Chondritis of the auricles (9/10 patients) and arthropathy (8/10 patients) are the most common presenting signs. Chondritis was also seen in the nose (6/10) and the upper respiratory tract involving the larynx and trachea (4/10). Cochlear and vestibular damage and ocular inflammation were each seen in 5/10 patients. Patients were treated with steroids and/or Dapsone. Both drugs were reliable in abating episodes of activity and in decreasing recurrences. These results further support Dapsone as an alternate form of treatment for RP.

Adolescent↗

PubMed1984

[Iliac-femoral artery involvement in relapsing polychondritis].

A patient presenting a relapsing polychondritis with ruptured iliac arterial aneurysm and lower limbs arteritis is described. The analysis of the results compared with the literature's cases confirm the no response for surgical and medical treatments.

Adult↗

PubMed1976

Hypocomplementemic ear effusion in relapsing polychondritis.

In a case of relapsing polychondritis it was possible to aspirate a collection of subcutaneous fluid from the patient's involved ear. A determination of total hemolytic complement activity of this fluid was low, suggesting that activation of the complement system may have occurred in the course of the patient's disease and might be related to the pathogenesis of this disorder.

Complement System Proteins↗