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We describe a case of relapsing polychondritis associated with Reiter's Syndrome in a 46-year-old white man. Relapsing polychondritis is a rare disorder of unknown etiology associated in about 30% of cases with other rheumatic disorders such as connective tissue diseases, vasculitis and exceptionally, seronegative spondyloarthritis. We found this association only once in our review of the literature.
We describe a case of relapsing polychondritis with laryngo-tracheal involvement, occurring after ear piercing in a 39-year-old woman. Polychondritis was clearly time-related to ear piercing. This association draws attention to the risk of relapsing polychondritis during body art practices with cartilage trauma.
Central nervous system manifestations are rare in patients with relapsing polychondritis. We report a case of encephalitis associated with relapsing polychondritis. The patient presented with bilateral auricular chondritis first and then tracheal chondritis, and encephalitis developed. The symptoms and signs of encephalitis were felt mostly likely to be the result of vasculitis and improved dramatically after pulse corticosteroid therapy. We believe that early recognition of central nervous system involvement in relapsing polychondritis is extremely important. High-dose corticosteroids should be given to suspected cases as early as possible.
Generalized normolipemic plane xanthomatosis is a rare cutaneous disorder, frequently associated with reticuloendothelial diseases and some disorders with inflammation. Relapsing polychondritis is also a rare disease that shows an association with various immune-mediated diseases. We report a case of generalized normolipemic plane xanthomatosis associated with relapsing polychondritis in a 56-year-old Japanese man. We have already reported the clinical picture of relapsing polychondritis as well as an increase in urinary glycosaminoglycans excretion in this patient. During subsequent treatment with various immunosuppressive therapy, including prednisone, methotrexate, azathioprine, or aurothiomalate, multiple elevated yellowish erythematous plaques appeared on his neck approximately 32 months after the onset of relapsing polychondritis. Histologically, these eruptions consisted of perivascular neutrophilic infiltrate with nuclear dust and multiple foam cells among collagen bundles, compatible with those of generalized normolipemic plane xanthomatosis. This combination of two rare diseases has not been reported in the literature to our knowledge.
The authors reviewed the records of 11 patients with relapsing polychondritis associated with active scleritis and analyzed the immunopathologic characteristics of ocular tissue from three of these. Seven patients (63%) required cytotoxic drugs, alone or in combination with low-dose oral corticosteroids. Only one patient was treated successfully with systemic corticosteroids alone. Two patients were controlled with dapsone and one with an oral nonsteroidal anti-inflammatory drug. Dapsone, which has been reported to be effective in the treatment of relapsing polychondritis, did not control the destructive scleral inflammation in six (75%) of eight patients; two (50%) of four patients with diffuse anterior scleritis were controlled with this drug. Patients with nodular and necrotizing scleritis were controlled with azathioprine and cyclophosphamide, respectively. These data suggest that the ocular manifestations of relapsing polychondritis, especially nodular and necrotizing scleritis, are less amenable to treatment with systemic corticosteroids and/or dapsone and that more potent immunosuppressants (azathioprine and cyclophosphamide) may be required to treat these lesions successfully. Results of histologic and immunofluorescent examination of conjunctival and/or scleral biopsy specimens from three patients confirmed the vasculitic nature of the eye lesions in this disease.
We report the case of a 54-year-old woman suspected of relapsing polychondritis who underwent a nasal septum biopsy under general anaesthesia. Tracheal intubation was unremarkable, but extubation attempts failed because of upper airway inspiratory collapse induced ventilatory distress. Definitive tracheostomy placement was mandatory. Cartilage biopsy confirmed severely developed relapsing polychondritis. Even though relapsing polychondritis is rare, anaesthesiologists should be aware of this pathology and preanaesthetic evaluation may focus on particular risks associated with upper airway cartilaginous structure anatomical and histological modifications.
Auricular pain, redness, and swelling are usually the result of infectious cellulitis. However, relapsing polychondritis is another cause of this symptom complex and is the result of an autoimmune phenomenon. It presents with inflammation and destruction of both articular and nonarticular cartilage, with the external ear and joint cartilage most often involved. Although usually indolent with multiple acute exacerbations, relapsing polychondritis can be rapidly fatal if the airway or cardiovascular system is involved. We present a case of relapsing polychondritis initially mistaken for auricular cellulitis. The clinical manifestations and course of relapsing polychondritis are reviewed, along with the emergency department management.
The authors describe a case of "relapsing polychondritis" showing that the characteristic manifestation of the disease on nasal and ear cartilage took place at a later stage than in the eye vessels, the inner ear and the trunk and valves of the aorta. The authors therefore consider the term "relapsing polychondritis" to be somewhat restrictive and claim that owing to the systemic diffusion of the disease in all vessels, R.P. should be included among systemic vasculitis diseases.
A case of relapsing polychondritis with aortic aneurysm is presented and relevant literature reviewed. Only one case of relapsing polychondritis has been previously reported in Indian literature. Increased awareness of this relatively rare, multi-system disease is warranted, to facilitate the diagnosis.
OBJECTIVE: To report the atypical ocular symptoms (arterialized conjunctival vessels, exudative retinal detachment) that can be the presenting manifestations of relapsing polychondritis. DESIGN: Observational case report and literature review. METHODS: A complete ocular and systemic evaluation was performed on a patient with relapsing polychondritis and exudative retinal detachment. MAIN OUTCOME MEASURES: Retinal, choroidal, and scleral findings. RESULTS: A 73-year-old man with relapsing polychondritis presented with a unilateral large bullous exudative retinal detachment and marked choroidal and scleral thickening bilaterally. CONCLUSIONS: Ophthalmologists should consider relapsing polychondritis in the differential diagnosis of exudative retinal detachment. A combination of echographic and laboratory findings can assist in the accurate diagnosis of this rare condition.
Relapsing polychondritis is a rare autoimmune disease characterized by inflammation and degeneration of cartilaginous tissue. We describe the first reported cases of relapsing polychondritis in patients with human immunodeficiency virus (HIV) and no associated connective tissue diseases. The relationship between autoimmune and HIV diseases is complex and unclear. Treatment of HIV disease with antiretroviral therapy and subsequent immune restoration may lead to the development of autoimmune diseases in genetically susceptible individuals.
We report an unusual case of a patient with a myelodysplastic syndrome associated with life threatening relapsing polychondritis. The improvement in symptoms attributable to the relapsing polychondritis during treatment of the hematologic disorder suggests that relapsing polychondritis is a paraneoplastic syndrome associated with myelodysplastic syndromes.
Central nervous system (CNS) involvement is a rare condition in relapsing polychondritis. We report a case of meningoencephalitis that revealed relapsing polychondritis in a 70-year-old woman. A vasculitic process is presumed. Patient was treated with steroids and dapsone, and no relapse was observed after a follow-up of 2 years.
We have generated transgenic (tg) mice expressing HLA-DQ8alphabeta (DQA1*0301/DQB*0302) or HLA-DQ6alphabeta (DQA1*0103/DQB1*0601) molecules lacking endogenous murine class II expression (A beta0) to investigate the ability of these HLA class II to present type II collagen (CII) and induce collagen-induced arthritis. The DQ8alphabeta tg mice responded strongly to CII, developing severe arthritis, while DQ6alphabeta tg mice were nonresponsive to CII. The addition of the mixed haplotype DQ8alpha6beta molecule did not significantly influence CII reactivity. To examine the interaction of DQ6alphabeta and DQ8alphabeta molecules in vivo, we generated double tg DQ6alphabeta/8alphabeta (A beta0) mice expressing both the alpha- and beta-chains of DQ6 and DQ8 molecules by mating DQ6alphabeta (A beta0) and DQ8alphabeta (A beta0) tg mice. CII-immunized DQ6alphabeta/8alphabeta tg mice developed severe experimental polychondritis, exhibiting both polyarthritis and auricular chondritis. The clinical, serologic, and histologic manifestations of experimental polychondritis are similar to those symptoms in human relapsing polychondritis. The susceptibility of DQ6alphabeta/8alphabeta tg mice compared with resistance in the parental strains suggests that expression of both the DQ6alphabeta and DQ8alphabeta tgs, unique to the DQ6alphabeta8alphabeta tg strain, is important in susceptibility to experimental polychondritis. The DQ6alphabeta/8alphabeta tg mice provide a model to investigate putative autoantigens and the mechanisms of pathogenesis involved in relapsing polychondritis as well as the influence of the expression of multiple HLA class II molecules on the disease process.
A 69-year-old man with a history of relapsing polychondritis developed a widespread cutaneous eruption that was clinically and histologically typical of erythema elevatum diutinum. This eruption was accompanied by a recurrence of arthralgias. The significance of this association between relapsing polychondritis and erythema elevatum diutinum, which has not been previously reported, is discussed.
A case of relapsing polychondritis with perichondrial IgG and C3, dermoepidermal IgG, and a coexisting pseudocyst of the auricle is reported. The use of direct immunofluorescence testing in establishing a diagnosis of relapsing polychondritis and the expected findings are reviewed. In some cases direct immunofluorescence may allow a diagnosis of relapsing polychondritis before clinical criteria or a routine histologic evaluation suggests a definite diagnosis.
The authors report a case of relapsing polychondritis associated with antiphospholipid antibodies. The initial manifestations were recurrent venous thrombosis which occurred prior to the chondritis. Antiphospholipid antibodies are found in various diseases, some of which are sometimes associated to relapsing polychondritis. However, antiphospholipid-antibodies have not yet been reported in this disease.
Aseptic meningitis is a rare complication of relapsing polychondritis. We describe a 60-year-old man who developed a prolonged episode of aseptic meningitis for which no cause could be determined and, that resolved spontaneously. He then developed classic relapsing polychondritis 14 months later. He subsequently had another episode of prolonged meningitis complicated by hydrocephalus. No infectious cause for the meningitis could be determined after extensive investigation including meningeal biopsy. The patient responded to corticosteroids and antituberculous therapy.