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Recurrent auricular chondritis and cartilage repair
  1. A P Rozin1,
  2. E Gez2,
  3. R Bergman3
  1. 1B Shine Department of Rheumatology, Rambam Medical Centre and B Rappaport Faculty of Medicine, Israel-Technion Institute of Technology, Haifa, Israel
  2. 2Department of Oncology, Rambam Medical Centre and B Rappaport Faculty of Medicine, Israel-Technion Institute of Technology, Haifa, Israel
  3. 3Department of Dermatology, Rambam Medical Centre and B Rappaport Faculty of Medicine, Israel-Technion Institute of Technology, Haifa, Israel
  1. Correspondence to:
    Dr A P Rozin
    a_rozinrambam.health.gov.il

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We present a case of recurrent auricular chondritis, which developed after two injections of a luteinising hormone-releasing hormone (LH-RH) analogue buserelin combined with oral treatment of a pure antiandrogen bicalutamide (Casodex). The patient was treated successfully with a continuous moderate dose of corticosteroids together with azathioprine and methotrexate. Complete repair of the deformed ear followed 7 months after starting the treatment.

Relapsing polychondritis (RP) is a chronic autoimmune cartilaginous inflammation. Auricular chondritis is a presenting sign in over 85% of patients, in which patients’ ears become red, swollen, and tender. We observed a painless form of recurrent auricular chondritis complicated by severe cartilage damage.

CASE REPORT

A 69 year old man had a history of old myocardial infarction and prostatic adenocarcinoma (Gleason score VI, stage T2B NO MO). He underwent two injections of an LH-RH analogue buserelin (Suprefact Depot, Aventis Pharma), combined with a pure antiandrogen bicalutamide (Casodex; AstraZeneca).

Five months after starting treatment he presented with a painless redness and swelling of his right ear, which spared the lobe. This inflammation, showing inflammatory mononuclear infiltrate, progressed to include dropping and deformity of the upper part of the ear, and resolved spontaneously after 3 months (figs 1A and 2). The patient was then referred to the rheumatologist.

Figure 1

 (A) Droop of the pinna after a prolonged (3 months) painless episode of auricular chondritis. The biopsy point is seen on the upper posterior portion of the auricular cartilage. (B) Complete repair of the normal auricular form and cartilage followed 7 months of treatment.

Figure 2

 An inflammatory cell infiltrate obscures the chondrodermal interface and partially replaces the auricular cartilage: the findings are associated with RP (haematoxylin-eosin, ×400).

Besides the dropped pinna, no systemic manifestations were found on physical examination and the patient’s neurological status was unremarkable, including normal pain perception. Retinal screening showed no vasculitic changes. Routine laboratory investigation was normal except for raised inflammatory markers: erythrocyte sedimentation rate and C reactive protein. Serum immunoelectrophoresis, antinuclear antibodies, antineutrophil cytoplasmic antibodies, rheumatoid factor, prostate-specific antigen were normal. Viral serology, tuberculin test, and Venereal Disease research Laboratory test were negative. Ultrasound of large vessels and echocardiography were normal. A chest x ray examination and computed tomography (CT), abdominal CT, and bone scan were unremarkable.

Owing to serious coronary disease, the lack of active auricular inflammation and systemic disease, and because buserelin treatment had been stopped, it was decided to observe the patient without treatment. However, painless auricular chondritis of the opposite side occurred 3 months later, supporting an initial suspicion of relapsing polychondritis. Prednisone 40 mg/day and azathioprine 100 mg/day were given. Corticosteroids were tapered to 20 mg/day for 1 month after reduction of inflammation in the opposite ear. Azathioprine treatment was stopped when the liver enzyme level was significantly raised after 2 months of treatment. A daily dose of prednisone 20 mg/day was continued, and oral methotrexate 7.5 mg/week was started after the liver function returned to normal. Partial repair of the auricular cartilage was noted 2 months later and full repair of the deformed ear was seen 7 months after the start of corticosteroids and second line treatment (fig 1B). No auricular deformity of the second ear developed.

DISCUSSION

A case of RP which followed 5 months of antiandrogen treatment with another LH-RH analogue, goserelin (Zoladex) was reported a few years ago.1 A hormonal precipitating factor in RP has been suggested by reports of patients whose disease worsened during pregnancy or during chorionic gonadotropin treatment.2,3 However, RP affects men and women equally.

It is still unclear, what component of antiandrogen treatment might be responsible for the auricular chondritis in our case, but the precedent with other LH-RH analogues points to buserelin as possible offender. Is there really a relation with prostate cancer treatment, when only a few cases are reported among the millions who receive this type of treatment? The question should be further investigated.

We think, that deep suppression of the autoimmune process with a moderate dose of prednisone (20 mg/day) and second line treatment may be the background for effective cartilage repair.4 Corticosteroids, carrying androgen properties, might also promote cartilage restoration.5,6 It has been shown that corticosteroids might stimulate glycosaminoglycan and DNA synthesis in chondrocytes.7,8 It is well known that articular cartilage in adults has a limited ability for self repair. However, if the damage extends beyond the subchondral bone, a repair process ensues in which mesenchymal progenitor cells migrate into the injured site and undergo chondrogenic differentiation.9 Highly vascularised auricular skin and perichondral tissue may provide the cartilage with such progenitor cells and growth factors.10 Painless inflammation of cartilage tissue in the patient with normal pain perception may indicate early destruction of receptors. If such occult damage involves cartilage tissue of large vessels, the consequences may be dramatic. This concealed process should be considered in planning follow up and prophylaxis of a flare.

REFERENCES

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