Taking severe relapsing polychondritis by the ear

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Rheumawire

Dec 23, 2003

Taking severe relapsing polychondritis by the ear

Marseille and Paris, France - The continuing fashion for piercing,

slicing, and tattooing, collectively known as " body art, " is producing

novel clinical sequelae. In most localities there is little if any

statutory regulation of such practices, despite the documented health

risks. One is a severe, treatment-resistant, relapsing polychondritis

that follows piercing of the upper third of the ear. TNF inhibitors may

be required when other modalities fail to control the inflammation.

Dr Jacques Serratrice (Service de Médecine Interne, CHU Timone,

Marseille, France) reports a typical case of ear-piercing-related

relapsing polychondritis in the December 2003 Journal of Rheumatology

[1]. Dr Saadoun (Hôpital Cochin, Paris, France) reports that such

cases, which can be refractory to such treatments as prednisone,

azathioprine, and methotrexate, may respond to infliximab [2].

Serratrice's patient was a 39-year-old women first seen in February 2002

with inspiratory dyspnea with wheezing, hoarseness, weakness of the

voice, inflamed nose and ears, and a " cauliflower ear " due to

destruction of the cartilage of the auricle of the right ear and

collapse of the concha. " Palpation of the ribs was painful and palpation

of the thyroidal cartilage was followed by coughing, " Serratrice

reports.

Three years previously the patient, during a first pregnancy, had

undergone a jewelry-shop piercing in the upper third of the pinna with

insertion of a stainless steel ring. The hole in the cartilage remained

painful, and the next month the patient changed the steel ring for a

silver one. Four weeks later she removed that because of persistent

inflammation that did not respond to topical antiseptics. The problem

continued and was diagnosed about 6 months later as local Staphylococcus

epidermidis but did not respond to pristinamycin. Following the birth of

a normal son, the patient complained of rib pain, and the chondritis of

the right ear had expanded to involvement of the nose, ribs, and

respiratory tract. Dapsone had no effect, and prednisone was only partly

effective. Serratrice notes that the patient had had silicone breast

implants for augmentation mammaplasty 6 years before.

Extensive workup showed that the patient was positive for human

anti-type II collagen antibodies. Computerized tomography showed

chondritis of thyroid and cricoid cartilages and inflammatory

involvement of the first tracheal ring.

" Faced with this typical relapsing polychondritis, which had started

near the location of an ear piercing, we decided to initiate steroid

therapy (prednisone 1 mg/kg per day) with methotrexate (12 mg/wk). After

12 months of follow-up, with slow tapering of steroids (prednisone 0.25

mg/kg), inflammation was mildly reduced, " Serratrice reports.

Although metal ions are generally assumed to be too small to trigger an

immune response, Serratrice thinks that the steel studs used in this

type of commercial ear piercing might become immunogenic after

conjugation with protein carriers during the long period that they are

left in the newly wounded cartilage. Animal studies have shown that

implanting such metal ear studs can cause chondritis in mice, apparently

due to an autoimmune response resulting from chronic inflammation at the

insertion site. Previous research has shown that commercial studs left

in contact with body fluids can trigger inflammatory reactions,

especially studs with low corrosion resistance and high cytotoxicity

[3].

Saadoun reported 2 similar cases of relapsing polychondritis in young

women. Both were inadequately controlled despite multiple treatments

including prednisone, hydroxychloroquine, gold salts, azathioprine (2.5

mg/kg per day), D-penicillamine, and methotrexate (20 mg weekly).

In the first case, TNF inhibition was tried because of polyarthritis

relapse, system inflammation, erythrocyte sedimentation of 53 mm/h,

C-reactive protein (CRP) of 42 mg/L, failure to control the ear and nose

cartilage inflammation, and hepatic toxicity due to azathioprine and

methotrexate. The first infusion of infliximab 5 mg/kg produced clinical

improvement and dropped CRP to the normal range. Eight infusions

produced sustained clinical and biological response and permitted

reduction of prednisone from 15 mg/day to 3 mg/day.

The second case had similar recurrent, refractory relapsing

polychondritis inadequately controlled by methotrexate, dapsone,

cyclosporine, azathioprine, or cyclophosphamide in addition to

prednisone. The first infusion of infliximab 5 mg/kg produced clinical

improvement, and 7 infusions produced sustained clinical and biological

response that allowed prednisone reduction from 20 mg/day to 8 mg/day.

" Both these patients had a history of therapy with immunosuppressive

agents, none of which had been sufficient to control clinical symptoms

or systemic inflammation. In both cases, repeated relapses required

long-term use of steroids. After the first infusion of infliximab,

clinical and biological improvement was observed. Prednisone dose was

reduced in both cases. Over a 9-month period sustained response was

observed, with no adverse events, " Saadoun writes.

Janis

Sources

1. Serratrice J, Ene N, Granel B, et al. Severe relapsing polychondritis

occurring after ear piercing. J Rheumatol 2003; 30:2716-2717.

2. Saadoun D, Deslandre CJ, Allanore Y, Pham XV, Kahan A. Sustained

response to infliximab in 2 patients with refractory relapsing

polychondritis. J Rheumatol 2003 Jun; 30(6):1394-5.

3. o SO, Higa OZ, Saiki M, Correa OV, Costa I. Cytotoxicity due to

corrosion of ear piercing studs. Toxicol In Vitro 2000 Dec;

14(6):497-504.