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Methotrexate as a steroid-sparing agent in coganaposs syndromecomment on the concise communication by richardson.

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Methotrexate as a steroid-sparing agent in Cogan’s
syndrome: comment on the concise communication by
To the Editor:
We read with interest the article on methotrexate
therapy for Cogan’s syndrome by Richardson (1). We would
like to report a similar experience in a patient with Cogan’s
syndrome, in which corticosteroids were poorly tolerated
and could not be tapered successfully.
The patient, a 33-year-old white woman, was first
seen in June 1990 when she presented with eye pain and
sudden decreased hearing acuity in the left ear associated
with tinnitus, nausea, vomiting, and vertigo. Three years
previously she had developed hearing loss in the right ear
and bilateral eye irritation, which were not treated. Since
that time, the eye manifestations had resolved, but almost
complete hearing loss in the right ear had persisted. The
patient’s medical history was otherwise unremarkable.
An audiologic evaluation confirmed the sensorineuronal hearing loss bilaterally. Audiometry demonstrated
almost complete hearing loss in the right ear and a loss of
25-60 decibels in the left ear, prevalent on high frequency. A
computed tomography scan of the head and evoked potentials ruled out retrocochlear abnormalities. Results of a
middle-ear evaluation with tympanometry and acoustic reflex measurement were normal. Electronystagmography disclosed minimal vestibular dysfunction. An eye examination
revealed interstitial keratitis bilaterally. There was no serologic evidence of syphilis, and results of other investigations,
including tests for antinuclear antibodies and rheumatoid
factor, a spinal fluid examination, and an echocardiography ,
were negative or normal.
A diagnosis of Cogan’s syndrome was considered,
and prednisone treatment at 1 mgkglday was instituted. The
interstitial keratitis healed gradually, and the patient noted
hearing improvement in the left ear, which was confirmed
with audiometry as a gain of 30 decibels. Minimal improvement was also noted in the right ear.
During the following 2 years, several attempts to
decrease the corticosteroid dosage were associated with
relapse of vertigo and fluctuating bilateral hearing loss, and
required massive increases in the prednisone dosage, as
much as 2 mgkg/day. The minimum dose of prednisone that
was needed to avoid relapses in this patient was always
above 20 mg/day. Therapy was poorly tolerated and the
patient developed cushingoid features and high blood pressure. In August 1992, low-dose methotrexate therapy (15
mg/week) was added to the regimen, which allowed for
tapering of the corticosteroid dosage without further decreases in hearing loss or recurrence of the vertigo or
interstitial keratitis. By November 1994, the patient was
receiving 9 mg of prednisone daily. Notably, results of a test
for anticochlear antibodies (a recently developed test) were
positive while the patient was receiving methotrexate (2).
Although spontaneous remission may occur in
Cogan’s syndrome, it is unlikely that this was the case in our
patient, since repeated attempts to decrease the corticosteroid dosage led to recurrence of hearing loss and vertigo.
Thus, we can reasonably assume that methotrexate was a
useful steroid-sparing agent in this patient.
The pathophysiology of Cogan’s syndrome remains
unknown, but in some cases, evidence of systemic necrotizing vasculitis has been reported. Therefore, as in rheumatoid
arthritis, in which methotrexate therapy has been associated
with accelerated nodulosis and cutaneous vasculitis (3), such
therapy should also be carefully monitored in Cogan’s syndrome. It is unlikely that a randomized controlled study of
this rare systemic disease could be done, and additional
individual experiences should be reported before methotrexate can be recommended widely.
Jacques Pouchot, MD
Philippe Vinceneux, MD
Hbpital Louis Mourier
University Paris VII
Colombes, France
Didier Bouccara, MD
Olivier Sterkers, MD, PhD
Hbpital Beaujon
University Paris VII Clichy, France
Bernard Bodelet, MD
Hbpital de St Dit des Vosges
St Die des Vosges, France
1. Richardson B: Methotrexate therapy for hearing loss in Cogan’s
syndrome. Arthritis Rheum 37:1559-1561, 1994
2. Mayot D, Btn6 MC, Pemn C, Faure GC: Immunologic alterations in patients with sensonneural hearing disorders. Clin
Immunol Immunopathol68:4145, 1993
3. Segal R, Caspi D, Tishler M, Fishel B, Yaron M: Accelerated
nodulosis and vasculitis during methotrexate therapy for rheumatoid arthritis. Arthritis Rheum 31:1182-1185, 1988
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