当前位置： SCI文献检索 > INTERNATIONAL JOURNAL OF PEDIATRIC OTORHINOLARYNGOLOGY期刊下所有文献 > Mycophenolate mofetil as a treatment of steroid dependent Cogan's syndrome in childhood.

Mycophenolate mofetil as a treatment of steroid dependent Cogan's syndrome in childhood.

Abstract：

:Cogan's syndrome is a rare chronic inflammatory disorder which typically associates an ophthalmological and vestibulocochlear involvement, with a risk of systemic symptoms in 50-70% of cases. Autoimmune origin has been consolidated by the recent discoveries of Lunardi as a result of the dysregulation of the response of B and T lymphocytes. We report here a pediatric case of Cogan's syndrome, with long term follow-up and complete vestibulocochlear and ophthalmologic examination. During the acute phase, early steroids treatment (prednisone 1mg/kg per day) was effective on ocular lesions and hearing loss. The patient required high steroid doses to maintain remission, suggesting the necessity for steroid sparing immunosuppressive agents. We introduced mycophenolate mofetil (MMF), an immunosuppressive agent mainly used in solid organ transplantation. After steroid withdrawal the patient remained in complete remission on MMF. This is the first report of MMF treatment in Cogan's syndrome. MMF produces a targeted inhibition of the proliferation of B and T lymphocytes, particularly interesting in Cogan's syndrome. Low long term toxicity and overall good tolerance make MMF a new treatment option in steroid dependent Cogan's syndrome.

journal_name

Int J Pediatr Otorhinolaryngol

journal_title

International journal of pediatric otorhinolaryngology

authors

Hautefort C,Loundon N,Montchilova M,Marlin S,Garabedian EN,Ulinski T

doi

10.1016/j.ijporl.2009.06.025

subject

Has Abstract

pub_date

2009-10-01 00:00:00

pages

1477-9

issue

10

eissn

0165-5876

issn

1872-8464

pii

S0165-5876(09)00372-3

journal_volume

73

pub_type

杂志文章

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