Document Detail


Development of canal cholesteatoma in a patient with prenatal isotretinoin exposure.
MedLine Citation:
PMID:  20638138     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
PURPOSE: To describe the clinical and radiologic findings in a case of isotretinoin embryopathy-like syndrome and discuss management of hearing loss, congenital external auditory canal (EAC) stenosis, and EAC cholesteatoma.
METHODS: Review of medical, audiological, and radiological records.
RESULTS: An 8 year old female presented with bilateral moderate conductive hearing loss, bilateral microtia, left EAC stenosis, and right EAC atresia, secondary to prenatal isotretinoin exposure. Comorbidities included developmental delay, ventricular septal defect, hypotonia, and retinal maldevelopment. The left EAC was sharply upsloping with a 2mm-diameter meatus. Computed tomography (CT) scan of the temporal bone demonstrated normal middle and inner ears bilaterally; serial CT scans over 6 years demonstrated progressive development of left canal cholesteatoma. Implantation of a right BAHA system was performed, followed by left canalplasty and excision of cholesteatoma with facial nerve monitoring. An endaural incision was utilized to avoid compromising future microtia repair. Postoperative left-sided hearing improved to mild low-frequency conductive hearing loss rising to normal at 2000 Hz and above.
CONCLUSIONS: Despite extensive precautions for its use, isotretinoin remains a cause of major birth defects, including sensorineural, conductive or mixed hearing loss. Congenital EAC stenosis is much less common than congenital atresia or acquired stenosis; optimal surgical approaches vary depending on hearing status and facial nerve anatomy. Close monitoring for development of canal cholesteatoma is necessary.
Authors:
Kathryn M Van Abel; Marc E Nelson; Ryan M Collar; Marci M Lesperance
Related Documents :
15294328 - The cisternal segment of the abducens nerve in man: three-dimensional mr imaging.
15956488 - Optic neuropathy secondary to cat scratch disease: distinguishing mr imaging features f...
1443398 - Myelination of the optic radiation in leber congenital amaurosis.
22826138 - The pattern and pace of hyperacute hemorrhage expansion.
10552728 - Relating x-ray absorption and some quality characteristics of mango fruit (mangifera in...
22918778 - Intraoperative somatostatin receptor detection after peptide receptor radionuclide ther...
Publication Detail:
Type:  Case Reports; Journal Article     Date:  2010-07-17
Journal Detail:
Title:  International journal of pediatric otorhinolaryngology     Volume:  74     ISSN:  1872-8464     ISO Abbreviation:  Int. J. Pediatr. Otorhinolaryngol.     Publication Date:  2010 Sep 
Date Detail:
Created Date:  2010-08-16     Completed Date:  2010-12-22     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8003603     Medline TA:  Int J Pediatr Otorhinolaryngol     Country:  Ireland    
Other Details:
Languages:  eng     Pagination:  1082-4     Citation Subset:  IM    
Copyright Information:
Copyright 2010 Elsevier Ireland Ltd. All rights reserved.
Affiliation:
Thomas Jefferson School of Medicine, USA.
Export Citation:
APA/MLA Format     Download EndNote     Download BibTex
MeSH Terms
Descriptor/Qualifier:
Abnormalities, Drug-Induced / etiology
Child
Cholesteatoma / chemically induced*,  radiography,  surgery
Ear / abnormalities*
Ear Canal* / radiography
Ear Diseases / chemically induced*,  radiography,  surgery
Female
Hearing Loss, Conductive
Humans
Isotretinoin / adverse effects*
Pregnancy
Prenatal Exposure Delayed Effects*
Teratogens*
Chemical
Reg. No./Substance:
0/Teratogens; 4759-48-2/Isotretinoin

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


Previous Document:  CCR7 is expressed in astrocytes and upregulated after an inflammatory injury.
Next Document:  The effect of lip strengthening exercises in children and adolescents with myotonic dystrophy type 1...