Development of canal cholesteatoma in a patient with prenatal isotretinoin exposure

International Journal of Pediatric Otorhinolaryngology
Kathryn M Van AbelMarci M Lesperance

Abstract

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. Review of medical, audiological, and radiological records. 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. Despite extensive precautions for its use, isotretinoin remains a...Continue Reading

References

Jun 1, 1990·The Laryngoscope·R R Cole, R A Jahrsdoerfer
Jul 1, 1987·The Laryngoscope·A F Jahn, K Ganti
Dec 22, 1999·Archives of Otolaryngology--head & Neck Surgery·K Ishijima, I Sando
Dec 14, 2007·Reproductive Toxicology·Facundo Garcia-BournissenMarco De Santis

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