Document Detail


Congenital trigeminal anesthesia in two siblings and their long-term follow-up.
MedLine Citation:
PMID:  10653422     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
PURPOSE: To alert ophthalmologists to congenital trigeminal anesthesia as a cause of corneal scarring and amblyopia and its effective treatment with tarsorrhaphies. METHODS: Case reports. A 2-month-old infant presented with bilateral corneal erosions and complete corneal anesthesia. Her sister presented at age 3 years with a corneal ulcer and corneal hypoesthesia (sensation markedly decreased). The father and paternal grandmother of the siblings also had corneal hypoesthesia. RESULTS: Further investigation of the infant revealed bilateral hearing loss, swallowing difficulties, and decreased sensation in the trigeminal nerve distribution. A diagnosis of congenital trigeminal anesthesia was made. The corneal erosions of the patient resolved with bilateral two-thirds width tarsorrhaphies. The girl continues to do well now at 10 years of age with ocular lubrication and superficial corneal scar removal. Her older sister initially required antibiotic ointment for her corneal ulcer but now requires only ocular lubrication for congenital trigeminal anesthesia. CONCLUSION: This study describes the earliest reported use of tarsorrhaphies in an infant with congenital trigeminal anesthesia. The presence of this condition in her sister and relatives makes it one of the few reports of congenital trigeminal anesthesia in more than two generations. Early recognition of this condition is essential in the preservation of useful vision.
Authors:
V A Wong; R A Cline; P J Dubord; M Rees
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of ophthalmology     Volume:  129     ISSN:  0002-9394     ISO Abbreviation:  Am. J. Ophthalmol.     Publication Date:  2000 Jan 
Date Detail:
Created Date:  2000-02-03     Completed Date:  2000-02-03     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  0370500     Medline TA:  Am J Ophthalmol     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  96-8     Citation Subset:  AIM; IM    
Affiliation:
Department of Ophthalmology, Vancouver Hospital and Health Sciences Centre, BC, Canada. anthonyw@unixg.ubc.ca
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MeSH Terms
Descriptor/Qualifier:
Amblyopia / congenital*,  diagnosis,  surgery
Child, Preschool
Cornea / innervation*
Corneal Diseases / congenital*,  diagnosis,  surgery
Deglutition Disorders / congenital
Eyelids / surgery
Female
Follow-Up Studies
Hearing Loss, Bilateral / congenital
Humans
Hypesthesia / congenital*,  diagnosis,  surgery
Infant
Nuclear Family
Trigeminal Nerve / abnormalities*,  pathology
Trigeminal Nerve Diseases / congenital*,  diagnosis

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


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