Literature DB >> 16116518

Two cases of lacrimal gland agenesis in the same family--clinicoradiologic findings and management.

Seung Hyun Kim1, Sangjoon Hwang, Soonyoung Kweon, Tack-Koon Kim, Jaeryung Oh.   

Abstract

BACKGROUND: Lacrimal gland agenesis is extremely rare. This condition is occasionally associated with salivary gland agenesis and abnormalities in the lacrimal drainage system, particularly occlusions of the lacrimal puncta and canaliculi. The detailed presentation of clinicoradiologic findings and treatment modality has not been previously reported.
METHODS: A 6-year-old boy was presented to our department complaining of severe foreign-body sensations in both eyes. Punctate epithelial erosions and mucous filaments were noted in both eyes. His tear break-up time (TBUT) was less than 1 second, and a Schirmer's test (both eyes) detected less than 1 mm of wetting in 5 minutes. His mother had similar complaints dating back to childhood.
RESULTS: Orbital computed tomography (CT) scan and magnetic resonance imaging (MRI) revealed the absence of both lacrimal glands and all major salivary glands. After the insertion of a lacrimal punctal plug, punctate epithelial erosions and mucous filaments decreased remarkably within 2 weeks. MRI scans of the patient's mother confirmed the absence of both lacrimal glands and salivary glands. She had been using artificial tears for a long time.
INTERPRETATION: The absence of lacrimal glands can be confirmed by clinicoradiologic findings, and the punctal plug may be an effective tool for the treatment of patients who retain a patent lacrimal drainage system.

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Year:  2005        PMID: 16116518     DOI: 10.1016/S0008-4182(05)80014-6

Source DB:  PubMed          Journal:  Can J Ophthalmol        ISSN: 0008-4182            Impact factor:   1.882


  4 in total

1.  Isolated form of congenital bilateral lacrimal gland agenesis.

Authors:  Nedime Sahinoglu; Samuray Tuncer; Nilufer Alparslan; Gonul Peksayar
Journal:  Indian J Ophthalmol       Date:  2011 Nov-Dec       Impact factor: 1.848

2.  Identification of AAAS gene mutation in Allgrove syndrome: A report of three cases.

Authors:  Wenjing Li; Chunxiu Gong; Zhan Qi; D I Wu; Bingyan Cao
Journal:  Exp Ther Med       Date:  2015-08-10       Impact factor: 2.447

3.  Isolated bilateral congenital lacrimal gland agenesis - Report of two cases.

Authors:  Manar Alwohaib; Silvana Artioli Schellini; Sahar M Elkhamary; Osama Al Shaikh
Journal:  Saudi J Ophthalmol       Date:  2017-05-03

4.  Waardenburg syndrome with dry eyes: A rare association.

Authors:  Anupam Singh; Sanjeev Kumar Mittal; Ajai Agrawal; Rupal Verma; Preeti Yadav
Journal:  Taiwan J Ophthalmol       Date:  2019-09-12
  4 in total

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