Literature DB >> 28539756

Neurotrophic keratitis in a patient with disseminated lymphangiomatosis.

Jared E Knickelbein1,2, Susan T Stefko1, Puwat Charukamnoetkanok1.   

Abstract

INTRODUCTION: Neurotrophic keratitis, a degenerative corneal disease caused by trigeminal nerve impairment, has many etiologies and remains very difficult to treat.
METHODS: Case report of a 23-year-old male with a right corneal ulcer that failed to improve despite broad-spectrum antimicrobials.
RESULTS: Prior diagnosis of disseminated lymphangiomatosis with a lesion in the right petrous apex effacing Meckel's (trigeminal) cave in conjunction with a history of nonhealing corneal abrasions suggested a neurotrophic etiology. Drawstring temporary tarsorrhaphy, in addition to antibiotics and autologous serum, lead to successful clearing of the infection and resolution of the corneal ulcer. Visual acuity improved from light perception (LP) at the peak of infection to 20/40 six weeks after treatment.
CONCLUSIONS: To our knowledge, we report the first case of neurotrophic keratitis in a patient with disseminated lymphangiomatosis that caused a mass effect in Meckel's (trigeminal) cave leading to compression of the trigeminal nerve. The antibiotic-resistant corneal ulcer was successfully treated with drawstring tarsorrhaphy, confirming the utility of this therapeutic measure in treating neurotrophic keratitis.

Entities:  

Keywords:  corneal abrasion; disseminated lymphangiomatosis; drawstring tarsorrhaphy; neurotrophic keratitis

Year:  2009        PMID: 28539756      PMCID: PMC5436160          DOI: 10.2147/eb.s6957

Source DB:  PubMed          Journal:  Eye Brain        ISSN: 1179-2744


  7 in total

1.  The drawstring temporary tarsorrhaphy technique.

Authors:  John Kitchens; John Kinder; Thomas Oetting
Journal:  Arch Ophthalmol       Date:  2002-02

Review 2.  Neurotrophic keratitis.

Authors:  S Bonini; P Rama; D Olzi; A Lambiase
Journal:  Eye (Lond)       Date:  2003-11       Impact factor: 3.775

Review 3.  Thoracic lymphangiomas, lymphangiectasis, lymphangiomatosis, and lymphatic dysplasia syndrome.

Authors:  J L Faul; G J Berry; T V Colby; S J Ruoss; M B Walter; G D Rosen; T A Raffin
Journal:  Am J Respir Crit Care Med       Date:  2000-03       Impact factor: 21.405

4.  Congenital abnormalities of the lymphatic system: a new clinical classification.

Authors:  R I Hilliard; J B McKendry; M J Phillips
Journal:  Pediatrics       Date:  1990-12       Impact factor: 7.124

5.  A systematic method of drawing corneal pathologic conditions.

Authors:  G O Waring; P R Laibson
Journal:  Arch Ophthalmol       Date:  1977-09

6.  Primary disorders of the lymphatic vessels--a unified concept.

Authors:  C Levine
Journal:  J Pediatr Surg       Date:  1989-03       Impact factor: 2.545

7.  Mucus in the healthy and diseased eye.

Authors:  P Wright; I A Mackie
Journal:  Trans Ophthalmol Soc U K       Date:  1977-04
  7 in total
  1 in total

1.  Neurotrophic Keratopathy in Systemic Diseases: A Case Series on Patients Treated With rh-NGF.

Authors:  Alessandro Meduri; Giovanni William Oliverio; Antonio Valastro; Claudia Azzaro; Umberto Camellin; Francesco Franchina; Leandro Inferrera; Anna Roszkowska; Pasquale Aragona
Journal:  Front Med (Lausanne)       Date:  2022-05-30
  1 in total

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