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A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer

PURPOSE: To report a rare case of lattice corneal dystrophy type 1 (LCD1) with bilateral Mooren's ulcer. OBSERVATIONS: This case involved a 62-year-old male patient with LCD1 who presented with the primary complaint of experiencing pain and photophobia in both eyes for 2 months prior to his ini...

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Autores principales: Kayukawa, Kanae, Kitazawa, Koji, Wakimasu, Koichi, Sotozono, Chie, Kinoshita, Shigeru
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9883143/
https://www.ncbi.nlm.nih.gov/pubmed/36718435
http://dx.doi.org/10.1016/j.ajoc.2023.101796
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author Kayukawa, Kanae
Kitazawa, Koji
Wakimasu, Koichi
Sotozono, Chie
Kinoshita, Shigeru
author_facet Kayukawa, Kanae
Kitazawa, Koji
Wakimasu, Koichi
Sotozono, Chie
Kinoshita, Shigeru
author_sort Kayukawa, Kanae
collection PubMed
description PURPOSE: To report a rare case of lattice corneal dystrophy type 1 (LCD1) with bilateral Mooren's ulcer. OBSERVATIONS: This case involved a 62-year-old male patient with LCD1 who presented with the primary complaint of experiencing pain and photophobia in both eyes for 2 months prior to his initial visit. Upon examination, a peripheral corneal ulcer was observed in both eyes covering more than 3 of the 4 quadrants, accompanied with ciliary injection and severe corneal infiltration. He was diagnosed with Mooren's ulcer, and treatment with 0.1% betamethasone and 0.5% levofloxacin eye drops and systemic cyclosporine and betamethasone was initiated. At 1-month post treatment initiation, a remaining ulceration ridge was observed on the corneal surface in his left eye, which was subsequently resected. Complete epithelialization was achieved at 1-month postoperative in the left eye and after 6-months of conservative topical treatment in the right eye. At 8–9 years post onset of Mooren's ulcer, the patient underwent penetrating keratoplasty in both eyes while undergoing treatment with oral cyclosporine administration for severe corneal opacity due to progression of lattice dystrophy. Post treatment, there has been no recurrence of ulcerations, even though more that 10 years has passed since the onset of Mooren's ulcer. CONCLUSIONS AND IMPORTANCE: To the best of our knowledge, this is the first reported case of LCD1 with bilateral Mooren's ulcer, and in this rare case, the patient was successfully treated with a combination of steroid, cyclosporine, and peripheral superficial keratectomy, and a good visual outcome was achieved after penetrating keratoplasty (PK) under the use of systemic cyclosporine.
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spelling pubmed-98831432023-01-29 A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer Kayukawa, Kanae Kitazawa, Koji Wakimasu, Koichi Sotozono, Chie Kinoshita, Shigeru Am J Ophthalmol Case Rep Case Report PURPOSE: To report a rare case of lattice corneal dystrophy type 1 (LCD1) with bilateral Mooren's ulcer. OBSERVATIONS: This case involved a 62-year-old male patient with LCD1 who presented with the primary complaint of experiencing pain and photophobia in both eyes for 2 months prior to his initial visit. Upon examination, a peripheral corneal ulcer was observed in both eyes covering more than 3 of the 4 quadrants, accompanied with ciliary injection and severe corneal infiltration. He was diagnosed with Mooren's ulcer, and treatment with 0.1% betamethasone and 0.5% levofloxacin eye drops and systemic cyclosporine and betamethasone was initiated. At 1-month post treatment initiation, a remaining ulceration ridge was observed on the corneal surface in his left eye, which was subsequently resected. Complete epithelialization was achieved at 1-month postoperative in the left eye and after 6-months of conservative topical treatment in the right eye. At 8–9 years post onset of Mooren's ulcer, the patient underwent penetrating keratoplasty in both eyes while undergoing treatment with oral cyclosporine administration for severe corneal opacity due to progression of lattice dystrophy. Post treatment, there has been no recurrence of ulcerations, even though more that 10 years has passed since the onset of Mooren's ulcer. CONCLUSIONS AND IMPORTANCE: To the best of our knowledge, this is the first reported case of LCD1 with bilateral Mooren's ulcer, and in this rare case, the patient was successfully treated with a combination of steroid, cyclosporine, and peripheral superficial keratectomy, and a good visual outcome was achieved after penetrating keratoplasty (PK) under the use of systemic cyclosporine. Elsevier 2023-01-12 /pmc/articles/PMC9883143/ /pubmed/36718435 http://dx.doi.org/10.1016/j.ajoc.2023.101796 Text en © 2023 Published by Elsevier Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Kayukawa, Kanae
Kitazawa, Koji
Wakimasu, Koichi
Sotozono, Chie
Kinoshita, Shigeru
A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer
title A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer
title_full A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer
title_fullStr A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer
title_full_unstemmed A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer
title_short A case of lattice corneal dystrophy type 1 with bilateral Mooren's ulcer
title_sort case of lattice corneal dystrophy type 1 with bilateral mooren's ulcer
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9883143/
https://www.ncbi.nlm.nih.gov/pubmed/36718435
http://dx.doi.org/10.1016/j.ajoc.2023.101796
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