| Peer-Reviewed

A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation

Received: 8 March 2021    Accepted: 30 March 2021    Published: 16 April 2021
Views:       Downloads:
Abstract

Purpose-To report a rare presentation of Vogt-Koyanagi-Harada (VKH) disease in a 55 year old female patient with unilateral limbal stem cell deficiency (LSCD) with corneal perforation. Observations-Patient presented to the out patient department with recurrent episodes of watering, pain, redness in the left eye. Examination revealed LSCD with areas of pannus and infiltration. She was treated medically. Patient came back after a year with a corneal perforation in her left eye. The perforation was sealed using cyanoacrylate glue. Six weeks later, she presented with a drop in vision in both eyes. Optical Coherence Tomography (OCT) in both eyes showed multiple sub retinal fluid pockets and Fundus Fluorescein Angiography (FFA) revealed multiple pinpoint leakages typical of Harada disease. The patient was investigated for systemic associations, which were found to be normal. She was managed with tapering doses of oral steroids. OCT showed resolution of fluid pockets at one month. There was a good control of the disease, with no signs of recurrence at 6 months follow up. Conclusion-The clinical association between the two entities has not been published in literature till date. Further data or reports on such cases would help throw light on any possible association between LSCD and VKH disease.

Published in International Journal of Ophthalmology & Visual Science (Volume 6, Issue 2)
DOI 10.11648/j.ijovs.20210602.12
Page(s) 72-74
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2024. Published by Science Publishing Group

Keywords

Vogt-Koyanagi Harada Disease, Limbal Stem Cell Deficiency, Corneal Perforation

References
[1] Marcelo TT, Iguban EB. A case report on Vogt-Koyanagi-Harada disease seen at a tertiary hospital in the Philippines. Ophthalmol Open J. 2018; 3 (1): 1-4.
[2] Sheriff F, Narayanan NS, Huttner AJ, Baehring JM. Vogt-Koyanagy-Harada syndrome: A novel case and a brief review of focal neurologic presentations. Neurol Neuroimmunol Neuroinflamm. 2014; 1 (4).
[3] Kunjal Sejpal, Pejman Bakhtiari, and Sophie X. Deng. Presentation, Diagnosis and Management of Limbal Stem Cell Deficiency. Middle East Afr J Ophthalmol. 2013 Jan-Mar; 20 (1): 5-10.
[4] Puangsricharern V, Tseng SC. Cytologic evidence of corneal diseases with limbal stem cell deficiency. Ophthalmology. 1995; 102: 1476-85.
[5] Sangwan VS, Jain V, Vemuganti GK, Murthy SI. Vernal keratoconjunctivitis with limbal stem cell deficiency. Cornea. 2011; 30: 491-6.
[6] Bhatia RP, Srivastava R, Ghosh A. Limbal stem cell study in contact lens wearers. Ann Ophthalmol (Skokie) 2009; 41: 87-92.
[7] Sridhar MS, Vemuganti GK, Bansal AK, Rao GN. Impression cytology-proven corneal stem cell deficiency in patients after surgeries involving the limbus. Cornea. 2001; 20: 145-8.
[8] Dua HS, Azuara-Blanco A. Autologous limbal transplantation in patients with unilateral corneal stem cell deficiency. Br J Ophthalmol. 2000; 84: 273-8.
[9] Read RW. Vogt-Koyanagy-Harada Disease. Ophthalmol Clin N Am 15. 2002; 333-341.
[10] Olivier Calvetti, Caroline Laurent-Coriat, Michel Paques. Vogt-Koyanagi-Harada disease. Orphanet. 2009.
[11] Rao NA, Gupta A, Dustin L, et al. Frequency of distinguishing clinical features in Vogt-Koyanagi-Harada disease. Ophthalmology. 2010; 117 (3): 591-599.
[12] Castillo TR, Noche RR, Fajardo RV. Vogt - Koyanagi - Harada disease in the Philippines. Philippine Journal of Ophthalmology. 1998; 23 (4): 159-162.
[13] Yao J, Chen Y, Shao T, et al. Bilateral acute angle closure glaucoma as a presentation of vogt-koyanagi-harada syndrome in four chinese patients: a small case series. Ocul Immunol Inflamm 2013; 21: 286-291.
[14] Read RW, Yu F, Accorinti M, et al. Evaluation of the effect on outcomes of the route of administration of corticosteroids in acute Vogt-Koyanagi-Harada disease. Am J Ophthalmol. 2006; 142: 119124.
[15] Fang W, Yang P, Vogt-Koyanagi-Harada Syndrome. Current Eye Research, 2008; 33 (7): 517-523.
[16] Lin, D., Chen, W., Zhang, G. et al. Comparison of the optical coherence tomographic characters between acute Vogt-Koyanagi-Harada disease and acute central serous chorioretinopathy. BMC Ophthalmol. 2014; 87.
[17] Nicula C, Szabo I. Vogt-Koyanagi-Harada syndrome Case report. Rom J Ophthalmol. 2016; 60 (3): 181-183.
[18] Brouzas D, Chatzoulis D, Galina E, Liaskou A, Koukoulomatis P. Corneal anesthesia in a case with Vogt-Koyanagi-Harada syndrome. Acta Ophthalmol Scand. 1997 Aug; 75 (4): 464-5.
Cite This Article
  • APA Style

    Shreesha Kumar Kodavoor, Soundarya Balajee, Ramamurthy Dandapani, Muralidhar Rajamani, Thenarasun Seethalakshmi Asaithambi. (2021). A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation. International Journal of Ophthalmology & Visual Science, 6(2), 72-74. https://doi.org/10.11648/j.ijovs.20210602.12

    Copy | Download

    ACS Style

    Shreesha Kumar Kodavoor; Soundarya Balajee; Ramamurthy Dandapani; Muralidhar Rajamani; Thenarasun Seethalakshmi Asaithambi. A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation. Int. J. Ophthalmol. Vis. Sci. 2021, 6(2), 72-74. doi: 10.11648/j.ijovs.20210602.12

    Copy | Download

    AMA Style

    Shreesha Kumar Kodavoor, Soundarya Balajee, Ramamurthy Dandapani, Muralidhar Rajamani, Thenarasun Seethalakshmi Asaithambi. A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation. Int J Ophthalmol Vis Sci. 2021;6(2):72-74. doi: 10.11648/j.ijovs.20210602.12

    Copy | Download

  • @article{10.11648/j.ijovs.20210602.12,
      author = {Shreesha Kumar Kodavoor and Soundarya Balajee and Ramamurthy Dandapani and Muralidhar Rajamani and Thenarasun Seethalakshmi Asaithambi},
      title = {A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation},
      journal = {International Journal of Ophthalmology & Visual Science},
      volume = {6},
      number = {2},
      pages = {72-74},
      doi = {10.11648/j.ijovs.20210602.12},
      url = {https://doi.org/10.11648/j.ijovs.20210602.12},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ijovs.20210602.12},
      abstract = {Purpose-To report a rare presentation of Vogt-Koyanagi-Harada (VKH) disease in a 55 year old female patient with unilateral limbal stem cell deficiency (LSCD) with corneal perforation. Observations-Patient presented to the out patient department with recurrent episodes of watering, pain, redness in the left eye. Examination revealed LSCD with areas of pannus and infiltration. She was treated medically. Patient came back after a year with a corneal perforation in her left eye. The perforation was sealed using cyanoacrylate glue. Six weeks later, she presented with a drop in vision in both eyes. Optical Coherence Tomography (OCT) in both eyes showed multiple sub retinal fluid pockets and Fundus Fluorescein Angiography (FFA) revealed multiple pinpoint leakages typical of Harada disease. The patient was investigated for systemic associations, which were found to be normal. She was managed with tapering doses of oral steroids. OCT showed resolution of fluid pockets at one month. There was a good control of the disease, with no signs of recurrence at 6 months follow up. Conclusion-The clinical association between the two entities has not been published in literature till date. Further data or reports on such cases would help throw light on any possible association between LSCD and VKH disease.},
     year = {2021}
    }
    

    Copy | Download

  • TY  - JOUR
    T1  - A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation
    AU  - Shreesha Kumar Kodavoor
    AU  - Soundarya Balajee
    AU  - Ramamurthy Dandapani
    AU  - Muralidhar Rajamani
    AU  - Thenarasun Seethalakshmi Asaithambi
    Y1  - 2021/04/16
    PY  - 2021
    N1  - https://doi.org/10.11648/j.ijovs.20210602.12
    DO  - 10.11648/j.ijovs.20210602.12
    T2  - International Journal of Ophthalmology & Visual Science
    JF  - International Journal of Ophthalmology & Visual Science
    JO  - International Journal of Ophthalmology & Visual Science
    SP  - 72
    EP  - 74
    PB  - Science Publishing Group
    SN  - 2637-3858
    UR  - https://doi.org/10.11648/j.ijovs.20210602.12
    AB  - Purpose-To report a rare presentation of Vogt-Koyanagi-Harada (VKH) disease in a 55 year old female patient with unilateral limbal stem cell deficiency (LSCD) with corneal perforation. Observations-Patient presented to the out patient department with recurrent episodes of watering, pain, redness in the left eye. Examination revealed LSCD with areas of pannus and infiltration. She was treated medically. Patient came back after a year with a corneal perforation in her left eye. The perforation was sealed using cyanoacrylate glue. Six weeks later, she presented with a drop in vision in both eyes. Optical Coherence Tomography (OCT) in both eyes showed multiple sub retinal fluid pockets and Fundus Fluorescein Angiography (FFA) revealed multiple pinpoint leakages typical of Harada disease. The patient was investigated for systemic associations, which were found to be normal. She was managed with tapering doses of oral steroids. OCT showed resolution of fluid pockets at one month. There was a good control of the disease, with no signs of recurrence at 6 months follow up. Conclusion-The clinical association between the two entities has not been published in literature till date. Further data or reports on such cases would help throw light on any possible association between LSCD and VKH disease.
    VL  - 6
    IS  - 2
    ER  - 

    Copy | Download

Author Information
  • Cornea Services, The Eye Foundation, Coimbatore, India

  • Cornea, Refractive and Cataract Surgery, The Eye Foundation, Coimbatore, India

  • Refractive and Cataract Surgery, The Eye Foundation, Coimbatore, India

  • Glaucoma, Neuro Ophthalmology, Paediatric Ophthalmology and Squint, The Eye Foundation, Coimbatore, India

  • Vitreoretina Services, the Eye Foundation, Coimbatore, India

  • Sections