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Successive onset of Vogt-Koyanagi-Harada syndrome in father and son

BACKGROUND: Vogt‒Koyanagi‒Harada (VKH) disease is a multifactorial systemic autoimmune disorder against melanocytes that is characterized by panuveitis. Familial occurrence of VKH disease is rare. Here, we report two cases of a father and his son with characteristic manifestations of VKH disease. CA...

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Autores principales: Li, Hougang, Sun, Shuo, Zhang, Yanrui, Liu, Jinfeng, Zhao, Xuzheng, Zhao, Guixia
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10029775/
https://www.ncbi.nlm.nih.gov/pubmed/36944975
http://dx.doi.org/10.1186/s12886-023-02851-2
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author Li, Hougang
Sun, Shuo
Zhang, Yanrui
Liu, Jinfeng
Zhao, Xuzheng
Zhao, Guixia
author_facet Li, Hougang
Sun, Shuo
Zhang, Yanrui
Liu, Jinfeng
Zhao, Xuzheng
Zhao, Guixia
author_sort Li, Hougang
collection PubMed
description BACKGROUND: Vogt‒Koyanagi‒Harada (VKH) disease is a multifactorial systemic autoimmune disorder against melanocytes that is characterized by panuveitis. Familial occurrence of VKH disease is rare. Here, we report two cases of a father and his son with characteristic manifestations of VKH disease. CASE PRESENTATION: A 53-year-old male with typical clinical symptoms of VKH disease was referred to Tangshan Eye Hospital. Examination showed the presence of ciliochoroidal effusion and exudative retinal detachment in both eyes. The patient was given intravenous methylprednisolone 120 mg for 2 days and intravenous methylprednisolone 80 mg for 1 day followed by 48 mg (1 mg/kg/day) oral methylprednisolone daily, accompanied by oral azathioprine 50 mg daily. Cycloplegic agent (0.5% tropicamide three times daily [TID]) was added. The patient was free of symptoms and recurrence within more than 1-year-follow-up period, the best corrected visual acuity (BVCA) was increased and maintained in both eyes with complete resolution of subretinal fluid. One year and nine months later, case 2 (his son) also presented with the typical clinical symptoms of VKH disease at 29 years of age. The son also recovered from VKH disease after routine and standard treatment. CONCLUSIONS: To the best of our knowledge, this is the first VKH disease case report of a father-son relationship. Although genetic factors have been demonstrated to be involved in the pathogenesis of VKH disease, the different inheritance modes of VKH patients need to be further explored and studied.
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spelling pubmed-100297752023-03-21 Successive onset of Vogt-Koyanagi-Harada syndrome in father and son Li, Hougang Sun, Shuo Zhang, Yanrui Liu, Jinfeng Zhao, Xuzheng Zhao, Guixia BMC Ophthalmol Case Report BACKGROUND: Vogt‒Koyanagi‒Harada (VKH) disease is a multifactorial systemic autoimmune disorder against melanocytes that is characterized by panuveitis. Familial occurrence of VKH disease is rare. Here, we report two cases of a father and his son with characteristic manifestations of VKH disease. CASE PRESENTATION: A 53-year-old male with typical clinical symptoms of VKH disease was referred to Tangshan Eye Hospital. Examination showed the presence of ciliochoroidal effusion and exudative retinal detachment in both eyes. The patient was given intravenous methylprednisolone 120 mg for 2 days and intravenous methylprednisolone 80 mg for 1 day followed by 48 mg (1 mg/kg/day) oral methylprednisolone daily, accompanied by oral azathioprine 50 mg daily. Cycloplegic agent (0.5% tropicamide three times daily [TID]) was added. The patient was free of symptoms and recurrence within more than 1-year-follow-up period, the best corrected visual acuity (BVCA) was increased and maintained in both eyes with complete resolution of subretinal fluid. One year and nine months later, case 2 (his son) also presented with the typical clinical symptoms of VKH disease at 29 years of age. The son also recovered from VKH disease after routine and standard treatment. CONCLUSIONS: To the best of our knowledge, this is the first VKH disease case report of a father-son relationship. Although genetic factors have been demonstrated to be involved in the pathogenesis of VKH disease, the different inheritance modes of VKH patients need to be further explored and studied. BioMed Central 2023-03-21 /pmc/articles/PMC10029775/ /pubmed/36944975 http://dx.doi.org/10.1186/s12886-023-02851-2 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Li, Hougang
Sun, Shuo
Zhang, Yanrui
Liu, Jinfeng
Zhao, Xuzheng
Zhao, Guixia
Successive onset of Vogt-Koyanagi-Harada syndrome in father and son
title Successive onset of Vogt-Koyanagi-Harada syndrome in father and son
title_full Successive onset of Vogt-Koyanagi-Harada syndrome in father and son
title_fullStr Successive onset of Vogt-Koyanagi-Harada syndrome in father and son
title_full_unstemmed Successive onset of Vogt-Koyanagi-Harada syndrome in father and son
title_short Successive onset of Vogt-Koyanagi-Harada syndrome in father and son
title_sort successive onset of vogt-koyanagi-harada syndrome in father and son
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10029775/
https://www.ncbi.nlm.nih.gov/pubmed/36944975
http://dx.doi.org/10.1186/s12886-023-02851-2
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