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

• Published in: BMC ophthalmology 2023-03, Vol.23 (1), p.113-113, Article 113
• Main Authors: Li, Hougang, Sun, Shuo, Zhang, Yanrui, Liu, Jinfeng, Zhao, Xuzheng, Zhao, Guixia
• Format: Article
• Language: English
• Subjects: Acuity; Adult; Antigens; Autoimmune diseases; Azathioprine; Case Report; Case reports; Coronaviruses; Corticosteroids; COVID-19; Disease; Edema; Effusion; Eye; Familial cases; Family medical history; Fathers; Genetic aspects; Genetic factors; Glaucoma; Glucocorticoids - therapeutic use; Humans; Infections; Intravenous administration; Leukocytes; Male; Medical imaging; Melanocytes; Methylprednisolone; Methylprednisolone - therapeutic use; Middle Aged; Nuclear Family; Ophthalmology; Pathogenesis; Patients; Retinal detachment; Tomography; Treatment Outcome; Uveomeningoencephalitic Syndrome - complications; Uveomeningoencephalitic Syndrome - diagnosis; Uveomeningoencephalitic Syndrome - drug therapy; Viral infections; Visual Acuity; Vogt‒Koyanagi‒Harada syndrome
• ISSN: 1471-2415; 1471-2415
• DOI: 10.1186/s12886-023-02851-2

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. 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. 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.