About MEAJO | Editorial board | Search | Ahead of print | Current Issue | Archives | Instructions to authors | Online submission | Subscribe | Advertise | Contact | Login 
Middle East African Journal of Ophthalmology Middle East African Journal of Ophthalmology
Users Online: 277   Home Print this page Email this page Small font sizeDefault font sizeIncrease font size


 
  Table of Contents 
CASE REPORT
Year : 2019  |  Volume : 26  |  Issue : 2  |  Page : 117-119  

Case report of a 4-year-old child with complicated Vogt-Koyanagi-Harada at a tertiary eye hospital


1 College of Medicine, King Saud bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia
2 Department of Pediatric Ophthalmology, King Khalid Eye Specialist Hospital, Riyadh, Saudi Arabia

Date of Web Publication26-Aug-2019

Correspondence Address:
Dr. Mohammed Al Shamrani
Department of Pediatric Ophthalmology, King Khalid Eye Specialist Hospital, P.O. Box 7191, Riyadh 11462
Saudi Arabia
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/meajo.MEAJO_244_18

Rights and Permissions
   Abstract 


Vogt-Koyanagi-Harada (VKH) disease is a chronic, bilateral, granulomatous panuveitis associated with cutaneous, neurologic, and auditory manifestations. We report a 4-year-old Saudi boy who developed severe ocular complications by 5 years of age. He presented to King Khalid Eye Specialist Hospital at the age of 4 years and was previously operated on elsewhere for cataract with intraocular lens implantation in his right eye at the age of 3 years. He consecutively had iris capture and membrane formation around the intraocular lens. Examination revealed band keratopathy, posterior synechiae, and fundus depigmentation in both eyes with cataract formation in his left eye. At the age of 5.5 years, he developed subretinal neovascular membrane formation in the left eye. To the best of our knowledge, this patient is youngest VKH case that manifested most of the major complications at a young age as 5 years old.

Keywords: Children, uveitis, Vogt-Koyanagi-Harada


How to cite this article:
AlQahtani DS, Al Shamrani M. Case report of a 4-year-old child with complicated Vogt-Koyanagi-Harada at a tertiary eye hospital. Middle East Afr J Ophthalmol 2019;26:117-9

How to cite this URL:
AlQahtani DS, Al Shamrani M. Case report of a 4-year-old child with complicated Vogt-Koyanagi-Harada at a tertiary eye hospital. Middle East Afr J Ophthalmol [serial online] 2019 [cited 2019 Sep 17];26:117-9. Available from: http://www.meajo.org/text.asp?2019/26/2/117/265369




   Introduction Top


Vogt-Koyanagi-Harada (VKH) disease is a chronic, bilateral, granulomatous panuveitis associated with cutaneous, neurologic, and auditory manifestations.[1] Patients present initially with ocular complaints such as blurry vision, photophobia, and ocular pain in the absence of trauma or surgical history. Typical ocular findings are iridocyclitis, cellular vitreous infiltrate, serous retinal detachment, and disc hyperemia.[2] Although most of VKH patients are adults, literature shows that children can be affected as well.[1],[3] Long-standing, poorly controlled uveitis can be vision threatening and leads to invariable complications such as cataract, glaucoma, subretinal fibrosis, and subretinal neovascular membranes.[1] We report a case of a child who developed most of the major VKH complications by 5 years of age which rarely manifest in children.


   Case Report Top


A 4-year-old Saudi boy was referred to King Khaled Eye Specialist Hospital as VKH case after cataract extraction with intraocular lens implantation (IOL) in his right eye at another health-care facility at the age of 3.5 years. He was on daily 10 mg of prednisone, prednisolone ophthalmic drops four times daily, and daily 70 mg of cyclosporine.

On examination, he was poorly fixating and following in both eyes. The intraocular pressure (IOP) was digitally within normal values. Slit-lamp examination of the anterior segment showed clear cornea in the right eye and band keratopathy nasally in the left eye. The anterior chamber was deep with +3 cells in both eyes. Posterior synechiae were evident in both eyes, with iris atrophy in the left eye. The right eye was pseudophakic with iris capture and membrane around the IOL [Figure 1]. The left lens had early cataract changes. There was no view to the posterior segment. Ultrasound B-scan showed mild vitreous opacity and flat retina in both eyes. He also had systemic involvement manifested as vitiligo and polisosis.
Figure 1: Right eye: Iris capture with intraocular lens implant

Click here to view


Routine uveitis investigations were normal. The child was started on adalimumab 20 mg subcutaneously every 2 weeks, methotrexate injections (started with 10 mg weekly and increased to 15 mg weekly), and folic acid supplements. After 5 months on systemic medications, his eyes were quiet, and he was scheduled for cataract surgery to the left eye and IOL removal in the right eye. The child was prepared with three doses of daily intravenous methylprednisolone 15 mg/kg/day and hourly and topical prednisolone ophthalmic drops. Lens aspiration and anterior vitrectomy with intravitreal triamcinolone 2 mg and subconjunctival triamcinolone 20 mg were applied for the left eye. After 1 week, the IOL was removed from the right eye with intravitreal triamcinolone 2 mg and subconjunctival triamcinolone 20 mg. He had expected mild inflammation postoperatively that was controlled with treatment. Three months after surgery, best-corrected visual acuity (BCVA) was 20/60 in both eyes. Two months later (at age 5.5 years), BCVA dropped to 3/200 in the left eye. He had mild aqueous flare in both eyes and subretinal blood in the macula of the left eye. Fundus fluorescein angiography, optical coherence tomography, and fundus photograph confirmed the diagnosis of choroidal neovascular membrane in the macula of the left eye [Figure 2]. Thus, the patient was treated with intravitreal bevacizumab (Avastin) injection (1.25 mg) to the left eye under general anesthesia. Two months after the injection, the membrane was scarred with no activity [Figure 3]. In his last visit after 7 months (6 years of age), he continued to have inactive VKH using adalimumab 20 mg/2 weeks, methotrexate injections 15 mg/week, and topical prednisone ophthalmic drops twice daily in both eyes. The patient's BCVA was 20/30 in the right eye and 20/80 in the left eye with normal IOP both eyes.
Figure 2: (a) Fundus fluorescein angiography, (b) wide field color fundus photograph

Click here to view
Figure 3: Left eye optical coherence tomography. (a) choroidal neovascular membrane before treatment. (b) Two months after treatment

Click here to view



   Discussion Top


VKH is a disease uncommonly affects the pediatric age group. Only 13%–15% of VKH patients were found to be children.[3],[4] The visual prognosis in this population is variably reported in the literature with poor visual prognosis attributed to younger age at presentation and high number of complications.[1],[3],[5] Shorter duration between the onset of disease and initiation of treatment (<2 weeks) is reported to be associated with better visual outcome and lesser complications.[1]

Our patient developed several complications and was operated with probably poor control of his disease before he presented to our center. On the initial examination at the age of 4.4 years, he had band-shaped keratopathy, posterior synechiae, and fundus depigmentation in both eyes. Furthermore, he had IOL capture in the right eye and cataract in the left eye. After stabilizing the patient for several months, he developed subretinal neovascular membrane at the age of 5.5 years. Subretinal neovascularization was reported in 2 of 23 patients in Abu El-Asrar et al. series.[1] Different subretinal changes (neovascularization, scar, and retinal pigment epithelial migration) were reported in 7 of 13 patients in Tobbara et al.[3] However, in both series, the age for subretinal neovascularization development was not indicated.

To the best of our knowledge, our patient is the youngest reported VKH case that developed subretinal neovascular membrane at this young age. The only younger published case was a 3-year-old boy with bilateral hyperemia and serous retinal detachments for which he was treated and stabilized with no further relapses or complications.[6]


   Conclusion Top


Early diagnosis and treatment of the disease can prevent most of the complications and lead to a favorable visual outcome. However, the nature of this age group and difficulties in expressing their problems can delay the diagnosis and lead to serious visual compromise.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Abu El-Asrar AM, Al-Kharashi AS, Aldibhi H, Al-Fraykh H, Kangave D. Vogt-Koyanagi-Harada disease in children. Eye (Lond) 2008;22:1124-31.  Back to cited text no. 1
    
2.
Forster DJ, Green RL, Rao NA. Unilateral manifestation of the Vogt-Koyanagi-Harada syndrome in a 7-year-old child. Am J Ophthalmol 1991;111:380-2.  Back to cited text no. 2
    
3.
Tabbara KF, Chavis PS, Freeman WR. Vogt-Koyanagi-Harada syndrome in children compared to adults. Acta Ophthalmol Scand 1998;76:723-6.  Back to cited text no. 3
    
4.
Tugal-Tutkun I, Ozyazgan Y, Akova YA, Sullu Y, Akyol N, Soylu M, et al. The spectrum of Vogt-Koyanagi- Harada disease in Turkey: VKH in turkey. Int Ophthalmol 2007;27:117-23.  Back to cited text no. 4
    
5.
Ohno S, Minakawa R, Matsuda H. Clinical studies of Vogt-Koyanagi-Harada's disease. Jpn J Ophthalmol 1988;32:334-43.  Back to cited text no. 5
    
6.
Takada S, Tanaka R, Kurita N, Ishii K, Kaburaki T. Vogt-Koyanagi-Harada disease in 3-year-old boy. Clin Exp Ophthalmol 2015;43:593-4.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
  
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed88    
    Printed2    
    Emailed0    
    PDF Downloaded16    
    Comments [Add]    

Recommend this journal