Long-Term Control of Macular Edema With Adalimumab After Cataract Surgery in a Japanese Child With Juvenile Idiopathic Arthritis: Case Report and Review of 26 Japanese Patients

Abstract

Juvenile idiopathic arthritis-associated uveitis is rare in the Japanese population. In this article, we report a child whose macular edema was controlled for years after cataract surgery with adalimumab, and reviewed 26 Japanese patients in the literature. In this case report, a 4-year-old boy developed band keratopathy, posterior iris synechiae, and complicated cataract in both eyes. Oral prednisolone prescribed at another hospital was discontinued due to high intraocular pressure in both eyes as a steroid responder. At the age of 5 years, he started oral methotrexate 8 mg weekly for recurrent bilateral iridocyclitis and then underwent lensectomy with core vitrectomy in both eyes. Planned intraocular lens implantation was cancelled at surgery because the anterior vitreous had severe inflammatory opacity with diffuse retinal edema in both eyes. Due to persistent macular edema in both eyes 5 months postoperatively, at the age of 6 years, he began to use adalimumab injection 20 mg every 2 weeks. The macular structure depicted by optical coherence tomography became normal in 2 months. At final visit at the age of 11 years, he had the best-corrected visual acuity of 0.8 in the right eye and 0.4 in the left eye, with adalimumab 40 mg every 2 weeks and methotrexate 8 mg weekly. In conclusion, macular edema persistent despite oral methotrexate after cataract surgery could be controlled for long term by adalimumab in a child with juvenile idiopathic arthritis. In the Japanese literature, only 26 additional cases with juvenile idiopathic arthritis-associated uveitis have been reported so far.

Keywords: JIA; Japanese; adalimumab; cataract surgery; juvenile idiopathic arthritis; literature review; macular edema; methotrexate; steroid responder; uveitis.

Figure 1.

Slit-lamp biomicroscopic photographs. At the age of 5 years and 6 months (A, right eye; B, left eye) when oral methotrexate was started. At the age of 5 years and 8 months just before cataract surgery (C, right eye; D, left eye) and 5 days after the surgery (E, right eye; F, left eye). At the age of 7 years (G, right eye; H, left eye), 1 year after the start of adalimumab. Note that dense band keratopathy in both eyes (C, D) has been scaled off and that inflammation has subsided in aphakic eyes (E, F).

Figure 2.

Optical coherence tomography. At the age of 5 years of 11 months (A, right eye; B, left eye), 1 month before the start of adalimumab. At the age of 6 years and 2 months (C, right eye; D, left eye), 2 months after the start of adalimumab. At the age of 8 years and 1 month (E, right eye; F, left eye). Note that macular edema in both eyes (A, B) has been resolved in 2 months (C, D) after the start of adalimumab. Note also that ellipsoid zone at the fovea (arrows in E, F), indicative of photoreceptor outer segments, is more well visualized in the right eye (E) compared with the left eye (F), explaining better visual acuity in the right eye.