Bruton Tyrosine Kinase Inhibitor in 2 Patients With Vitreoretinal Lymphoma.

This case report describes the use of tirabrutinib to treat 2 individuals with vitreoretinal lymphoma.

Retinal Vasculitis Associated with SAPHO Syndrome Responding to Treatment with Adalimumab.

PURPOSE: To report a case of bilateral retinal vasculitis successfully managed with adalimumab in a patient with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. CASE REPORT: SAPHO syndrome was diagnosed in a 48-year-old female with bilateral blurred vision refractory to steroid eye drops. A preliminary ophthalmic examination revealed bilateral intermediate uveitis with vitreous opacity, and fluorescein angiography (FA) revealed dye leakage from peripheral retinal vessels. Because oral antirheumatic drugs were ineffective in treating her osteitis, her internist prescribed adalimumab, which resulted in rapid normalization of C-reactive protein and improvement of osteitis. Following treatment with adalimumab for 5 months, FA revealed significant improvement of retinal vasculitis. This is the first report on the use of adalimumab for retinal vasculitis associated with SAPHO syndrome. CONCLUSION: We described a rare case of retinal vasculitis associated with SAPHO syndrome. Adalimumab treatment was effective in treating both osteitis and retinal vasculitis.

Case Report: A Rare Orbital Abscess Caused by Dacryocystitis After Administration of Rebamipide Ophthalmic Suspension.

Background: Rebamipide ophthalmic suspension was launched in Japan in 2012 and is used for the treatment of dry eye. Case Presentation: We report two cases of orbital abscess, which resulted from dacryocystitis that occurred after administration of rebamipide ophthalmic suspension. Computed tomography images showed an eyeball deformity in one case and exophthalmos in the other. In both cases, light microscopy, scanning electron microscopy and energy dispersive X-ray examinations revealed lacrimal sac concretions, which contained calcium phosphate crystals that were surrounded by microorganisms. Lacrimal sac concretion removal from the lacrimal sacs during dacryocystorhinostomies was performed on both patients. Although the postoperative outcome was favorable in one case, vision was lost in the other case due to the development of retinal artery occlusion as a complication of the orbital abscess, despite lacrimal sac concretion removal and administration of antimicrobials. Conclusions: This is the first case report to highlight that rebamipide ophthalmic suspension can cause an orbital abscess via development of lacrimal sac concretion. Ophthalmologists should be aware that rebamipide ophthalmic suspension might induce the formation of concretion in the lacrimal sac.

Total retinal detachment and contractile movement of the disc in eyes with morning glory syndrome.

PURPOSE: This study aimed to report clinical characteristics, intraoperative findings, and surgical outcomes of 2 eyes of 2 patients with retinal detachment (RD) associated with morning glory syndrome (MGS) and contractile movement of the disc. OBSERVATIONS: Case 1 was a 3-year-old Japanese boy who presented with congenital microphthalmos in the left eye. The presence of total RD with MGS and contractile movement of the disc was confirmed by ophthalmoscopic examination. During vitrectomy, migration of perfluorocarbon liquid into the subretinal space was observed, suggesting a communication between the vitreous cavity and the subretinal space. Here, the retina was reattached briefly after the surgery; however, it detached again 2 months after surgery. Case 2 was a 3-month-old Japanese girl who was referred to our hospital for evaluation of RD and MGS in her left eye. Partial RD, MGS, and contractile movement of the disc were confirmed by ophthalmoscopic examination. The RD remained unchanged up to 18 months from the first visit, but atresia of the pupil was found 21 months after the first visit. Severe proliferative vitreoretinopathy and contractile movement of the disc were found after lensectomy. It was impossible to completely remove the membrane despite meticulous effort of peeling it off. At the 15-month follow-up, the retinal configuration improved but remained detached. CONCLUSION AND IMPORTANCE: Total RD with contractile movement of the disc associated with MGS is a rare condition that seems difficult to treat.

Intravitreal injection of bevacizumab for retinopathy of prematurity in an infant with peters anomaly.

PURPOSE: To report our findings in an infant with Peters anomaly type II whose retinopathy of prematurity (ROP) was treated with an anti-VEGF agent and surgeries. CASE REPORT: A male infant weighing 548 g was born prematurely at 23 weeks and 1 day with corneal opacity and shallow anterior chambers in both eyes. At the postmenstrual age of 35 weeks and 3 days, the infant was tentatively diagnosed with stage 3 ROP because of a dilated tunica vasculosa lentis and ultrasonographic findings. The boy was treated with bilateral intravitreal injections of bevacizumab (IVB) because laser photocoagulation of the retina could not be performed due to the corneal opacity. The retina in the right eye detached 3 times, namely 5 days, 16 days, and 7 months after the IVB; encircling the scleral buckle and a vitrectomy with endolaser photocoagulation were therefore required. In his left eye, the retina was reattached after the initial IVB, and no additional treatment was required. ROP was not reactivated in both eyes until the last examination at the age of 2 years and 6 months. CONCLUSIONS: Our results showed that IVB is a useful treatment for ROP in patients with Peters anomaly. However, a retinal detachment can be a complication after IVB. The optimal timing of IVB for ROP in infants with hazy media needs to be determined.

Optical coherence tomographic findings at the fixation point in a case of bilateral congenital macular coloboma.

BACKGROUND: Congenital macular coloboma is a rare ocular disease that consists of atrophic lesions in the macula with well-circumscribed borders. We report the findings of spectral domain optical coherence tomography (SD-OCT) at the fixation point in a case of bilateral macular coloboma. CASE REPORT: The subject is a 4-year-old boy. He visited our hospital at age 1 year and 4 months for the evaluation of strabismus. The fundus examination of both eyes showed round-shaped sharply-demarcated atrophic lesions involving the macula with large choroidal vessels and bared sclera at the base. Immunologic tests including toxoplasmosis, rubella, varicella, herpes virus, and human T-cell leukemia virus were all negative. At age 4 years and 1 month, cycloplegic refraction showed insignificant refractive errors and his best corrected visual acuity was 0.6 bilaterally. The SD-OCT showed a crater-like depression accompanying atrophic neurosensory retina, and the absence of retinal pigment epithelium and choroid. Examination of the fixation behavior by visuscope showed steady fixation with an area 0.5° nasal to the nasal edge of the atrophic lesion bilaterally. The SD-OCT findings at fixation area showed remaining normal retinal structures involving inner segment-outer segment (IS/OS) junction line. CONCLUSION: The findings of SD-OCT have been shown to be useful in the diagnosis of macular coloboma. In the fixation point, the structure of retina and choroid were well preserved.