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Intravitreal ranibizumab for choroidal neovascularization secondary to acute multifocal posterior placoid pigment epitheliopathy

2009; Wiley; Volume: 88; Issue: 2 Linguagem: Inglês

10.1111/j.1755-3768.2009.01541.x

1755-3768

Nikolaos Mavrakanas, Efstratios Mendrinos, Cyrus Tabatabay, Constantin J. Pournaras,

Glaucoma and retinal disorders

Editor, Acute multifocal posterior placoid pigment epitheliopathy (AMPPPE) is an acute self-limiting chorioretinal inflammatory disorder characterized by multifocal yellow-white, placoid lesions at the level of the retinal pigment epithelium (RPE). It has a good long-term prognosis for visual acuity, although most patients have residual symptoms and paracentral scotomas (Wolf et al. 1991). Choroidal neovascularization (CNV) is a very rare complication of AMPPPE (Bowie et al. 2005). We report the case of a 14-year-old girl who developed CNV as a complication of AMPPPE, and who was treated effectively with a single intravitreal (IVT) ranibizumab injection. A 14-year-old girl diagnosed 2 years previously with bilateral AMPPPE presented with a 2-week history of decreased visual acuity and metamorphopsia in her left eye (LE). Best-corrected visual acuity (BCVA) was 20/30 right eye (RE) and 20/40 LE. Slit-lamp examination of the anterior segment was without any abnormal findings and intraocular pressure was within normal limits for both eyes. Fundus biomicroscopy of the RE revealed RPE changes in the posterior pole and an extrafoveal pigmented scar (Fig. 1A). Fundus biomicroscopy (Fig. 1B), fluorescein angiography (FA) (Fig. 1D) and optical coherence tomography (OCT) (Fig. 1F) of the LE revealed CNV associated with neuroretinal serous detachment. After the risks and benefits of off-label IVT ranibizumab injection and alternate available treatment modalities were discussed with the patient and her parents, she elected to be treated with IVT ranibizumab 0.5 mg after providing informed consent. (A) Colour fundus photography showing a nasal extrafoveal pigmented scar and retinal pigment epithelial (RPE) changes in the right eye. (B) Colour fundus photography showing extrafoveal choroidal neovascularization (CNV) (black arrow) with neuroretinal serous detachment and RPE changes in the left eye (LE). (C) LE colour fundus photography 1 month after intravitreal (IVT) ranibizumab injection showing regression of the neuroretinal serous detachment and granular extrafoveal pigmentation corresponding to the involuted CNV (black arrow). (D) Late-phase fluorescein angiography (FA) showing leakage from a classic CNV before treatment, with diffuse RPE changes corresponding to healed acute multifocal posterior placoid pigment epitheliopathy lesions. (E) Late-phase FA 1 month after IVT ranibizumab showing staining but no CNV leakage. (F) Macular optical coherence tomography (OCT) shows a hyper-reflective subretinal complex above the RPE (white arrows) corresponding to CNV, and increased retinal thickness with subretinal fluid. (G) OCT 1 month after IVT ranibizumab shows marked CNV regression (white arrow) and reduced subretinal fluid. (H) OCT 12 months after ranibizumab injection showing no CNV recurrence with further reduction of the subretinal fluid. One month following IVT ranibizumab injection, the patient had no more metamorphopsia and BCVA improved to 20/20 LE. There were no local or systemic adverse events. At that time, fundus biomicroscopy revealed fine, deep granular pigmentation over the involuted CNV with reduction of the neuroretinal serous detachment (Fig. 1C). Fluorescein angiography showed staining but no CNV leakage (Fig. 1E) and OCT demonstrated marked reduction of both the neovascular tissue (with decreased retinal thickening overlying the area of CNV) and subretinal fluid (Fig. 1G). Despite the persistence of subretinal fluid on OCT, we decided not to perform additional IVT ranibizumab injections but to observe the patient because of her excellent visual recovery, absence of metamorphopsia and absence of leakage from the CNV as demonstrated in FA (Fig. 1E). Thereafter, the patient was followed up regularly with no signs of CNV recurrence; at the follow-up visit 12 months following ranibizumab injection, BCVA was still 20/20 LE with further reduction of the residual subretinal fluid on OCT (Fig. 1H). The patient had no visual complaints. Choroidal neovascularization may complicate a multitude of different conditions ranging from age-related macular degeneration (AMD) to inflammatory choroidopathies. In the treatment of neovascular AMD, IVT ranibizumab been shown to be superior to PDT (Brown et al. 2006). Because vascular endothelial growth factor (VEGF) is a key molecule in the development of CNV, anti-VEGF therapies have been used increasingly in a variety of diseases complicated by neovascularization (Guthoff & Goebel 2008; Kurup et al. 2008; Querques et al. 2008). More specifically, the use of IVT ranibizumab and bevacizumab has been reported recently in other types of chorioretinal inflammatory white-dot disorders, i.e multiple evanescent white-dot syndrome (Rouvas et al. 2007), multifocal choroiditis (Fine et al. 2008) and serpiginous choroiditis (Song & Roh 2008), with significant therapeutic effects. A previously reported case of AMPPE-associated CNV was treated with sub-Tenon triamcinolone injection and photodynamic therapy (Bowie et al. 2005). There are potential disadvantages in treating CNV complicating chorioretinal inflammatory diseases with PDT. Photodynamic therapy can cause localized inflammation and increased VEGF production with a higher incidence of recurrent CNV. In addition, PDT is known to cause collateral damage to the choriocapillaris and RPE, which can result in permanent visual dysfunction. To the best of our knowledge, this is the first case of CNV secondary to AMPPPE to be treated with an IVT anti-VEGF. Moreover, in our patient, a single IVT injection of ranibizumab resulted in rapid and persistent regression of CNV with excellent visual outcomes, minimal macular scar formation and no scotoma. The speed and persistence of neovascular regression suggest that IVT ranibizumab may be an efficient treatment modality for this condition. IVT ranibizumab could be considered as a new and promising treatment for AMPPPE-associated CNV and merits further investigation.