J Korean Ophthalmol Soc.  2011 Jun;52(6):742-745. 10.3341/jkos.2011.52.6.742.

Regression of Choroidal Neovascularization in a Choroideremia Patient after Intravitreal Bevacizumab Injection: A Case Report

• Kim DB
• Jung MO
• Cho SW
• Choi GJ
• Kim DH

• KMID: 2214649
• DOI: http://doi.org/10.3341/jkos.2011.52.6.742

Abstract

PURPOSE
To report the first domestic case of choroidal neovascularization in a choroideremia patient treated with intravitreal bevacizumab injection.
CASE SUMMARY
A 29-year-old male presented with a sudden decline in vision in the left eye. Fundus examination revealed areas of choriocapillaries and retinal pigment epithelium atrophy with macular hemorrhage. Fluorescein angiogram revealed vascular hyperfluorescence in the juxtafoveal area. Neurosensory detachment around the macula and increased central macular thickness was also observed using optical coherence tomography. Upon the diagnosis of choroideremia with choroidal neovascularization, the patient was treated with 1.25 mg intravitreal bevacizumab. Visual acuity improved after four injections of intravitreal Bevacizumab with improvement in both detachment and fluorescein leakage.
CONCLUSIONS
In patients with choroideremia presenting sudden decline in vision, ophthalmologists should detect for possible choroidal neovascularization. The results from the present study show that judicious use of intravitreal Bevacizumab may be effective in such cases. Further studies with a large sample size and sufficiently long follow-up periods are required.

Keyword

Bevacizumab; Choroideremia; Neovascularization

MeSH Terms

Adult
Antibodies, Monoclonal, Humanized
Atrophy
Choroid
Choroidal Neovascularization
Choroideremia
Eye
Fluorescein
Follow-Up Studies
Hemorrhage
Humans
Male
Retinal Pigment Epithelium
Sample Size
Tomography, Optical Coherence
Vision, Ocular
Visual Acuity
Bevacizumab
Antibodies, Monoclonal, Humanized
Fluorescein

Figure

• 

  Figure 1. Fundus photographs of right eye (A) and left eye (B) at presentation show areas of choriocapillaries and retinal pigment epithelium atrophy in the mid-periphery and posterior pole relatively sparing the macula. Note the macular hemorrhage in the left eye (B). Fluorescein angiogram of right eye (C) and left eye (D) at presentation show symmetrical areas of retinal pigment epithelium and choriocapillary loss. The fovea is spared with surrounding areas of hyperfluorescence. Note the leakage from a juxtafo-veal choroidal neovascularization (CNV) in the left eye (D).

• 

  Figure 2. The optical coherence tomographs (OCT) of the patient's left eye at initial visit shows increased central retinal thickness owing to neurosensory detachment and shallow elevation of the pigmented epithelium.

• 

  Figure 3. Fluorescein angiogram of the left eye after 3 intravitreal bevacizumab injections shows leakage in the newly developed juxtafoveal choroidal neovascularization (CNV) just above the previous lesion.

• 

  Figure 4. Fluorescein angiogram (A) 2 months after the 4th intravitreal bevacizumab injection shows complete leakage resolution. OCT (optical coherence tomographs) image (B) also shows resolved detachment and marked decrease of central retinal thickness.

Reference

References

1. Roberts MF, Fishman GA, Roberts DK, et al. Retrospective, longitudinal, and cross sectional study of visual acuity impairment in choroideraemia. Br J Ophthalmol. 2002; 86:658–62.

2. Wakabayashi K, Yoneura D, Kawasaki K, Uyama M. New approach to electrodiagnosis in diseases of the retina choroideremia. Folia Ophthalmol Japan. 1984; 35:775–9.

3. Robinson D, Tiedeman J. Choroideremia associated with a subretinal neovascular membrane. Case report. Retina. 1987; 7:70–4.

4. Noro Y, Yamaguchi K, Tamai M. A case of choroideremia associated with subretinal proliferation. Jap J Clin Ophthalmol. 1992; 46:962–3.

5. Endo K, Yuzawa M, Ohba N. Choroideremia associated with subretinal neovascular membrane. Acta Ophthalmol Scand. 2000; 78:483–6.

6. Sawa M, Tamaki Y, Klancnik JM Jr, Yannuzzi LA. Intraretinal foveal neovascularization in choroideremia. Retina. 2006; 26:585–8.

7. Mauthner H. Ein fall von choroideremia. Ber Nat Med Ver Innsbruck. 1872; 2:191.

8. Krill AE, Archer D. Classification of the choroidal atrophies. Am J Ophthalmol. 1971; 72:562–85.