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| CASE REPORT |
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| Year : 2022 | Volume
: 30
| Issue : 3 | Page : 123-127 |
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Retinal Artery Macroaneurysm − An Uncommon Manifestation of Radiation Retinopathy
Divyansh Mishra, Mahesh Shanmugam, Sonali Lomte, Rajesh Ramanjulu, Surendra Pal
Department of Vitreoretina, Sankara Eye Hospital, Banglore, Karnataka, India
| Date of Submission | 19-Jun-2021 |
| Date of Decision | 19-Dec-2021 |
| Date of Acceptance | 28-Feb-2022 |
| Date of Web Publication | 09-Dec-2022 |
Correspondence Address:
Dr. Sonali Lomte
Department of Vitreoretina, Sankara Eye Hospital, Kundalhalli Gate, Airport, Varthur Road, Banglore 560037, Karnataka
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/njo.njo_38_21
To describe two cases of radiation retinopathy (RR) presenting with clinical and imaging characteristics of retinal artery macroaneurysm (RAM). The first case had radiotherapy to the right side of the brain 17 years prior to ocular manifestation, developed vitreous hemorrhage secondary to RAM in the right eye (RE) was managed with intravitreal anti-vascular endothelial growth factor. Second case had radiotherapy to right side of neck 6 years back, developed multiple tributary vein occlusions in RE, and was treated with sectoral laser. One year later, developed premacular and subretinal hemorrhage with RAM and managed with pars planavitrectomy and anti-VEGF. on subsequent follow up second patient had persistent cystoid macular edema which was managed with intravitreal triamcinolone acetonide and focal laser to RAM.
Keywords: Radiation retinopathy (RR), retinal artery macroaneurysm (RAM), Macular edema
How to cite this article:
Mishra D, Shanmugam M, Lomte S, Ramanjulu R, Pal S. Retinal Artery Macroaneurysm − An Uncommon Manifestation of Radiation Retinopathy. Niger J Ophthalmol 2022;30:123-7 |
Key Messages: Retinal artery macroaneurysm (RAM) although uncommon can present as radiation retinopathy.
| Introduction | |  |
Radiotherapy for cancers of the head and neck can result in radiation retinopathy (RR).[1] Therapeutic doses can cause RR between 6 months and 3 years postradiotherapy, with devastating visual outcomes.[2] Retinal artery macroaneurysms (RAMs) are round or fusiform dilatation of retinal arteries that occur within first three branches of retinal arteries. It is commonly observed in elderly females with hypertension.[3] In our cases, we highlight RAM as an unusual manifestation of RR which to the best of our knowledge has not been reported in any literature.
| Case Reports | | |
Case 1
A 33 year old male ,with history of radiotherapy to right side of brain 17 years before for hemangiopericytoma type 2 which is now in complete remission, presented with sudden painless diminution of vision in right eye (RE) of 2 days duration. He had no history of diabetes mellitus, hypertension, or any other systemic illness. External examination of the face showed right temporalis muscle atrophy [Figure 1]a,b. His best corrected visual acuity (BCVA) was 6/36 in RE and 6/6 in left eye (LE). Anterior segment (AS) of RE showed corneal filaments with punctate epithelial erosions and posterior chamber intraocular lens (had cataract surgery 17 years before), LE was normal. Schirmer test was 7 mm in RE and 19 mm in LE. Dilated fundus examination of RE demonstrated vitreous and preretinal hemorrhage with subretinal yellow deposits suggestive of hard exudates along the superior temporal arcade, LE was normal. He was advised to maintain restricted activity. On review 2 weeks later, vitreous hemorrhage had significantly regressed and fundus view was better than at the time of the presentation [Figure 1]c. Fundus fluorescein angiography (FFA) of RE [Figure 1]d–f showed perifoveal microaneurysm suggestive of microangiopathy and vitreous hemorrhage with no signs of neovascularization indicating haemorrhage secondary to RAM. RE optical coherence tomography (OCT) [Figure 2]a through fovea showed normal foveal contour, with preretinal hyperreflective material temporally suggestive of hemorrhage with some intraretinal hyperreflective foci and minimal retinal thickening. OCT angiography of RE [Figure 2]b did not show any preretinal neovascularization. Patient was treated with RE intravitreal anti-VEGF Bevacizumab (1.25 mg/0.05 mL). At 1 month of follow-up, BCVA in RE was 6/6. RE fundus examination showed significantly decreased vitreous and preretinal hemorrhage. OCT of RE [Figure 2]c revealed RAM which was masked by hemorrhage and dry fovea with minimal intraretinal fluid temporal to fovea. | Figure 1 (a,b) Radiation sequelae and (c) fundus photograph of right eye (RE) showing vitreous and preretinal hemorrhage, subretinal yellow deposits along superior temporal arcade, (d–f) fundus fluorescein angiography of RE showing focal hyperfluorescence due to microvascular abnormalities, blocked fluorescence due to preretinal and vitreous hemorrhage and late hyperfluorescence along the superotemporal artery likely RAM.
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 | Figure 2 (a) Optical coherence tomography (OCT) scan of right eye showing dry fovea with preretinal and intraretinal hyperreflective material, (b) no preretinal neovascularization observed on vitreoretinal slab of OCTA, (c) OCT following anti-VEGF showing round cavity with hyperreflective wall and dark lumen suggesting RAM (blue arrow) with intraretinal fluid temporal to fovea.
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Case 2
A 67-year-old male with history of intravenous chemotherapy and radiation to right side of the neck 6 years back for neck cancer came for routine examination. He had no history of hypertension, diabetes mellitus, or any other systemic illness. External examination of the face showed right torticollis with neck stiffness. His BCVA was 6/9 in RE and 6/6 in LE. AS of both eyes was normal. Dilated fundus examination of RE [Figure 3]a demonstrated microaneurysms at the posterior pole with multiple peripheral venous sclerosis. LE was normal [Figure 3]b. FFA [Figure 3]c–e of the RE revealed hyperfluorescence due to telangiectasia over posterior pole and multiple peripheral areas of hypofluorescence due to capillary nonperfusion (CNP), LE was normal. He was diagnosed to have nonproliferative RR and sectoral laser was performed to CNP areas. He was lost to follow-up and came 1 year later with complaints of diminution of vision in RE since 2 months. His BCVA was 6/60 in RE and 6/6 in LE. AS of RE showed nuclear sclerosis cataract and LE was normal. Dilated fundus examination of RE demonstrated hemorrhagic RAM along the superior temporal arcade with subhyaloid and subretinal hemorrhage, peripheral retina showed laser scars. LE was normal. The OCT [Figure 4]a showed preretinal material shadowing foveal details, intraretinal hyperreflectivity along the superior temporal vessel suggestive of RAM with subretinal fluid (SRF) and intraretinal hyperreflective material. He underwent RE phacoemulsification with intraocular lens (IOL) implantation, pars plana vitrectomy with internal limiting membrane peeling and intravitreal anti-VEGF. At 1 month of follow-up, RE revealed resolving hemorrhages and RAM lesion with peripheral laser scars and dull foveal reflex. OCT [Figure 4]b showed cystoid macular edema (CME) and RAM with minimal SRF. Intravitreal triamcinolone acetonide (IVTA) 2 mg with adjunct focal laser was performed to the RAM. One month after IVTA, BCVA was 6/24 in RE and 6/6 in LE. AS of RE showed posterior chamber intraocular lens (PCIOL), LE was normal. Dilated fundus examination of RE demonstrated involuted RAM with dry fovea and IVTA particles with peripheral laser marks, LE was normal. OCT RE [Figure 4]c showed resolved CME. | Figure 3 (a,b) Fundus photo of right eye (RE) showing perifoveal microaneurysms, left eye (LE) normal, (c,d) fundus fluorescein angiography (FFA) of RE showing hyperfluorescence due telangiectasia over posterior pole and hypofluorescence due to capillary nonperfusion areas, (e) LE FFA no significant abnormality noted.
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 | Figure 4 (a) Right eye optical coherence tomography (OCT) showing hyperreflective membrane in vitreoretinal interface suggestive of preretinal hemorrhage and intraretinal hyperreflectity suggestive of retinal artery macroaneurysm (RAM) with subretinal fluid (SRF), (b) OCT 1 month post-op showing cystoid macular edema (CME) and RAM with SRF, (c) OCT 1 month after intravitreal triamcinolone acetonide showing resolved CME and completely involuted RAM.
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| Discussion | | |
Radiation retinopathy, a frequently encountered complication of radiotherapy for ocular and periorbital cancers, is believed to be linked to dose, duration of treatment, and concomitant systemic factors.[4],[5],[6] Management of complications of RR such as macular edema and proliferative disease includes intravitreal anti-VEGF, IVTA, laser photocoagulation, hyperbaric oxygen, pentoxifylline, photodynamic therapy (PDT), and vitrectomy.[7],[8]
The RR typically presents with any combination of the following findings: macular edema, optic disc edema, telangiectasias, microaneurysms, cotton–wool spots, hard exudates, CNP, and retinal pigment epithelial atrophy, with advancement to proliferative disease which include vitreous hemorrhage, neovascular glaucoma, optic neuropathy, and tractional retinal detachment.[6]
However, to the best of our knowledge, RAM presenting as manifestation of RR has never been reported before. RAM is reported commonly in elderly females with hypertension and cardiovascular diseases. In our case, patient had radiation history to the face and he developed radiation sequelae to the face and eye on the same side. The factors that favor radiation retinopathy at the primary presentation are the faster progression to cataract when compared with other eye in both cases, multiple perifoveal microaneurysms in the first case and perifoveal microaneurysm with peripheral CNP areas in the second case. Thus, later development of RAM can also be associated with the RR and age acting as additive factor for the same in the second case.
Intracranial aneurysm following radiotherapy for head and neck cancer has been reported.[9] Similarly, coexistence between RAM and cerebral aneurysm has been reported.[10] Hence, we postulate that the pathophysiologic mechanism of radiation induced RAM is likely to resemble that of radiation-induced cerebral aneurysm, in which initial endothelial injury caused by radiation results in aneurysmal change.
Our patients did not have proliferative disease and vitreous hemorrhage was secondary to the RAM. These cases highlight the importance of careful review of past radiation history of an isolated RAM found in one eye of the patient. The treatment of radiation-induced RAM is same as that of other RAM, but patients should be followed up closely to check for the development of macular edema and proliferative disease due to radiation.
| Conclusion | | |
In our cases, patients with history of radiotherapy, in the absence of other systemic illnesses, clinical and imaging characteristics, rendered RAM secondary to radiation a likely diagnosis. RAM as sequel of radiotherapy and its management is not been reported before in literature.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Bagan SM, Hollenhorst RW. Radiation retinopathy after irradiation of intracranial lesions. Am J Ophthalmol 1979;88:694-7.  |
| 2. | Durkin SR, Roos D, Higgs B et al. Ophthalmic and adnexal complications of radiotherapy. Acta Ophthalmol Scand 2007;85:240-50. |
| 3. | Panton RW, Goldberg MF, Farber MD. Retinal arterial macroaneurysms: risk factors and natural history. Br J Ophthalmol 1990;74:595-600. |
| 4. | Brown GC, Shields JA, Sanborn G, Augsburger JJ, Savino PJ, Schatz NJ. Radiation retinopathy. Ophthalmology 1982;89:1494-501. |
| 5. | Viebahn M, Barricks ME, Osterloh MD. Synergism between diabetic and radiation retinopathy: case report and review. Br J Ophthalmol 1991;75;629-32. |
| 6. | Zamber RW, Kinyoun JL. Radiation retinopathy. West J Med 1992;157:530-3. |
| 7. | Giuliari GP, Sadaka A, Hinkle DM, Simpson ER. Current treatments for radiation retinopathy. Acta Oncol 2011;50:6-13. |
| 8. | Bansal AS, Bianciotto CG, Maguire JI, Regillo CD, Shields JA, Shields CL. Safety of pars plana vitrectomy in eyes with plaque-irradiated posterior uveal melanoma. Arch Ophthalmol 2012;130:1285-90. |
| 9. | Yang WH, Yang YH, Chen PC et al. Intracranial aneurysms formation after radiotherapy for head and neck cancer: a 10-year nationwide follow-up study. BMC Cancer 2019;537. |
| 10. | Serhad Nalcaci, Zafer Oztas, Cenk Eraslan, Cezmi Akkin. Are multiple retinal arterial macroaneurysms considered a sign of cerebral aneurysms? Ophthalmic Surg Lasers Imaging Retina 2017;48:79-82. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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