SUBRETINAL TRIAMCINOLONE ACETONIDE ASSOCIATED WITH IMPROVEMENT OF CYSTOID MACULAR EDEMA IN A PATIENT WITH RETINITIS PIGMENTOSA Kathleen E. Urban, MD,* Polly A. Quiram, MD, PHD,† Michael T. Trese, MD†
Purpose: To report a case of traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Methods: Single case report. Results: We describe a 17-year-old boy with retinitis pigmentosa and severe, persistent cystoid macular edema with foveal cysts resistant to surgical intervention who had marked clinical improvement after traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Conclusion: Subretinal delivery of triamcinolone acetonide may be more effective than preretinal steroid administration in reducing foveal cysts, particularly in retinitis pigmentosa. In this case, the duration of triamcinolone acetonide in the subretinal space seemed to be on the order of 3 months. RETINAL CASES & BRIEF REPORTS 3:47– 49, 2009
From *Beaumont Eye Institute, Beaumont Hospital, Royal Oak, Michigan; and †Associated Retinal Consultants, Beaumont Hospital, Royal Oak, Michigan.
treated with autologous plasmin enzyme–assisted vitrectomy without internal limiting membrane peeling. The patient was intolerant of acetazolamide treatment. Preoperatively, best-corrected visual acuity was 20/200 (Snellen visual acuity chart), with a foveal thickness of ⬎1,000 m measured by optical coherence tomography. Postoperatively, best-corrected visual acuity was 20/200, and optical coherence tomography findings were unchanged (data not shown). Because of the presence of persistent foveal cysts for several months after surgical intervention, 4 mg of intravitreal triamcinolone acetonide was injected in the right eye. Foveal thickness measured by optical coherence tomography decreased from ⬎1,000 m to ⬇600 m within 3 months. Best-corrected visual acuity improved from 20/200 to 20/100. Intravitreal triamcinolone acetonide injections were repeated every 4 months in the operating department. Two days after the fourth injection of intravitreal triamcinolone acetonide, the patient developed severe pain and photophobia and was diagnosed with endophthalmitis. Injection of intravitreal antibiotics and vitreous biopsy were performed. The same day, the patient was a passenger in severe car accident involving a fatality of another passenger. The patient had additional injuries including a cervical spine fracture. Two days after the accident, fundus examination of the right eye revealed a murky inferior vitreous cavity with no view of the inferior retina; the superior retina and macula were attached. After
C
onventional therapies for retinitis pigmentosa– related cystoid macular edema include acetazolamide, intravitreal triamcinolone, and vitrectomy with variable success.1,2 We report a case of severe, persistent cystoid macular edema with foveal cysts resistant to surgical intervention. The patient had marked clinical improvement after traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Case Report
A 17-year-old boy with retinitis pigmentosa and decreased visual acuity with large foveal cysts in the right eye (Fig. 1) was None of the authors have any proprietary interest to disclose. Reprint requests: Kathleen E. Urban, MD, 3535 West 13 Mile Road, Suite 555, Beaumont Eye Institute, William Beaumont Hospital, Royal Oak, MI 48073; e-mail: [email protected]
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Fig. 1. Optical coherence tomography image of the right eye demonstrating large foveal cysts preoperatively. Foveal thickness was ⬎1,000 m.
1 week of observation, the vitreous haze had partially cleared, and the patient was found to have a traumatic retinal detachment inferiorly, which was confirmed with B-scan ultrasonography. In addition, he had what appeared to be a thickened yellow area beneath the retina inferiorly (Fig. 2). At clinical examination, this was thought to be perhaps a granuloma, despite no known history of granulomatous disease; however, given the history of recent endophthalmitis, we could not rule out an infectious etiology for the yellow lesion. The patient was brought to the operating department for retinal detachment repair with scleral buckling and vitrectomy on the right eye. As he lay in the supine position on the operating department table, the yellow lesion was seen to migrate within the subretinal space to the nasal retina adjacent to the optic nerve. Intraoperatively, it was revealed that this was, in fact, a bolus of triamcinolone acetonide (injected intravitreally 3 weeks earlier) that had migrated into the subretinal space through a traumatic retinal dialysis inferiorly. Fine particles of triamcinolone acetonide were seen within the subretinal space inferior to the macula. The retina was repaired without incident. Three weeks postoperatively, optical coherence tomography revealed the fovea, adjacent to this bolus of subretinal steroid, to be
Fig. 2. Fundus photograph of the right eye showing traumatic displacement of triamcinolone acetonide into the subretinal space after successful retinal detachment repair.
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Fig. 3. Optical coherence tomography image of the right eye 3 weeks after surgical repair of a retinal detachment demonstrating complete resolution of the large foveal cyst. Best-corrected visual acuity was 20/100.
completely normal in thickness and contour (Fig. 3). Visual acuity remained 20/200. At ⬇3 months after intravitreal injection, the subretinal bolus of triamcinolone acetonide was decreasing in size. The foveal contour remained normal.
Discussion Pharmacologic therapies for retinitis pigmentosa– related cystoid macular edema include acetazolamide and intravitreal triamcinolone.1–3 We describe a patient with severe cystoid macular edema and large foveal cysts who had remarkable clinical improvement after traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Although we cannot draw any definitive conclusions from our patient’s complex clinical course, this case suggests that subretinal delivery of triamcinolone acetonide may be more effective than preretinal steroid administration in reducing foveal cysts, particularly in retinitis pigmentosa. In this case, the duration of triamcinolone acetonide in the subretinal space seemed to be on the order of 3 months. Why the nasally located subretinal bolus of triamcinolone acetonide had such a significant effect on the macula is not entirely clear. At clinical examination, particles of triamcinolone acetonide were visible inferior to the macula. It is possible that the inferior retinal detachment allowed triamcinolone acetonide to be widely distributed in the subretinal space, which consequently may have had a widespread effect on the retina. Novel drug delivery has been studied in humans and animal models.3–5 Subretinal steroid injection has been shown to be safe in rabbits.3 One pilot study used subretinal triamcinolone administration as an adjunct to vitrectomy for age-related macular degeneration; the results showed moderate visual improvement compared with controls.4 Ocular hypertension was the primary complication that was controlled with medical management. In addition, drug delivery by cannu-
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TRIAMCINOLONE ACETONIDE AND CME IN RP
lation of the suprachoroidal space has been studied in animal models.5 Further studies are needed to evaluate the potential side effects and/or retinal toxicity associated with subretinal triamcinolone treatment of retinal disease. It is unclear whether subretinal triamcinolone has an effect on visual acuity, but our patient did report a subjective improvement in vision after resolution of foveal cysts. Further, the potential untoward consequences for visual function of detaching the retina temporal to the macula or in the paramacular region in a patient with retinitis pigmentosa by the subretinal injection of a steroid need to be considered. Key words: subretinal space, triamcinolone acetonide, retinitis pigmentosa, cystoid macular edema.
References 1.
2.
3.
4.
5.
Fishman GA, Gilbert LD, Fiscella RG, et al. Acetazolamide for treatment of chronic macular edema in retinitis pigmentosa. Arch Ophthalmol 1989;107:1445–1452. Ke JE. Intravitreal triamcinolone acetonide for treatment of cystoid macular edema associated with retinitis pigmentosa. Retina 2006;26:1094–1096. Kozak I, Cheng L, Mendez T, et al. Evaluation of the toxicity of subretinal triamcinolone acetonide in the rabbit. Retina 2006;26:811–817. Kertes PJ, Coupland SG. The use of subretinal triamcinolone acetonide in the management of neovascular age-related macular degeneration: a pilot study. Can J Ophthalmol 2005;40: 573–584. Olsen TW, Feng X, Wabner K, et al. Cannulation of the suprachoroidal space: a novel drug delivery methodology to the posterior segment. Am J Ophthalmol 2006;142:777–787.
Purpose: To report a case of traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Methods: Single case report. Results: We describe a 17-year-old boy with retinitis pigmentosa and severe, persistent cystoid macular edema with foveal cysts resistant to surgical intervention who had marked clinical improvement after traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Conclusion: Subretinal delivery of triamcinolone acetonide may be more effective than preretinal steroid administration in reducing foveal cysts, particularly in retinitis pigmentosa. In this case, the duration of triamcinolone acetonide in the subretinal space seemed to be on the order of 3 months. RETINAL CASES & BRIEF REPORTS 3:47– 49, 2009
From *Beaumont Eye Institute, Beaumont Hospital, Royal Oak, Michigan; and †Associated Retinal Consultants, Beaumont Hospital, Royal Oak, Michigan.
treated with autologous plasmin enzyme–assisted vitrectomy without internal limiting membrane peeling. The patient was intolerant of acetazolamide treatment. Preoperatively, best-corrected visual acuity was 20/200 (Snellen visual acuity chart), with a foveal thickness of ⬎1,000 m measured by optical coherence tomography. Postoperatively, best-corrected visual acuity was 20/200, and optical coherence tomography findings were unchanged (data not shown). Because of the presence of persistent foveal cysts for several months after surgical intervention, 4 mg of intravitreal triamcinolone acetonide was injected in the right eye. Foveal thickness measured by optical coherence tomography decreased from ⬎1,000 m to ⬇600 m within 3 months. Best-corrected visual acuity improved from 20/200 to 20/100. Intravitreal triamcinolone acetonide injections were repeated every 4 months in the operating department. Two days after the fourth injection of intravitreal triamcinolone acetonide, the patient developed severe pain and photophobia and was diagnosed with endophthalmitis. Injection of intravitreal antibiotics and vitreous biopsy were performed. The same day, the patient was a passenger in severe car accident involving a fatality of another passenger. The patient had additional injuries including a cervical spine fracture. Two days after the accident, fundus examination of the right eye revealed a murky inferior vitreous cavity with no view of the inferior retina; the superior retina and macula were attached. After
C
onventional therapies for retinitis pigmentosa– related cystoid macular edema include acetazolamide, intravitreal triamcinolone, and vitrectomy with variable success.1,2 We report a case of severe, persistent cystoid macular edema with foveal cysts resistant to surgical intervention. The patient had marked clinical improvement after traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Case Report
A 17-year-old boy with retinitis pigmentosa and decreased visual acuity with large foveal cysts in the right eye (Fig. 1) was None of the authors have any proprietary interest to disclose. Reprint requests: Kathleen E. Urban, MD, 3535 West 13 Mile Road, Suite 555, Beaumont Eye Institute, William Beaumont Hospital, Royal Oak, MI 48073; e-mail: [email protected]
47
48
RETINAL CASES & BRIEF REPORTSℜ
Fig. 1. Optical coherence tomography image of the right eye demonstrating large foveal cysts preoperatively. Foveal thickness was ⬎1,000 m.
1 week of observation, the vitreous haze had partially cleared, and the patient was found to have a traumatic retinal detachment inferiorly, which was confirmed with B-scan ultrasonography. In addition, he had what appeared to be a thickened yellow area beneath the retina inferiorly (Fig. 2). At clinical examination, this was thought to be perhaps a granuloma, despite no known history of granulomatous disease; however, given the history of recent endophthalmitis, we could not rule out an infectious etiology for the yellow lesion. The patient was brought to the operating department for retinal detachment repair with scleral buckling and vitrectomy on the right eye. As he lay in the supine position on the operating department table, the yellow lesion was seen to migrate within the subretinal space to the nasal retina adjacent to the optic nerve. Intraoperatively, it was revealed that this was, in fact, a bolus of triamcinolone acetonide (injected intravitreally 3 weeks earlier) that had migrated into the subretinal space through a traumatic retinal dialysis inferiorly. Fine particles of triamcinolone acetonide were seen within the subretinal space inferior to the macula. The retina was repaired without incident. Three weeks postoperatively, optical coherence tomography revealed the fovea, adjacent to this bolus of subretinal steroid, to be
Fig. 2. Fundus photograph of the right eye showing traumatic displacement of triamcinolone acetonide into the subretinal space after successful retinal detachment repair.
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2009
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VOLUME 3
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NUMBER 1
Fig. 3. Optical coherence tomography image of the right eye 3 weeks after surgical repair of a retinal detachment demonstrating complete resolution of the large foveal cyst. Best-corrected visual acuity was 20/100.
completely normal in thickness and contour (Fig. 3). Visual acuity remained 20/200. At ⬇3 months after intravitreal injection, the subretinal bolus of triamcinolone acetonide was decreasing in size. The foveal contour remained normal.
Discussion Pharmacologic therapies for retinitis pigmentosa– related cystoid macular edema include acetazolamide and intravitreal triamcinolone.1–3 We describe a patient with severe cystoid macular edema and large foveal cysts who had remarkable clinical improvement after traumatic displacement of intravitreal triamcinolone acetonide into the subretinal space. Although we cannot draw any definitive conclusions from our patient’s complex clinical course, this case suggests that subretinal delivery of triamcinolone acetonide may be more effective than preretinal steroid administration in reducing foveal cysts, particularly in retinitis pigmentosa. In this case, the duration of triamcinolone acetonide in the subretinal space seemed to be on the order of 3 months. Why the nasally located subretinal bolus of triamcinolone acetonide had such a significant effect on the macula is not entirely clear. At clinical examination, particles of triamcinolone acetonide were visible inferior to the macula. It is possible that the inferior retinal detachment allowed triamcinolone acetonide to be widely distributed in the subretinal space, which consequently may have had a widespread effect on the retina. Novel drug delivery has been studied in humans and animal models.3–5 Subretinal steroid injection has been shown to be safe in rabbits.3 One pilot study used subretinal triamcinolone administration as an adjunct to vitrectomy for age-related macular degeneration; the results showed moderate visual improvement compared with controls.4 Ocular hypertension was the primary complication that was controlled with medical management. In addition, drug delivery by cannu-
49
TRIAMCINOLONE ACETONIDE AND CME IN RP
lation of the suprachoroidal space has been studied in animal models.5 Further studies are needed to evaluate the potential side effects and/or retinal toxicity associated with subretinal triamcinolone treatment of retinal disease. It is unclear whether subretinal triamcinolone has an effect on visual acuity, but our patient did report a subjective improvement in vision after resolution of foveal cysts. Further, the potential untoward consequences for visual function of detaching the retina temporal to the macula or in the paramacular region in a patient with retinitis pigmentosa by the subretinal injection of a steroid need to be considered. Key words: subretinal space, triamcinolone acetonide, retinitis pigmentosa, cystoid macular edema.
References 1.
2.
3.
4.
5.
Fishman GA, Gilbert LD, Fiscella RG, et al. Acetazolamide for treatment of chronic macular edema in retinitis pigmentosa. Arch Ophthalmol 1989;107:1445–1452. Ke JE. Intravitreal triamcinolone acetonide for treatment of cystoid macular edema associated with retinitis pigmentosa. Retina 2006;26:1094–1096. Kozak I, Cheng L, Mendez T, et al. Evaluation of the toxicity of subretinal triamcinolone acetonide in the rabbit. Retina 2006;26:811–817. Kertes PJ, Coupland SG. The use of subretinal triamcinolone acetonide in the management of neovascular age-related macular degeneration: a pilot study. Can J Ophthalmol 2005;40: 573–584. Olsen TW, Feng X, Wabner K, et al. Cannulation of the suprachoroidal space: a novel drug delivery methodology to the posterior segment. Am J Ophthalmol 2006;142:777–787.
