- Email: [email protected]
Central serous chorioretinopathy after bone marrow transplantation
founded this case, as seen in similar cases of Purtscher-like retinopathy associated with collagen vascular disease. A complement-mediated microemboli theory of pathogenesis has been proposed in both Purtscher retinopathy5 and mixed cryoglobulinemia1 with leukoemboli and immune complexes, respectively, causing vaso-occlusion. This case of Purtscher-like retinopathy in a young man with hepatitis C–associated cryoglobulinemia supports such a proposed pathogenic sequence. Immunosuppressive therapy may ameliorate such a complement-mediated pathway and is supported by the therapeutic response in 6 weeks. Clinicians should consider cryoglobulinemia in the differential diagnosis of a patient with Purtscher-like retinopathy and history of hepatitis C.
4 months after bone marrow transplantation for acute myelogenous leukemia. Other co-existing medical problems at the time of presentation included systemic hypertension and graft-versus-host-disease (GVHD), for which the patient was using both systemic corticosteroids and cyclosporine. CONCLUSION: Central serous chorioretinopathy is a rare cause of vision loss in patients after bone marrow transplantation. Previous descriptions of bone marrow transplantation-associated central serous chorioretinopathy in patients with thrombotic microangiopathy, as well as the occurrence of both systemic hypertension and the use of systemic corticosteroids and cyclosporine in our patient with bone marrow transplantation-associated central serous chorioretinopathy, support theories of choroidal vascular compromise in the pathogenesis of central serous chorioretinopathy. (Am J Ophthalmol 2001; 131:804 – 805. © 2001 by Elsevier Science Inc. All rights reserved.)
REFERENCES
1. Agnello V, Chung RT, Kaplan LM. A role for hepatitis C virus infection in type II cryoglobulinemia. N Engl J Med 1992;327:1490 –1495. 2. Gass JDM. Macular dysfunction caused by retinal vascular diseases. Traumatic retinopathy. In: Craven L, Cox K, editors. Stereoscopic atlas of macular diseases: diagnosis and treatment. St. Louis, MO: Mosby-Year Book, 1997:452– 454, 746 –747. 3. Cohen SM, Kokame GT, Gass JD. Paraproteinemias associated with serous detachments of the retinal pigment epithelium and neurosensory retina. Retina 1996;16:467– 473. 4. Case records of Massachusetts General Hospital. Weekly clinicopathological exercises. Case 13-1994. A 62-year-old man with epistaxis, confusion, renal failure, and bilateral central retinal-vein thrombosis. N Engl J Med 1994;330:920 – 927. 5. Jacob HS, Craddock PR, Hammerschmidt DE, Moldow CF. Complement-induced granulocyte aggregation: an unsuspected mechanism of disease. N Engl J Med 1980;302:789 – 794.
O
Central Serous Chorioretinopathy After Bone Marrow Transplantation Amani A. Fawzi, MD, and Emmett T. Cunningham, Jr, MD, PhD, MPH PURPOSE:
To describe central serous chorioretinopathy after bone marrow transplantation. METHODS: The medical records of the patient were reviewed retrospectively. RESULTS: A 46-year-old Filipino man developed multifocal central serous chorioretinopathy affecting his left eye Accepted for publication Nov 22, 2000. From the Francis I. Proctor Foundation and The Department of Ophthalmology, University of California at San Francisco, School of Medicine, San Francisco, California. Supported by a Career Development Award from Research to Prevent Blindness, Inc., New York (Dr. Emmett T. Cunningham, Jr). Inquiries to Emmett T. Cunningham, Jr, MD, PhD, MPH, The Pearl & Samuel J. Kimura Ocular Immunology Laboratory, The Francis I. Proctor Foundation, UCSF, School of Medicine, San Francisco, CA 94143-0944; fax: (415) 502-2521; e-mail: [email protected]
804
AMERICAN JOURNAL
CULAR COMPLICATIONS OF BONE MARROW TRANS-
plantation are common.1,2 Anterior segment complications of bone marrow transplantation occur most often and include keratoconjunctivitis sicca, conjunctival graftversus-host-disease, cataract, and corneal infections. Posterior segment complications may also follow bone marrow transplantation and include retinal microvasculopathy, optic disk edema resulting either from increased intracranial pressure or from cyclosporine toxicity, fungal retinitis or endophthalmitis, and cytomegalovirus retinitis.3 Central serous chorioretinopathy, a well-recognized complication of solid organ transplantation,4 is rare in the setting of bone marrow transplantation.3,5,6 A 46-year-old Filipino man presented with mildly decreased vision in the left eye for 1 week. Ocular history was unremarkable. Medical history was notable for acute myelogenous leukemia diagnosed 1 year before presentation, for which the patient underwent allogenic bone marrow transplant 4 months before presentation; for systemic hypertension, which developed 3 months after his bone marrow transplantation; and for graft-versus-host-disease, which the patient developed 1 month before the onset of his visual symptoms. Current medications included prednisone, 60 mg/day; cyclosporine, 400 mg/day; azathioprine, 75 mg/day; nifedipine XL 120mg/day; and acyclovir, 200 mg three times daily. On examination, best-corrected visual acuity was 20/20 in both eyes. No afferent pupillary defect existed. Intraocular pressure was RE: 20 and LE: 18. Slit-lamp examination revealed no evidence of anterior chamber or vitreous inflammation in either eye. The right eye was otherwise unremarkable. Left fundus examination showed three small serous macular detachments superior to the fovea (Figure 1A, arrowheads). Fluorescein angiography revealed multiple retinal pigment epithelium leak points underlying the detachment spaces (Figure 1B–D, arrows). The patient developed
OF
OPHTHALMOLOGY
JUNE 2001
FIGURE 1. Color (A) and serial fluorescein angiographic (B–D) photographs of the left fundus show three small serous macular detachments superior to the fovea (A; arrowheads) with multiple retinal pigment epithelial leak points underlying the detachment spaces (B–D, arrows).
disseminated cytomegalovirus infection and died 1 month later. No autopsy was performed. The pathogenesis of central serous chorioretinopathy is the topic of considerable debate.4 Although some authors have suggested that central serous chorioretinopathy results from an abnormality in the retinal pigment epithelium, many authorities believe that most cases of central serous chorioretinopathy represent dysregulation of the choroidal vasculature. Serous retinal detachment is a rare complication of bone marrow transplantation but has been described,3,5,6 particularly in the setting of thrombotic microvascular angiopathy5,6 of the sort observed in graftversus-host-disease. In addition, central serous chorioretinopathy has been reported in patients after solid organ transplants, where it has been associated with systemic hypertension and with use of corticosteroids and cyclosporine, each of which may contribute to choroidal microvascular compromise.4 Our patient had multiple possible contributing factors when he developed central serous chorioretinopathy, including systemic hypertension, reVOL. 131, NO. 6
cently active graft-versus-host-disease, and use of systemic corticosteroids and cyclosporine. REFERENCES
1. Suh DW, Ruttum MS, Stuckenschneider BJ, Mieler WF, Kivlin JD. Ocular findings after bone marrow transplantation in a pediatric population. Ophthalmology 1999;106:1564 –1570. 2. Ng JS, Lam DS, Li CK, et al. Ocular complications of pediatric bone marrow transplantation. Ophthalmology 1999;106:160–164. 3. Coskuncan NM, Jabs DA, Dunn JP, et al. The eye in bone marrow transplantation. VI. Retinal complications. Arch Ophthalmol 1994;112:372–379. 4. Cunningham, Jr ET, Alfred PR, Irvine AR. Central serous chorioretinopathy in patients with systemic lupus erythematosus. Ophthalmology 1996;103(12):2081–2090. 5. Nakai K, Tajima K, Kishimoto Y, et al. Bone marrow transplantation-associated thrombotic microangiopathy manifested by visual disturbance. Rinsho Ketsueki. Jpn J Clin Hematol 2000;41:25–31. 6. Binaghi M, Sterkers M, Vernant JP, Coscas G. Thrombotic microangiopathy and its ocular manifestations: an unusual complication of bone marrow graft. J Francais D Ophthalmol 1983;6:881– 888.
BRIEF REPORTS
805
4 months after bone marrow transplantation for acute myelogenous leukemia. Other co-existing medical problems at the time of presentation included systemic hypertension and graft-versus-host-disease (GVHD), for which the patient was using both systemic corticosteroids and cyclosporine. CONCLUSION: Central serous chorioretinopathy is a rare cause of vision loss in patients after bone marrow transplantation. Previous descriptions of bone marrow transplantation-associated central serous chorioretinopathy in patients with thrombotic microangiopathy, as well as the occurrence of both systemic hypertension and the use of systemic corticosteroids and cyclosporine in our patient with bone marrow transplantation-associated central serous chorioretinopathy, support theories of choroidal vascular compromise in the pathogenesis of central serous chorioretinopathy. (Am J Ophthalmol 2001; 131:804 – 805. © 2001 by Elsevier Science Inc. All rights reserved.)
REFERENCES
1. Agnello V, Chung RT, Kaplan LM. A role for hepatitis C virus infection in type II cryoglobulinemia. N Engl J Med 1992;327:1490 –1495. 2. Gass JDM. Macular dysfunction caused by retinal vascular diseases. Traumatic retinopathy. In: Craven L, Cox K, editors. Stereoscopic atlas of macular diseases: diagnosis and treatment. St. Louis, MO: Mosby-Year Book, 1997:452– 454, 746 –747. 3. Cohen SM, Kokame GT, Gass JD. Paraproteinemias associated with serous detachments of the retinal pigment epithelium and neurosensory retina. Retina 1996;16:467– 473. 4. Case records of Massachusetts General Hospital. Weekly clinicopathological exercises. Case 13-1994. A 62-year-old man with epistaxis, confusion, renal failure, and bilateral central retinal-vein thrombosis. N Engl J Med 1994;330:920 – 927. 5. Jacob HS, Craddock PR, Hammerschmidt DE, Moldow CF. Complement-induced granulocyte aggregation: an unsuspected mechanism of disease. N Engl J Med 1980;302:789 – 794.
O
Central Serous Chorioretinopathy After Bone Marrow Transplantation Amani A. Fawzi, MD, and Emmett T. Cunningham, Jr, MD, PhD, MPH PURPOSE:
To describe central serous chorioretinopathy after bone marrow transplantation. METHODS: The medical records of the patient were reviewed retrospectively. RESULTS: A 46-year-old Filipino man developed multifocal central serous chorioretinopathy affecting his left eye Accepted for publication Nov 22, 2000. From the Francis I. Proctor Foundation and The Department of Ophthalmology, University of California at San Francisco, School of Medicine, San Francisco, California. Supported by a Career Development Award from Research to Prevent Blindness, Inc., New York (Dr. Emmett T. Cunningham, Jr). Inquiries to Emmett T. Cunningham, Jr, MD, PhD, MPH, The Pearl & Samuel J. Kimura Ocular Immunology Laboratory, The Francis I. Proctor Foundation, UCSF, School of Medicine, San Francisco, CA 94143-0944; fax: (415) 502-2521; e-mail: [email protected]
804
AMERICAN JOURNAL
CULAR COMPLICATIONS OF BONE MARROW TRANS-
plantation are common.1,2 Anterior segment complications of bone marrow transplantation occur most often and include keratoconjunctivitis sicca, conjunctival graftversus-host-disease, cataract, and corneal infections. Posterior segment complications may also follow bone marrow transplantation and include retinal microvasculopathy, optic disk edema resulting either from increased intracranial pressure or from cyclosporine toxicity, fungal retinitis or endophthalmitis, and cytomegalovirus retinitis.3 Central serous chorioretinopathy, a well-recognized complication of solid organ transplantation,4 is rare in the setting of bone marrow transplantation.3,5,6 A 46-year-old Filipino man presented with mildly decreased vision in the left eye for 1 week. Ocular history was unremarkable. Medical history was notable for acute myelogenous leukemia diagnosed 1 year before presentation, for which the patient underwent allogenic bone marrow transplant 4 months before presentation; for systemic hypertension, which developed 3 months after his bone marrow transplantation; and for graft-versus-host-disease, which the patient developed 1 month before the onset of his visual symptoms. Current medications included prednisone, 60 mg/day; cyclosporine, 400 mg/day; azathioprine, 75 mg/day; nifedipine XL 120mg/day; and acyclovir, 200 mg three times daily. On examination, best-corrected visual acuity was 20/20 in both eyes. No afferent pupillary defect existed. Intraocular pressure was RE: 20 and LE: 18. Slit-lamp examination revealed no evidence of anterior chamber or vitreous inflammation in either eye. The right eye was otherwise unremarkable. Left fundus examination showed three small serous macular detachments superior to the fovea (Figure 1A, arrowheads). Fluorescein angiography revealed multiple retinal pigment epithelium leak points underlying the detachment spaces (Figure 1B–D, arrows). The patient developed
OF
OPHTHALMOLOGY
JUNE 2001
FIGURE 1. Color (A) and serial fluorescein angiographic (B–D) photographs of the left fundus show three small serous macular detachments superior to the fovea (A; arrowheads) with multiple retinal pigment epithelial leak points underlying the detachment spaces (B–D, arrows).
disseminated cytomegalovirus infection and died 1 month later. No autopsy was performed. The pathogenesis of central serous chorioretinopathy is the topic of considerable debate.4 Although some authors have suggested that central serous chorioretinopathy results from an abnormality in the retinal pigment epithelium, many authorities believe that most cases of central serous chorioretinopathy represent dysregulation of the choroidal vasculature. Serous retinal detachment is a rare complication of bone marrow transplantation but has been described,3,5,6 particularly in the setting of thrombotic microvascular angiopathy5,6 of the sort observed in graftversus-host-disease. In addition, central serous chorioretinopathy has been reported in patients after solid organ transplants, where it has been associated with systemic hypertension and with use of corticosteroids and cyclosporine, each of which may contribute to choroidal microvascular compromise.4 Our patient had multiple possible contributing factors when he developed central serous chorioretinopathy, including systemic hypertension, reVOL. 131, NO. 6
cently active graft-versus-host-disease, and use of systemic corticosteroids and cyclosporine. REFERENCES
1. Suh DW, Ruttum MS, Stuckenschneider BJ, Mieler WF, Kivlin JD. Ocular findings after bone marrow transplantation in a pediatric population. Ophthalmology 1999;106:1564 –1570. 2. Ng JS, Lam DS, Li CK, et al. Ocular complications of pediatric bone marrow transplantation. Ophthalmology 1999;106:160–164. 3. Coskuncan NM, Jabs DA, Dunn JP, et al. The eye in bone marrow transplantation. VI. Retinal complications. Arch Ophthalmol 1994;112:372–379. 4. Cunningham, Jr ET, Alfred PR, Irvine AR. Central serous chorioretinopathy in patients with systemic lupus erythematosus. Ophthalmology 1996;103(12):2081–2090. 5. Nakai K, Tajima K, Kishimoto Y, et al. Bone marrow transplantation-associated thrombotic microangiopathy manifested by visual disturbance. Rinsho Ketsueki. Jpn J Clin Hematol 2000;41:25–31. 6. Binaghi M, Sterkers M, Vernant JP, Coscas G. Thrombotic microangiopathy and its ocular manifestations: an unusual complication of bone marrow graft. J Francais D Ophthalmol 1983;6:881– 888.
BRIEF REPORTS
805