Correspondence incurrence of subcutaneous tissue adhesion at the resection site, leading to other cosmetic problems. In conclusion, the AFI for facial augmentation widely performed as a convenient and safe cosmetic procedure at present can cause unexpected complications such as inflammation and lipogranuloma formation at the periorbital area. Therefore, it requires caution, and sufficient explanation should be given to the patient before the surgery. Yi-Ryeung Park, Jin A. Choi, Tae-Yoon La St. Vincent’s Hospital, College of Medicine, The Catholic University of Korea, Suwon, Korea Correspondence to: Tae-Yoon La, MD: [email protected] REFERENCES
3. Thaunat O, Thaler F, Loirat P, et al. Cerebral fat embolism induced by facial fat injection. Plast Reconstr Surg. 2004;113:2235-6. 4. Park TH, Seo SW, Kim JK, Chang CH. Clinical experience with hyaluronic acid-filler complications. J Plast Reconstr Aesthetic Surg. 2011;64:892-6. 5. Cheng NX, Xu SL, Deng H, et al. Migration of implants: a problem with injectable polyacrylamide gel in aesthetic plastic surgery. Aesthetic Plast Surg. 2006;30:215-25. 6. Cho N, Kim EK, Oh KK, et al. Migrated foreign body granulomas on mammography after injection in the cervicofacial area. Clin Radiol. 2004;59:835-40. 7. Coleman SR. Structural fat graft: the ideal filler? Clin Plast Surg. 2001;28:111-9. 8. Wolter TP, von Heimberg D, Stoffels I, et al. Cryopreservation of mature human adipocytes: in vitro measurement of viability Ann Plast Surg. 2005;55:408-13. 9. Son DG, Oh JH, Choi TH, et al. Viability of fat cells over time after syringe suction lipectomy: the effects of cryopreservation. Ann Plast Surg. 2010;65:354-60. 10. Sa HS, Woo KI, Suh YL, Kim YD. Periorbital lipogranuloma: a previously unknown complication of autologous fat injections for facial augmentation. Br J Opthalmol. 2011;95:1259-63.
1. Kim SM, Kim YS, Hong JW, et al. An analysis of the experiences of 62 patients with moderate complications after full-face fat injection for augmentation. Plast Reconstr Surg. 2012;129:1359-68. 2. Park SH, Sun HJ, Choi KS. Sudden unilateral visual loss after autologous fat injection into the nasolabial fold. Clin Ophthalmol. 2008;2:679-83.
Monoclonal gammopathy of undetermined significance maculopathy Monoclonal gammopathy of unknown significance (MGUS), a premalignant condition with relative hyperviscosity, has few reported ophthalmologic findings (based on a MEDLINE search). In this report, we present a young female with documented MGUS and with unusual ophthalmic manifestations. A 37-year-old Afro-Caribbean female with no ocular history presented with several weeks of photophobia and bilateral vision loss. Her medical history was significant for 3 cerebrovascular accidents (CVAs) in the past, which left her with no residual deficit. Systemic evaluation for the CVAs had never been performed in her country. The remainder of her medical history and review of systems were noncontributory. On examination, her best corrected visual acuity was 20/40 OD and 20/60 OS. Slit-lamp examination revealed 2þ cell and flare. Applanation tensions were within normal limits. Dilated examination revealed 2þ vitritis, macular elevation (Fig. 1A, 1B), and the absence of vasculitis or shifting fluid. Spectral-domain optical coherence tomography confirmed bilateral serous macular detachments (Fig. 1C, 1D). Fluorescein angiography confirmed the absence of vasculitis, the absence of any choroidal leakage, and demonstrated late staining of the optic nerve (Fig. 2A, 2B). Magnetic resonance imaging (MRI) of the brain and orbit were performed with and without gadolinium, and revealed mild nonspecific diffuse smooth thickening and enhancement of the choroid in the
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globes bilaterally. No mass lesion is noted in the orbits. The retrobulbar fat appeared unremarkable bilaterally. There were multiple old cortical infarcts in different vascular distributions, with focal volume loss and encephalomalacia. A comprehensive systemic evaluation was initiated and included evaluation for autoimmune diseases, infectious entities, and sarcoidosis. Serum protein electrophoresis (SPEP) was performed and was abnormal (albumin, 3.47 g/dL; α-1 globulin, 0.23 g/dL; α-2 globulin 0.77 g/dL; γ-globulin, 3.21 g/dL; M-spike, 1.94 g/dL). Bone marrow biopsy was performed to further clarify the abnormal SPEP, and it confirmed the diagnosis of MGUS. Because of the significant iritis, vitritis, and choroidal thickening on MRI, the possibility of ocular lymphoma was considered. A vitreous biopsy was performed, and it revealed a paucicellular inflammatory infiltrate predominantly of macrophages, with the absence of granulomas. There was no staining with Congo red stain or birefringence on polarization, findings consistent with absence of amyloid. Immunohistochemical evaluation of the cell block confirmed the presence of CD68þ macrophages and was negative for CD20 and CD3 immunostains for B and T lymphocytes. There was no evidence of malignancy. Therapy was initiated with oral prednisone at a dosage of 1 mg/kg, complimented by topical Pred Forte 1% therapy every hour bilaterally. A rapid improvement of the iritis, vitritis, and serous macular detachments ensued, with a full return of visual acuity (Fig. 3A, 3B). However, despite a slow, lengthy taper of the oral and topical prednisone, there was a recurrence of inflammation and the serous
CAN J OPHTHALMOL — VOL. 48, NO. 6, DECEMBER 2013
Correspondence
Fig. 1 — A and B, Fundus photography. C and D, Optical coherence tomography imaging demonstrating neurosensory detachment before treatment.
macular elevation. Oral prednisone at 1 mg/kg and topical prednisone therapy were reinitiated, with a rapid and complete resolution of the inflammation and maculopathy. Because of steroid toxicity, rituximab therapy was initiated and maintained, allowing for complete withdrawal of the systemic and topical prednisone. The patient has remained free of systemic malignancy and recurrence of her ocular findings for more than 9 months. MGUS is a premalignant plasma cell dyscrasia, which primarily affects a geriatric population. The prognosis for MGUS in the geriatric population is variable, as there is a risk for progression to multiple myeloma or other lymphoproliferative disorders of 1% per year.1 The occurrence of MGUS in people younger than 40 years is rare, so much so that exact data regarding the incidence and prognosis in this age group are lacking.2 Our patient’s youth at presentation and constellation of physical findings suggest that the disease in this age group, and hence this patient, may manifest differently,
thus explaining why similar findings have not been previously reported. The reported ocular manifestations of MGUS include corneal stromal deposits and copper deposition in the Descemet membrane (MEDLINE search). Neither bilateral exudative maculopathy nor uveitis in association with MGUS has been previously reported (MEDLINE search). This is in contrast with the well-documented retinal manifestations of patients with polyclonal gammopathies.3 Our comprehensive systemic evaluation and fluorescein angiographic findings ruled out the classic autoimmune conditions, syphilis, tuberculosis, sarcoidosis, lymphoma, central serous, and malignant hypertension. VogtKoyanagi-Harada disease is unlikely given the absence of poliosis, cutaneous manifestations, or neurologic symptoms, and its association with MGUS has never been reported. Acute exudative polymorphous vitelliform maculopathy is equally unlikely given the absence of vitelliformtype lesions4 and its lack of association with uveitis. We
CAN J OPHTHALMOL — VOL. 48, NO. 6, DECEMBER 2013
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Correspondence
Fig. 2 — Fluorescein angiography.
Fig. 3 — A and B, OD and OS optical coherence tomography imaging demonstrating resolution of detachments with steroid treatment.
therefore conclude that the findings we describe are as a result of MGUS. We hypothesize that the circulating paraproteins that are present in this condition as a result of MGUS may act as autoantibodies against Z1 retinal structures, such as retinal antigens or the retinal pigment epithelium,5 thus inciting an inflammatory reaction, leading to steroidresponsive uveitis and exudative maculopathy. Our case illustrates that early and aggressive use of steroids or other immune-modulating agents may be required in young patients with MGUS who manifest similar findings. Norman Saffra, Aleksandr Rakhamimov, William B. Solomon, Joshua Scheers-Masters
REFERENCES 1. Kyle RA, Therneau TM, Rajkumar SV, et al. A long-term study of prognosis in monoclonal gammopathy of undetermined significance. N Engl J Med. 2002;346:564-9. 2. Wadhera RK, Rajkumar SV. Prevalence of monoclonal gammopathy of undetermined significance: a systematic review. Mayo Clin Proc. 2010;85:933-42. 3. Ho AC, Benson WE, Wong J. Unusual immunogammopathy maculopathy. Ophthalmology. 2000;107:1099-103. 4. Chan CK, Gass JD, Lin SG. Acute exudative polymorphous vitelliform maculopathy syndrome. Retina. 2003;23:453-62. 5. Koreen L, He SX, Johnson MW, Hackel RE, Khan NW, Heckenlively JR. Anti-retinal pigment epithelium antibodies in acute exudative polymorphous vitelliform maculopathy. Arch Ophthalmol. 2011;129:23-9.
Maimonides Medical Center, Brooklyn, NY Correspondence to: Aleksandr Rakhamimov, MD: Aleksandr.Rakhamimov@ gmail.com
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Can J Ophthalmol 2013;48:e168–e170 0008-4182/13/$-see front matter & 2013 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2013.07.018
3. Thaunat O, Thaler F, Loirat P, et al. Cerebral fat embolism induced by facial fat injection. Plast Reconstr Surg. 2004;113:2235-6. 4. Park TH, Seo SW, Kim JK, Chang CH. Clinical experience with hyaluronic acid-filler complications. J Plast Reconstr Aesthetic Surg. 2011;64:892-6. 5. Cheng NX, Xu SL, Deng H, et al. Migration of implants: a problem with injectable polyacrylamide gel in aesthetic plastic surgery. Aesthetic Plast Surg. 2006;30:215-25. 6. Cho N, Kim EK, Oh KK, et al. Migrated foreign body granulomas on mammography after injection in the cervicofacial area. Clin Radiol. 2004;59:835-40. 7. Coleman SR. Structural fat graft: the ideal filler? Clin Plast Surg. 2001;28:111-9. 8. Wolter TP, von Heimberg D, Stoffels I, et al. Cryopreservation of mature human adipocytes: in vitro measurement of viability Ann Plast Surg. 2005;55:408-13. 9. Son DG, Oh JH, Choi TH, et al. Viability of fat cells over time after syringe suction lipectomy: the effects of cryopreservation. Ann Plast Surg. 2010;65:354-60. 10. Sa HS, Woo KI, Suh YL, Kim YD. Periorbital lipogranuloma: a previously unknown complication of autologous fat injections for facial augmentation. Br J Opthalmol. 2011;95:1259-63.
1. Kim SM, Kim YS, Hong JW, et al. An analysis of the experiences of 62 patients with moderate complications after full-face fat injection for augmentation. Plast Reconstr Surg. 2012;129:1359-68. 2. Park SH, Sun HJ, Choi KS. Sudden unilateral visual loss after autologous fat injection into the nasolabial fold. Clin Ophthalmol. 2008;2:679-83.
Monoclonal gammopathy of undetermined significance maculopathy Monoclonal gammopathy of unknown significance (MGUS), a premalignant condition with relative hyperviscosity, has few reported ophthalmologic findings (based on a MEDLINE search). In this report, we present a young female with documented MGUS and with unusual ophthalmic manifestations. A 37-year-old Afro-Caribbean female with no ocular history presented with several weeks of photophobia and bilateral vision loss. Her medical history was significant for 3 cerebrovascular accidents (CVAs) in the past, which left her with no residual deficit. Systemic evaluation for the CVAs had never been performed in her country. The remainder of her medical history and review of systems were noncontributory. On examination, her best corrected visual acuity was 20/40 OD and 20/60 OS. Slit-lamp examination revealed 2þ cell and flare. Applanation tensions were within normal limits. Dilated examination revealed 2þ vitritis, macular elevation (Fig. 1A, 1B), and the absence of vasculitis or shifting fluid. Spectral-domain optical coherence tomography confirmed bilateral serous macular detachments (Fig. 1C, 1D). Fluorescein angiography confirmed the absence of vasculitis, the absence of any choroidal leakage, and demonstrated late staining of the optic nerve (Fig. 2A, 2B). Magnetic resonance imaging (MRI) of the brain and orbit were performed with and without gadolinium, and revealed mild nonspecific diffuse smooth thickening and enhancement of the choroid in the
e168
Can J Ophthalmol 2013;48:e166–e168 0008-4182/13/$-see front matter & 2013 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2013.08.002
globes bilaterally. No mass lesion is noted in the orbits. The retrobulbar fat appeared unremarkable bilaterally. There were multiple old cortical infarcts in different vascular distributions, with focal volume loss and encephalomalacia. A comprehensive systemic evaluation was initiated and included evaluation for autoimmune diseases, infectious entities, and sarcoidosis. Serum protein electrophoresis (SPEP) was performed and was abnormal (albumin, 3.47 g/dL; α-1 globulin, 0.23 g/dL; α-2 globulin 0.77 g/dL; γ-globulin, 3.21 g/dL; M-spike, 1.94 g/dL). Bone marrow biopsy was performed to further clarify the abnormal SPEP, and it confirmed the diagnosis of MGUS. Because of the significant iritis, vitritis, and choroidal thickening on MRI, the possibility of ocular lymphoma was considered. A vitreous biopsy was performed, and it revealed a paucicellular inflammatory infiltrate predominantly of macrophages, with the absence of granulomas. There was no staining with Congo red stain or birefringence on polarization, findings consistent with absence of amyloid. Immunohistochemical evaluation of the cell block confirmed the presence of CD68þ macrophages and was negative for CD20 and CD3 immunostains for B and T lymphocytes. There was no evidence of malignancy. Therapy was initiated with oral prednisone at a dosage of 1 mg/kg, complimented by topical Pred Forte 1% therapy every hour bilaterally. A rapid improvement of the iritis, vitritis, and serous macular detachments ensued, with a full return of visual acuity (Fig. 3A, 3B). However, despite a slow, lengthy taper of the oral and topical prednisone, there was a recurrence of inflammation and the serous
CAN J OPHTHALMOL — VOL. 48, NO. 6, DECEMBER 2013
Correspondence
Fig. 1 — A and B, Fundus photography. C and D, Optical coherence tomography imaging demonstrating neurosensory detachment before treatment.
macular elevation. Oral prednisone at 1 mg/kg and topical prednisone therapy were reinitiated, with a rapid and complete resolution of the inflammation and maculopathy. Because of steroid toxicity, rituximab therapy was initiated and maintained, allowing for complete withdrawal of the systemic and topical prednisone. The patient has remained free of systemic malignancy and recurrence of her ocular findings for more than 9 months. MGUS is a premalignant plasma cell dyscrasia, which primarily affects a geriatric population. The prognosis for MGUS in the geriatric population is variable, as there is a risk for progression to multiple myeloma or other lymphoproliferative disorders of 1% per year.1 The occurrence of MGUS in people younger than 40 years is rare, so much so that exact data regarding the incidence and prognosis in this age group are lacking.2 Our patient’s youth at presentation and constellation of physical findings suggest that the disease in this age group, and hence this patient, may manifest differently,
thus explaining why similar findings have not been previously reported. The reported ocular manifestations of MGUS include corneal stromal deposits and copper deposition in the Descemet membrane (MEDLINE search). Neither bilateral exudative maculopathy nor uveitis in association with MGUS has been previously reported (MEDLINE search). This is in contrast with the well-documented retinal manifestations of patients with polyclonal gammopathies.3 Our comprehensive systemic evaluation and fluorescein angiographic findings ruled out the classic autoimmune conditions, syphilis, tuberculosis, sarcoidosis, lymphoma, central serous, and malignant hypertension. VogtKoyanagi-Harada disease is unlikely given the absence of poliosis, cutaneous manifestations, or neurologic symptoms, and its association with MGUS has never been reported. Acute exudative polymorphous vitelliform maculopathy is equally unlikely given the absence of vitelliformtype lesions4 and its lack of association with uveitis. We
CAN J OPHTHALMOL — VOL. 48, NO. 6, DECEMBER 2013
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Correspondence
Fig. 2 — Fluorescein angiography.
Fig. 3 — A and B, OD and OS optical coherence tomography imaging demonstrating resolution of detachments with steroid treatment.
therefore conclude that the findings we describe are as a result of MGUS. We hypothesize that the circulating paraproteins that are present in this condition as a result of MGUS may act as autoantibodies against Z1 retinal structures, such as retinal antigens or the retinal pigment epithelium,5 thus inciting an inflammatory reaction, leading to steroidresponsive uveitis and exudative maculopathy. Our case illustrates that early and aggressive use of steroids or other immune-modulating agents may be required in young patients with MGUS who manifest similar findings. Norman Saffra, Aleksandr Rakhamimov, William B. Solomon, Joshua Scheers-Masters
REFERENCES 1. Kyle RA, Therneau TM, Rajkumar SV, et al. A long-term study of prognosis in monoclonal gammopathy of undetermined significance. N Engl J Med. 2002;346:564-9. 2. Wadhera RK, Rajkumar SV. Prevalence of monoclonal gammopathy of undetermined significance: a systematic review. Mayo Clin Proc. 2010;85:933-42. 3. Ho AC, Benson WE, Wong J. Unusual immunogammopathy maculopathy. Ophthalmology. 2000;107:1099-103. 4. Chan CK, Gass JD, Lin SG. Acute exudative polymorphous vitelliform maculopathy syndrome. Retina. 2003;23:453-62. 5. Koreen L, He SX, Johnson MW, Hackel RE, Khan NW, Heckenlively JR. Anti-retinal pigment epithelium antibodies in acute exudative polymorphous vitelliform maculopathy. Arch Ophthalmol. 2011;129:23-9.
Maimonides Medical Center, Brooklyn, NY Correspondence to: Aleksandr Rakhamimov, MD: Aleksandr.Rakhamimov@ gmail.com
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Can J Ophthalmol 2013;48:e168–e170 0008-4182/13/$-see front matter & 2013 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2013.07.018
