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Show journal of Neuro- Ophthalmology 16( 1): 68- 72, 1996. © 1996 Lippincott- Raven Publishers, Philadelphia Letters to the Editor Human T- lymphotropic Virus Type I ( HTLV- I) To the Editor: Human T- lymphotropic virus type I ( HTLV- I) is a human retrovirus that is highly epidemic in some regions of the world: southern Japan, Central Africa, and the Caribbean islands. In Martinique ( French West Indies), the seroprevalence in the general population is - 2%. HTLV- I has been shown to be responsible for two distinct systemic diseases: adult T- cell leukemia ( ATL) and tropical spastic paraparesis or HTLV- I- associated myelopathy ( TSP/ HAM). During a 9- month period in 1992 through 1993, a prospective study found eight cases of uveitis in a consecutive series of 50 patients with TSP/ HAM. This is the first ophthalmological study performed outside the Japanese area. Our study included 50 patients ( 32 women, 18 men), all black people born in Martinique, with a mean age of 60.5 years ( 31 to 92 years). All of them were systematically referred from the Department of Neurology to the Department of Ophthalmology of Fort de France University Hospital. They all had HTLV- I antibodies assessed by two methods: enzyme- linked immunosorbent assay ( ELISA) ( Sanofi Pasteur diagnosis) and Western blot ( Abbott laboratories). Of 50 patients, we observed eight patients ( 16%) with uveitis: two cases of uveitis optic- disc neuritis with loss of visual acuity, abnormal visual field testing, and dyschromatopsia ( cases 1 and 2), two cases of isolated anterior uveitis ( cases 5 and 8), one case of uveitis associated with a hyalitis ( case 6), one case of uveitis associated with hyalitis and papillary hyperhemia ( case 3), and two cases of hyalitis associated with papillary hyperhemia ( cases 4 and 7). Table 1 summarizes their characteristics. The patients with uveitis were younger ( mean age; 46.6 years) than the patients without uveitis ( mean age; 63.1 years). In five cases, the uveitis developed in both eyes. Examination showed neither lens iris synechia, pars plana exudative deposits, nor peripheral or mottled chorioretinitis. The clinical course and the laboratory results were not consistent with the usual etiologies of anterior uveitis and uveitis optic- disc neuritis. They were as follows: hemogram, erythrocyte sedimentation rate, serum proteins electrophoresis, serologies of syphilis, toxoplasmosis, leptospiro-sis, herpes simplex, varicella zoster, cytomegalovirus, human immunodeficiency virus ( HIV), anti-streptolysin- O, angiotensin converting enzyme, analysis of human leukocyte antigen ( HLA), rheumatoid factor, antinuclear antibodies, and skin reaction to tuberculin. When the case is found after routine ophthalmological examination, the loss of visual acuity is low. Five of the eight patients showed a recurrent form of the disease responding well to topical and systemic corticosteroid therapy, which resulted in short- term improvement of the visual acuity. In the first report by Vernant et al. ( 1) in 1986, which established a relationship between TSP/ HAM and HTLV- I, one case of uveitis was mentioned among a total of 22 TSP/ HAM. Since that time, many Japanese authors have reported cases of anterior and posterior uveitis among TSP/ HAM patients. Thus of 17 TSP/ HAM patients, Ohba et al. ( 2) observed isolated vitreous opacity in one case and granulomatous anterior uveitis in two cases. Among 34 TSP/ HAM patients, Nakao et al. ( 3) described three cases of anterior uveitis associated with hyalitis. Moreover, HTLV- I seroprevalence should be significantly higher in patients suffering from etiologically unknown uveitis than in the entire adult population or than in patients suffering from etiologically known uveitis ( 3,4). Many characteristics mentioned in these reports are also present in our patients: every clinical type is observed, there is no lens- iris synechia, the vitreous opacities are very frequent ( 5), and the outcome consists of frequent mild relapses that respond well to corticosteroid therapy. Thus the uveal inflammatory reaction should argue in favor of the hypothesis of a disorder of the autoimmune mechanism in the TSP/ HAM. In endemic areas, HTLV- I infection may be a significant cause of uveitis, sometimes associated with optic- disc neuritis, whether or not a TSP/ HAM- associated feature. Harold Merle, M. D. * Didier Smadja, M. D. * Jean- Claude Vernant, M. D. Departments of Ophthalmology and ^ Neurology Central Hospitalier Universitaire de Fort de France, BP 632 97261 Fort de France Martinique, French West Indies 68 LETTERS TO THE EDITOR 69 TABLE 1. Clinical characteristics of eight cases of uveitis observed among a population of 50 TSP/ HAM in Martinique Case no. 1 2 3 4 5 6 7 8 Age 43 47 42 36 50 52 49 54 Sex F F F F F M F F Anterior uveitis R Yes Yes - - Yes Yes - Yes L Yes Yes Yes - Yes Yes - Yes Vitreous opacity R L Yes Yes - Yes - Yes - - Yes Yes Yes - - Yes Yes - Hyperhemia of optic R Yes - - Yes - - - - disk L Yes Yes Yes - - - Yes - Evolution Recurrent form since 5 years old Recurrent form since 15 years old Recurrent form since 1 year old Routine examination Routine examination Recurrent form since 1 year old Routine examination Recurrent form since 1 year old TSP/ HAM, tropical spastic paraparesis/ human T- lymphotropic virus type l- associated myelopathy. REFERENCES Vernant JC, Gessain A, Gout O, et al. Parapareses spas-tiques tropicales en Martinique: haute prevalence d'anti-corps HTLV- I. Presse Med 1986; 15: 419- 22. Ohba N, Matsumoto M, Sameshima M, et al. Ocular manifestations in patients infected with human T- lymphotropic virus type I. Jpn J Ophthalmol 1989; 33: 1- 12. Nakao K, Ohba N, Matsumoto M. Non infectious anterior uveitis in patients infected with human T- lymphotropic virus type I. Jpn J Ophthalmol 1989; 33: 472- 81. Mochizuki M, Watanabe T, Yamaguchi K, et al. Uveitis associated with human T- cell lymphotropic virus type I. Am J Ophthalmol 1992; 114: 123- 9. Yoshimura K, Mochizuki M, Araki S, et al. Clinical and immunologic features of human T- cell lymphotropic virus type- I uveitis. Am J Ophthalmol 1993; 116: 156- 63. Comment on " Isolated Trochlear Nerve Palsy Secondary to Dural Carotid- Cavernous Sinus Fistula" by Aki K. Selky and Valerie A. Purvin ( JNO 14( 1): 52- 54, 1994) Dear Editors: I read with great interest and pleasure the report of Drs. Selky and Purvin. It recalled a case of mine ( unfortunately not reported in the printed medical literature). I first examined the patient, a 60- year-old woman on March 23, 1970. She complained of a 5- week history of double vision, worse when looking down and to her left, as well as a pounding headache. Examination revealed mild redness of the conjunctiva, gross paresis of the right superior oblique muscle with 10 prism diopter ( p. d.) of right hypertropia on forward gaze, increasing to 17 p. d. on right head tilt. Among my initial diagnostic impressions were diabetic mononeuropathy, temporal arteritis, myasthenia gravis, episcleritis, and dysthyroid ophthalmopathy. Initially there was no exophthalmos or asymmetry in intraocular pressures. Five months later, the diplopia had resolved, but 8 months later right exophthalmos developed with maximum relative proptosis of 5 mm. At 11 months after onset, intraocular pressure had risen to 25 mm O. D. ( 15 mm O. S.). A B- scan performed by Dr. Jackson Coleman 12 months after onset revealed an abnormal optic nerve pattern consistent with " vascular anomaly" such as " carotid- cavernous sinus fistula." An orbital venogram 16 months after onset of symptoms showed obstruction of the superior ophthalmic vein on the right, with a large collateral proceeding laterally and posteriorly. The left superior ophthalmic vein was normal. A bilateral carotid arteriogram done 20 months after onset revealed a right dural carotid- cavernous fistula. Feeding vessels were from small meningeal branches of the right internal carotid system and by ascending pharyngeal, nasopharyngeal, and middle meningeal branches of the right external carotid artery. Venous drainage was shown to be exclusively on the right side, anteriorly, through the superior ophthalmic vein and facial vein. Clinical course: 6 months after the arteriogram, the proptosis, redness, and asymmetric pressures resolved. Over the next 22 years, a relentless drop of acuity secondary to cataract occurred, along with recurrence of redness, and dilation of fundus veins. At no time during the entire 24- year course has there ever been objective or subjective bruits. At the time of diagnosis ( 1971), the only therapeutic option offered her by Dr. Charles Wilson was electrothrombosis of the right cavernous sinus, which the patient respectfully declined. Although this case obviously would have taken precedence had it been reported, I can hardly fault Drs. Selky and Purvin for not citing it. Congratulations to them! Richard L. Sogg, M. D. Clinical Professor of Neuro- Ophthalmology / Neuro- Ophthalmol, Vol. 16, No. 1, 1996 |