HTLV-I Associated uveitis, myelopathy, rheumatoid arthritis and Sjögren's syndrome

Pinheiro, Sônia Regina A. A.; Lana-Peixoto, Marco Aurélio; Proietti, Anna B. Carneiro; Oréfice, Fernando; Lima-Martins, Maria V.; Proietti, Fernando A.

doi:10.1590/S0004-282X1995000500011

Abstracts

A 62 year-old white female presented with a 10-year-history of slowly progressive spastic paraparesis, pain and dysesthesia in the lower limbs and sphincter disturbance. A few years after the onset of the neurologic symptoms she developped migratory arthritis with swelling of the knees and pain on palpation of knees and fingers, dry eyes, mouth and skin. Two months before admission she presented bilateral nongranulomatous anterior uveitis. Examination revealed spastic paraparesis with bilateral Babinski sign, a decreased sensation level below L3, decreased vibration sense in the lower extremities, and a postural tremor of the upper limbs. Laboratory work-up disclosed HTLV-I positive tests in the blood and cerebrospinal fluid (CSF), and a mild pleocytosis in the CSF with a normal protein content. Nerve conduction velocity studies were normal. The present case shows the association of uveitis, arthritis and Sjögren's syndrome in a patient with tropical spastic paraparesis / human T-cell lymphotropic virus type I (HTLV-I) associated myelopathy (TSP/HAM), and illustrates the wide spectrum of clinical manifestations which may accompany this infection with this virus.

HTLV-I/II; uveitis; myelopathy; spastic paraparesis; Sjögren's syndrome

Uma mulher branca de 62 anos foi internada apresentando história de paraparesia lentamente progressiva durante 10 anos. Dois meses antes da internação ela apresentou uveíte anterior não granulomatosa bilateral. Poucos anos após o início dos sintomas neurológicos, ela desenvolveu artrite migratória com edema dos joelhos e dor a palpação dos joelhos e dedos dos pés, boca, pele e olhos secos. Ao exame físico foi observado paraparesia espástica com sinal de Babinski positivo, sensibilidade diminuída abaixo de L3, diminuição da sensação de vibração nas extremidades inferiores, e tremor postural dos membros superiores. Apresentou testes positivos para o HTLV-I no sangue. O estudo do líquido cefalorraquidiano mostrou discreta pleocitose, proteínas normais e ELISA e Western blot positivos para o HTLV-I. O estudo da velocidade de condução dos nervos foi normal. Este caso mostra a associação de uveíte, artrite e síndrome de Sjogren em uma paciente com paraparesia espástica tropical (HTLV-I) associada a mielopatia (TSP/HAM), e ilustra o largo espectro das manifestações clínicas que podem acompanhar a infecção por este vírus.

HTLV-I/II; uveite; mielopatia; paraparesia espástica; síndrome de Sjogren

HTLV-I Associated uveitis, myelopathy, rheumatoid arthritis and Sjögren's syndrome

Uveite, mielopatia, artrite reumatóide e sindrome de Sjogren associadas ao HTLV-I

Sônia Regina A. A. Pinheiro^I; Marco Aurélio Lana-Peixoto^II; Anna B. Carneiro Proietti^III; Fernando Oréfice^I; Maria V. Lima-Martins^III; Fernando A. Proietti^IV

^IDepartments of Ophthalmology

^IINeurology

^IIIFederal University of Minas Gerais Medical School and State Center of Hematology and Hemotherapy (Fundação Hemominas); Belo Horizonte, MG, Brazil

^IVPreventive and Social Medicine

SUMMARY

A 62 year-old white female presented with a 10-year-history of slowly progressive spastic paraparesis, pain and dysesthesia in the lower limbs and sphincter disturbance. A few years after the onset of the neurologic symptoms she developped migratory arthritis with swelling of the knees and pain on palpation of knees and fingers, dry eyes, mouth and skin. Two months before admission she presented bilateral nongranulomatous anterior uveitis. Examination revealed spastic paraparesis with bilateral Babinski sign, a decreased sensation level below L3, decreased vibration sense in the lower extremities, and a postural tremor of the upper limbs. Laboratory work-up disclosed HTLV-I positive tests in the blood and cerebrospinal fluid (CSF), and a mild pleocytosis in the CSF with a normal protein content. Nerve conduction velocity studies were normal. The present case shows the association of uveitis, arthritis and Sjögren's syndrome in a patient with tropical spastic paraparesis / human T-cell lymphotropic virus type I (HTLV-I) associated myelopathy (TSP/HAM), and illustrates the wide spectrum of clinical manifestations which may accompany this infection with this virus.

Key words:HTLV-I/II, uveitis, myelopathy, spastic paraparesis, Sjögren's syndrome.

RESUMO

Uma mulher branca de 62 anos foi internada apresentando história de paraparesia lentamente progressiva durante 10 anos. Dois meses antes da internação ela apresentou uveíte anterior não granulomatosa bilateral. Poucos anos após o início dos sintomas neurológicos, ela desenvolveu artrite migratória com edema dos joelhos e dor a palpação dos joelhos e dedos dos pés, boca, pele e olhos secos. Ao exame físico foi observado paraparesia espástica com sinal de Babinski positivo, sensibilidade diminuída abaixo de L3, diminuição da sensação de vibração nas extremidades inferiores, e tremor postural dos membros superiores. Apresentou testes positivos para o HTLV-I no sangue. O estudo do líquido cefalorraquidiano mostrou discreta pleocitose, proteínas normais e ELISA e Western blot positivos para o HTLV-I. O estudo da velocidade de condução dos nervos foi normal. Este caso mostra a associação de uveíte, artrite e síndrome de Sjogren em uma paciente com paraparesia espástica tropical (HTLV-I) associada a mielopatia (TSP/HAM), e ilustra o largo espectro das manifestações clínicas que podem acompanhar a infecção por este vírus.

Palavras-chave:HTLV-I/II, uveite, mielopatia, paraparesia espástica, síndrome de Sjogren.

Texto completo disponível apenas em PDF.

Full text available only in PDF format.

Aceite: 27-junho-1995.

Dra. Sônia Regina A. A. Pinheiro - Rua São Paulo 409/601 - 30170-130 Belo Horizonte MG - Brasil. FAX 031 2918526.

1. Bhagavati S, Ehrlich G, Kula RW, Kwok S, Sninsky J, Udani V, Poiesz BJ. Detection of T-cell lymphoma/ leukemia virus type I DNA and antigen in spinal fluid and blood of patients with chronic progresssive myelopathy. N Engl J Med 1988; 318:1141-1147.
2. DeCastro Costa CM, Carton H, Goubau P, Almeida JAC. Brazilian studies on tropical spastic paraparesis: a meta-analysis. Arq Neuropsiquiatr 1994; 52:585-591.
3. Gessain A, Vernant JC, Maurs L, Barin F, Gout O, Calender A, De Thé G. Antibodies to human T-lymphotropic virus type I in patients with tropical spastic paraparesis. Lancet 1985; 2:156-157.
4. Kanazawa H, Ijichi S, Eiraku N, Tadahiko I, Higuchi I, Nakagawa M, Kuriyama M, Tanaka S, Osame M. Behcet's disease and Sjögren syndrome in a patient with HTLV-I-associated myelopathy. J Neurol Sci 1993; 119:121-122.
5. Kira J, Nakamura M, Sawada T, Koyanagi Y, Ohori N, Itoyama Y, Yamamoto M, Sakaki Y, Goto I. Antibody titers to HTLV-I-p40^lax protein and gag-env hybrid protein in HTLV-I associated myelopathy/tropical spastic paraparesis: correlation with HTLV-I proviral DNA load. J Neurol Sci 1992; 107:98-104.
6. Melo A, Gomes I, Mattos K. Mielopatias por HTLV-I na cidade de Salvador, Bahia. Arq Neuropsiquiatr 1994; 52:320-325.
7. Mochizuki M, Watanabe T, Yamaguchi K, Tajima K, Yoshimura K, Nakashima S, Shirao M, Araki S, Miyata N, Mori S, Takatsuki K. Uveitis associated with human T lymphotropic virus type I: seroepidemiologic, clinical and virologic studies. J Infect Dis 1992; 166:943-944.
8. Mochizuki M, Watanabe T, Yamaguchi K, Yosshimura K, Nakashima S, Shirao M, Araki S, Takatsuki K, Mori S, Miyata N. Uveitis associated with human T-cell lymphotropic virus type I. Am J Ophthalmol 1992; 114:123-129.
9. Moreira ED Jr, Ribeiro TT, Swanson P, Sampaio C, Melo A, Brites C, Badaró R, Toedter G, Lee H, Harrington W Jr. Seroepidemiology of human T-cell lymphotropic virus type I /II in Northeastern Brazil. J AIDS 1993; 959-963.
10. Nakao K, Ohba N. Clinical features of HTLV-I associated uveitis. Br J Ophthalmol 1993; 77:374-279.
11. Nakao K, Ohba N, Otsuka S, Okubo A, Yanagita T, Hashimoto N, Arimura H. HTLV-I associated uveitis and hyperthyreoidism. Jpn J Ophthalmol 1994; 38:56-61.
12. Nishioka K, Maruyama I, Sato K, Kitajima I, Nakajima Y, Osame M. Chronic inflammatory arthropathy associated with HTLV-I. Lancet. 1989; 25:441.
13. Ohba N, Matsumoto M, Sameshima M, Kabayama Y, Nakao K, Unoki K, Uehara F, Kawano K, Maruyama I, Osame M. Ocular manifestations in patients infected with human T-lymphotropic virus type I. Jpn J Ophtalmol 1989; 33:1-12.
14. Ohba N, Nakao K Isashiki Y, Osame M, Sonoda S, Yashiki S, Yamaguchi K, Tagima K and The Study Group for HTLV-I Associated Ocular Diseases. A multicenter case-control study of HTLV-I associated uveitis. Jpn J Ophthalmol 1994; 38:162-167.
15. Ohba N, Nakao K, Iashiki Y, Kaminagayoshi T, Sonoda S, Yashiki S, Osame M, and The Study Group for HTLV-I Associated Ocular Diseases. Clinical features of HTLV-I associated uveitis determined in multicenter collaborative study. Jpn J Ophthalmol 1994; 38: 168-174.
16. Osame M, Igata A, Matsumoto M, Usuku A, Kitajima I, Katahashi K. On the discovery of a new clinical entity: humam T-cell Iymphotropic virus type I-associated myelopathy (HAM). Adv Neurol Sci 1987; 31:727-745.
17. Osame M, McArthur JC. Neurologic manifestations of infection with human T-cell Iymphotropic virus type I. In Asbury AK, McKahnn GM, McDonald WI (eds). Diseases of the nervous system: clinical neurobiology. Philadelphia: Saunders, 1992; pl331-1339.
18. Rodgers PEB. The clinical features and aetiology of the neuropathic syndrome in Jamaica. West Ind Med J 1965; 14:36-47.
19. Roman GC, Spencer PS, Schoenberg BS, Hugon J, Ludolph A, Rodgers-Jonhson P, Osuntokun BO, Shamlaye CF. Tropical spastic paraparesis in the Seychelles Islands: a clinical and case-control neuroepidemiologic study. Neurology 1987; 37:1323-1328.
20. Roman GC. Retrovirus-associated myelopathies. Arch Neurol 1987; 44:659-663.
21. Roman GC, Roman LN. Tropical spastic paraparesis: a clinical study of 50 patients from Tumaco (Colombia) and review of the worldwide features of the syndrome. J Neurol Sci 1988; 87:121-138.
22. Roman GC. The neuroepidemiology of tropical spastic paraparesis. Ann Neurol 1988; 23(Suppl): 113-130.
23. Roman GC Tropical spastic paraparesis and HTLV-I myelitis. In Vinken PJ, Bruyn BW, Klawans HL(eds). Handbook of clinical neurology.Amsterdam: Elsevier, vol.56, p 525-542.
24. Sheremata WA, Berger JR, Harrington WJ Jr, Ayyar DR, Stafford M, DeFreitas E. Human T-lymphotropic virus type I-associated myelopathy. A report of ten patients bom in the United States. Arch Neurol 1992; 49:1113-1118.
25. Spina-França A, Livramento JA, Machado LR, Gomes HR, Viana LS, Castro LHM, Nóbrega JPS, Bacheschi LA. HTLV-I antibodies in serum and cerebrospinal fluid in tropical spastic paraparesis in Brazil. Arq Neuropsiquiatr 1990; 48:441-447.
26. Sugimoto M, Nakashima H,Watanabe H, Uyama E, Tanaka F, Ando M, Kawasaki S. T-lymphocyte alveolitis in HTLV-I associated myelopathy. Lancet 1987; 2:1220.
27. Vernant JC, Maurs L, Gout O, Buisson G, Plumelle Y, Neisson-Vemant C, Monplaisir N, Roman GC. HTLV-I associated tropical spastic paraparesis in Martinique: a reappraisal. Ann Neurol 1988; 23(Suppl):133-135
28. Wayne Moore GR, Traugott U, Scheinberg LC, Raine CS. Tropical spastic paraparesis: a model of virus-induced cytotoxic T-cell-mediated demyelination? Ann Neurol 1989; 26:523-530.

Publication Dates

Publication in this collection
08 Dec 2010
Date of issue
Dec 1995

This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

[1] 1. Bhagavati S, Ehrlich G, Kula RW, Kwok S, Sninsky J, Udani V, Poiesz BJ. Detection of T-cell lymphoma/ leukemia virus type I DNA and antigen in spinal fluid and blood of patients with chronic progresssive myelopathy. N Engl J Med 1988; 318:1141-1147.

[2] 2. DeCastro Costa CM, Carton H, Goubau P, Almeida JAC. Brazilian studies on tropical spastic paraparesis: a meta-analysis. Arq Neuropsiquiatr 1994; 52:585-591.

[3] 3. Gessain A, Vernant JC, Maurs L, Barin F, Gout O, Calender A, De Thé G. Antibodies to human T-lymphotropic virus type I in patients with tropical spastic paraparesis. Lancet 1985; 2:156-157.

[4] 4. Kanazawa H, Ijichi S, Eiraku N, Tadahiko I, Higuchi I, Nakagawa M, Kuriyama M, Tanaka S, Osame M. Behcet's disease and Sjögren syndrome in a patient with HTLV-I-associated myelopathy. J Neurol Sci 1993; 119:121-122.

[5] 5. Kira J, Nakamura M, Sawada T, Koyanagi Y, Ohori N, Itoyama Y, Yamamoto M, Sakaki Y, Goto I. Antibody titers to HTLV-I-p40^lax protein and gag-env hybrid protein in HTLV-I associated myelopathy/tropical spastic paraparesis: correlation with HTLV-I proviral DNA load. J Neurol Sci 1992; 107:98-104.

[6] 6. Melo A, Gomes I, Mattos K. Mielopatias por HTLV-I na cidade de Salvador, Bahia. Arq Neuropsiquiatr 1994; 52:320-325.

[7] 7. Mochizuki M, Watanabe T, Yamaguchi K, Tajima K, Yoshimura K, Nakashima S, Shirao M, Araki S, Miyata N, Mori S, Takatsuki K. Uveitis associated with human T lymphotropic virus type I: seroepidemiologic, clinical and virologic studies. J Infect Dis 1992; 166:943-944.

[8] 8. Mochizuki M, Watanabe T, Yamaguchi K, Yosshimura K, Nakashima S, Shirao M, Araki S, Takatsuki K, Mori S, Miyata N. Uveitis associated with human T-cell lymphotropic virus type I. Am J Ophthalmol 1992; 114:123-129.

[9] 9. Moreira ED Jr, Ribeiro TT, Swanson P, Sampaio C, Melo A, Brites C, Badaró R, Toedter G, Lee H, Harrington W Jr. Seroepidemiology of human T-cell lymphotropic virus type I /II in Northeastern Brazil. J AIDS 1993; 959-963.

[10] 10. Nakao K, Ohba N. Clinical features of HTLV-I associated uveitis. Br J Ophthalmol 1993; 77:374-279.

[11] 11. Nakao K, Ohba N, Otsuka S, Okubo A, Yanagita T, Hashimoto N, Arimura H. HTLV-I associated uveitis and hyperthyreoidism. Jpn J Ophthalmol 1994; 38:56-61.

[12] 12. Nishioka K, Maruyama I, Sato K, Kitajima I, Nakajima Y, Osame M. Chronic inflammatory arthropathy associated with HTLV-I. Lancet. 1989; 25:441.

[13] 13. Ohba N, Matsumoto M, Sameshima M, Kabayama Y, Nakao K, Unoki K, Uehara F, Kawano K, Maruyama I, Osame M. Ocular manifestations in patients infected with human T-lymphotropic virus type I. Jpn J Ophtalmol 1989; 33:1-12.

[14] 14. Ohba N, Nakao K Isashiki Y, Osame M, Sonoda S, Yashiki S, Yamaguchi K, Tagima K and The Study Group for HTLV-I Associated Ocular Diseases. A multicenter case-control study of HTLV-I associated uveitis. Jpn J Ophthalmol 1994; 38:162-167.

[15] 15. Ohba N, Nakao K, Iashiki Y, Kaminagayoshi T, Sonoda S, Yashiki S, Osame M, and The Study Group for HTLV-I Associated Ocular Diseases. Clinical features of HTLV-I associated uveitis determined in multicenter collaborative study. Jpn J Ophthalmol 1994; 38: 168-174.

[16] 16. Osame M, Igata A, Matsumoto M, Usuku A, Kitajima I, Katahashi K. On the discovery of a new clinical entity: humam T-cell Iymphotropic virus type I-associated myelopathy (HAM). Adv Neurol Sci 1987; 31:727-745.

[17] 17. Osame M, McArthur JC. Neurologic manifestations of infection with human T-cell Iymphotropic virus type I. In Asbury AK, McKahnn GM, McDonald WI (eds). Diseases of the nervous system: clinical neurobiology. Philadelphia: Saunders, 1992; pl331-1339.

[18] 18. Rodgers PEB. The clinical features and aetiology of the neuropathic syndrome in Jamaica. West Ind Med J 1965; 14:36-47.

[19] 19. Roman GC, Spencer PS, Schoenberg BS, Hugon J, Ludolph A, Rodgers-Jonhson P, Osuntokun BO, Shamlaye CF. Tropical spastic paraparesis in the Seychelles Islands: a clinical and case-control neuroepidemiologic study. Neurology 1987; 37:1323-1328.

[20] 20. Roman GC. Retrovirus-associated myelopathies. Arch Neurol 1987; 44:659-663.

[21] 21. Roman GC, Roman LN. Tropical spastic paraparesis: a clinical study of 50 patients from Tumaco (Colombia) and review of the worldwide features of the syndrome. J Neurol Sci 1988; 87:121-138.

[22] 22. Roman GC. The neuroepidemiology of tropical spastic paraparesis. Ann Neurol 1988; 23(Suppl): 113-130.

[23] 23. Roman GC Tropical spastic paraparesis and HTLV-I myelitis. In Vinken PJ, Bruyn BW, Klawans HL(eds). Handbook of clinical neurology.Amsterdam: Elsevier, vol.56, p 525-542.

[24] 24. Sheremata WA, Berger JR, Harrington WJ Jr, Ayyar DR, Stafford M, DeFreitas E. Human T-lymphotropic virus type I-associated myelopathy. A report of ten patients bom in the United States. Arch Neurol 1992; 49:1113-1118.

[25] 25. Spina-França A, Livramento JA, Machado LR, Gomes HR, Viana LS, Castro LHM, Nóbrega JPS, Bacheschi LA. HTLV-I antibodies in serum and cerebrospinal fluid in tropical spastic paraparesis in Brazil. Arq Neuropsiquiatr 1990; 48:441-447.

[26] 26. Sugimoto M, Nakashima H,Watanabe H, Uyama E, Tanaka F, Ando M, Kawasaki S. T-lymphocyte alveolitis in HTLV-I associated myelopathy. Lancet 1987; 2:1220.

[27] 27. Vernant JC, Maurs L, Gout O, Buisson G, Plumelle Y, Neisson-Vemant C, Monplaisir N, Roman GC. HTLV-I associated tropical spastic paraparesis in Martinique: a reappraisal. Ann Neurol 1988; 23(Suppl):133-135

[28] 28. Wayne Moore GR, Traugott U, Scheinberg LC, Raine CS. Tropical spastic paraparesis: a model of virus-induced cytotoxic T-cell-mediated demyelination? Ann Neurol 1989; 26:523-530.