HTLV-I associated myelopathy in patients from Brazil and Iran: neurological manifestations and cerebrospinal fluid findings

Puccioni-Sohler, Marzia; Kitze, Bernid; Felgenhauer, Klaus

doi:10.1590/S0004-282X1995000200005

Abstracts

We analysed sera and cerebrospinal fluid (CSF) of 20 cases of human T-cell lymphotropic virus type (HTLV-I) associated myelopathy/tropical spastic paraparesis (HAM/TSP) from Brazil and Iran and as controls, 16 Brazilian HTLVI seronegative individuals afflicted with other neurological diseases. It was observed in the HAM/TSP patients that: 1) all had an inflammatory reaction within the central nervous system (CNS); 2) 95% (19/20) showed oligoclonal bands reflecting intrathecal IgG synthesis; 3) 85% (17/20) presented a local synthesis of HTLVI antibodies; 4) 35% (7/20) had a measurable immunoglobulin (Ig) synthesis within the CNS. The CSF parameters of the HAM/TSP were compared with the clinical data (age at onset, duration of disease and disability level). Our data prove that CSF analysis is important for the diagnosis of HAM/TSP. There is no association between the severity of the disease and CSF findings.

HTLV-I; HAM; cerebrospinal fluid; multiple sclerosis

Foram analisadas amostras de líquido cefalorraquidiano (LCR) e plasma de 20 pacientes com mielopatia associada ao HTLV-I (HAM/TSP) provenientes do Brasil e Irã e como controle, 16 de indivíduos brasileiros soronegativos para HTLV-I acometidos por outras doenças neurológicas (esclerose múltipla, epilepsia idiopática e mielopatia de etiologia desconhecida). Observou-se no grupo de pacientes com HAM/TSP: 1) reação inflamatória no SNC em todos os casos; 2) 95% (19/20) mostravam bandas oligoclonais refletindo síntese intratecal de IgG no SNC; 3)85% (7/20) apresentavam síntese local de anticorpos para HTLV-I; 4)35% (tinham síntese mensurável de imunoglobulinas no SNC. Os parâmetros do LCR dos pacientes com HAM/TSP foram comparados aos dados clínicos (idade de início, duração da doença e grau de incapacidade). Os dados apresentados neste estudo indicam a importância da analise do LCR para diagnóstico de HAM/TSP. Entretanto nenhuma associação entre a gravidade da doença e os achados do LCR foi demonstrada.

HTLV-I; mielopatia; HAM; líquido cefalorraquidiano; esclerose múltipla

HTLV-I associated myelopathy in patients from Brazil and Iran: neurological manifestations and cerebrospinal fluid findings

Mielopatia associada a HTLV-I em pacientes do Brasil e do Irã: manifestações neurológicas e achados no líquido cefalorraquidiano

Marzia Puccioni-Sohler^{I, II}; Bernid Kitze^I; Klaus Felgenhauer^I

^IKlinik und Poliklinik für Neurologie der Georg-August-Universitát, Göttingen, Germany

^IIServiço de Neurologia do Hospital Universitário Clementino Fraga Filho da Universidade Federal do Rio de Janeiro (HUCFF/ UFRJ), CNPq/CAPES, Brasil, DAAD

SUMMARY

We analysed sera and cerebrospinal fluid (CSF) of 20 cases of human T-cell lymphotropic virus type (HTLV-I) associated myelopathy/tropical spastic paraparesis (HAM/TSP) from Brazil and Iran and as controls, 16 Brazilian HTLVI seronegative individuals afflicted with other neurological diseases. It was observed in the HAM/TSP patients that: 1) all had an inflammatory reaction within the central nervous system (CNS); 2) 95% (19/20) showed oligoclonal bands reflecting intrathecal IgG synthesis; 3) 85% (17/20) presented a local synthesis of HTLVI antibodies; 4) 35% (7/20) had a measurable immunoglobulin (Ig) synthesis within the CNS. The CSF parameters of the HAM/TSP were compared with the clinical data (age at onset, duration of disease and disability level). Our data prove that CSF analysis is important for the diagnosis of HAM/TSP. There is no association between the severity of the disease and CSF findings.

Key words:HTLV-I, HAM/TSP, cerebrospinal fluid, multiple sclerosis.

RESUMO

Foram analisadas amostras de líquido cefalorraquidiano (LCR) e plasma de 20 pacientes com mielopatia associada ao HTLV-I (HAM/TSP) provenientes do Brasil e Irã e como controle, 16 de indivíduos brasileiros soronegativos para HTLV-I acometidos por outras doenças neurológicas (esclerose múltipla, epilepsia idiopática e mielopatia de etiologia desconhecida). Observou-se no grupo de pacientes com HAM/TSP: 1) reação inflamatória no SNC em todos os casos; 2) 95% (19/20) mostravam bandas oligoclonais refletindo síntese intratecal de IgG no SNC; 3)85% (7/20) apresentavam síntese local de anticorpos para HTLV-I; 4)35% (tinham síntese mensurável de imunoglobulinas no SNC. Os parâmetros do LCR dos pacientes com HAM/TSP foram comparados aos dados clínicos (idade de início, duração da doença e grau de incapacidade). Os dados apresentados neste estudo indicam a importância da analise do LCR para diagnóstico de HAM/TSP. Entretanto nenhuma associação entre a gravidade da doença e os achados do LCR foi demonstrada.

Palavras-chave: HTLV-I, mielopatia, HAM/TSP, líquido cefalorraquidiano, esclerose múltipla.

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Acknowledgement

We thank Prof. Dr. H.O. Reiber for his helpfull support at the Neurochemistry Labor von der Klinik und Poliklinik für Neurologie der Georg-August-Universitãt, Gõttingen, Germany.

Aceite: 4-novembro-1994.

Dra. Marzia Puccioni-Sohler - R. Francisco Sá, 26/1201 - 22080-010 Rio de Janeiro RJ - Brasil. Fax: (55)(21) 5211175.

1. Cavalcanti M, Ferreira O, Puccioni M, Novis S, Schechter M. HTLV-I associated neurologic manifestations in four generations of a Brazilian family. J AIDS 1993, 6:213-217.
2. Ceroni M, Piccardo P, Rodgers-Johnson P et al. Intrathecal synthesis of IgG antibodies to HTLV-I supports an etiological role for HTLV-I in tropical spastic paraparesis. Ann Neurol 1988, 23(Suppl):S188-S191.
3. Gessain A, Barin F, Vernant JC et al. Antibodies to human T-lymphotropic virus type I in patients with tropical spastic paraparesis. Lancet 1985, 2:407-410.
4. Gessain A, Caudie C, Gout O et al. Intrathecal synthesis of antibodies to human T lymphotropic virus type I and the presence of oligoclonal bands in the cerebrospinal fluid of patients with endemic tropical spastic paraparesis. J Inf Dis 1988, 6:1226-1234.
5. Gessain A, Gout O. Chronic myelopathy associated with human T -lymphotropic virus type I (HTLV-I). Ann Intern Med 1992, 117:933-946.
6. Hauser SL, Dawson DM, Lehrich JR et al. Intensive immunossupression in progressive multiple sclerosis. N Engl J Med 1983, 308:173-180.
7. Kitze B, Turner RW, Burchhardt M, Poser S, Hunsmann G, Weber T. Differential diagnosis of HTLV-I associated myelopathy and multiple sclerosis in Iranian patients. Clin Investig 1992, 70:1013-1018.
8. Kuroda Y, Fujiyama F, Nagugumo F. Analysis of factors of relevance to rapid clinical progression in HTLV-I-associated myelopathy. J Neurol Sci 1991, 105:61-66
9. Iwasaki Y. Pathology of chronic myelopathy associated with HTLVI infection (HAM/TSP). J Neurol Sci 1990, 96:103-123.
10. Link H, Cruz M, Gessain A et al. Chronic progressive myelopathy associated with HTLVI: oligoclonal bands and anti-HTLVI IgG antibodies in cerebrospinal fluid and serum. Neurology 1989, 39:1566-1571.
11. Marti-Fabregas J, Martinez JM, Ilia I, Escartin A. Myelopathy of unknown etiology. Acta Neurol Scand 1989, 80:455-460.
12. McLean BN, Rudge P, Thompson EJ. Viral specific IgG and IgM antibodies in the CSF of patients with tropical spastic paraparesis. J Neurol 1989, 236: 351-352.
13. Nagasato K, Nakamura T, Ohishi K et al. Active production of anti-human T-lymphotropic virus type I (HTLV-I) IgM antibody in HTLV-I-associated myelopathy. J Neuroimmunol 1991, 32:105-109.
14. Osame M, Usuku K, Izumo S et al. HTLV-I associated myelopathy, a new clinical entity. Lancet 1986, 1:1031-1032.
15. Poser CM, Paty DW, Scheinberg L et al. New diagnostic criteria for multiple sclerosis: guidelines for research protocols. Ann Neurol 1983, 13:227-231.
16. Reiber H, Lange P. Quantification of virus specific antibodies in cerebrospinal fluid and serum: sensitive and specific detection of antibody synthesis in brain. Clin Chem 1991, 37:1153-1160.
17. Roman G, Roman LN, Osame M. Human T lymphotropic virus type I neurotropism. Prog Med Virol. Basel: Karger, 1990; 37: 190-210.
18. Sheremata WA, Berger JR, Harrington WJ, Ayyar R, Staford M, DeFreitas E. Human T lymphotropic virus type-I associated myelopathy. Arch Neurol 1992, 49:1113-1118.
19. Spina-Franca A, Livramento JA, Machado LR et al. HTLV-I antibodies in serum and cerebrospinal fluid in tropical spastic paraparesis in Brazil. Arq Neuropsiquiatr 1990, 48:441-447.

Publication Dates

Publication in this collection
20 Jan 2011
Date of issue
June 1995

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

[1] 1. Cavalcanti M, Ferreira O, Puccioni M, Novis S, Schechter M. HTLV-I associated neurologic manifestations in four generations of a Brazilian family. J AIDS 1993, 6:213-217.

[2] 2. Ceroni M, Piccardo P, Rodgers-Johnson P et al. Intrathecal synthesis of IgG antibodies to HTLV-I supports an etiological role for HTLV-I in tropical spastic paraparesis. Ann Neurol 1988, 23(Suppl):S188-S191.

[3] 3. Gessain A, Barin F, Vernant JC et al. Antibodies to human T-lymphotropic virus type I in patients with tropical spastic paraparesis. Lancet 1985, 2:407-410.

[4] 4. Gessain A, Caudie C, Gout O et al. Intrathecal synthesis of antibodies to human T lymphotropic virus type I and the presence of oligoclonal bands in the cerebrospinal fluid of patients with endemic tropical spastic paraparesis. J Inf Dis 1988, 6:1226-1234.

[5] 5. Gessain A, Gout O. Chronic myelopathy associated with human T -lymphotropic virus type I (HTLV-I). Ann Intern Med 1992, 117:933-946.

[6] 6. Hauser SL, Dawson DM, Lehrich JR et al. Intensive immunossupression in progressive multiple sclerosis. N Engl J Med 1983, 308:173-180.

[7] 7. Kitze B, Turner RW, Burchhardt M, Poser S, Hunsmann G, Weber T. Differential diagnosis of HTLV-I associated myelopathy and multiple sclerosis in Iranian patients. Clin Investig 1992, 70:1013-1018.

[8] 8. Kuroda Y, Fujiyama F, Nagugumo F. Analysis of factors of relevance to rapid clinical progression in HTLV-I-associated myelopathy. J Neurol Sci 1991, 105:61-66

[9] 9. Iwasaki Y. Pathology of chronic myelopathy associated with HTLVI infection (HAM/TSP). J Neurol Sci 1990, 96:103-123.

[10] 10. Link H, Cruz M, Gessain A et al. Chronic progressive myelopathy associated with HTLVI: oligoclonal bands and anti-HTLVI IgG antibodies in cerebrospinal fluid and serum. Neurology 1989, 39:1566-1571.

[11] 11. Marti-Fabregas J, Martinez JM, Ilia I, Escartin A. Myelopathy of unknown etiology. Acta Neurol Scand 1989, 80:455-460.

[12] 12. McLean BN, Rudge P, Thompson EJ. Viral specific IgG and IgM antibodies in the CSF of patients with tropical spastic paraparesis. J Neurol 1989, 236: 351-352.

[13] 13. Nagasato K, Nakamura T, Ohishi K et al. Active production of anti-human T-lymphotropic virus type I (HTLV-I) IgM antibody in HTLV-I-associated myelopathy. J Neuroimmunol 1991, 32:105-109.

[14] 14. Osame M, Usuku K, Izumo S et al. HTLV-I associated myelopathy, a new clinical entity. Lancet 1986, 1:1031-1032.

[15] 15. Poser CM, Paty DW, Scheinberg L et al. New diagnostic criteria for multiple sclerosis: guidelines for research protocols. Ann Neurol 1983, 13:227-231.

[16] 16. Reiber H, Lange P. Quantification of virus specific antibodies in cerebrospinal fluid and serum: sensitive and specific detection of antibody synthesis in brain. Clin Chem 1991, 37:1153-1160.

[17] 17. Roman G, Roman LN, Osame M. Human T lymphotropic virus type I neurotropism. Prog Med Virol. Basel: Karger, 1990; 37: 190-210.

[18] 18. Sheremata WA, Berger JR, Harrington WJ, Ayyar R, Staford M, DeFreitas E. Human T lymphotropic virus type-I associated myelopathy. Arch Neurol 1992, 49:1113-1118.

[19] 19. Spina-Franca A, Livramento JA, Machado LR et al. HTLV-I antibodies in serum and cerebrospinal fluid in tropical spastic paraparesis in Brazil. Arq Neuropsiquiatr 1990, 48:441-447.