Lymphocyte alveolitis in HAM/TSP patients: preliminary report

Abstract

SUMMARY -H.TLV-I associate d myelopath y ha s bee n describe d a s a systemi c diseas e characterized b y manifestation s i n severa l organ s outsid e th e nervou s system . W e repor t inflammator y pulmonary involvemen t i n patient s wit h diagnosi s o f HAM .KEY WORDS : HT'LV-I , HAM/TSP , alveolitis , pulmonar y involvement . HTLV-I 'associate d myelopath y (HAM ) ha s bee n describe d a s a neurologi c diseas e characterized b y spasti c paraparesis , sensitiv e syndromes , periphera l neuropathy , constipation , bladder disturbanc e an d impotenc e o r decrease d libid o 5,s. Ther e hav e bee n som e report s o f systemic non-neurologi c manifestation s a s vasculitis , uveitis , Sjogren' s syndrome , arthropathy , cryoglobulinemia, ichthyosis , polymyositi s an d adul t T-cel l leukemia/lymphom a associate d with HA M 7. Pulmonar y infiltrate s i n HA M patient s wer e firs t describe d i n native s o f Martinique 2. I n Japan , bronchoalveola r lavag e studie s showe d fiv e patients i wit h increase d tota l cell count s an d increase d proportio n o f lymphocyte s i n lavag e fluid , a s compare d wit h dat a found i n health y non-smoki n volunteer s 9 . On e o f thes e patients , wit h a n abnorma l ches t X-ray, ha d transbronchia l lun g biops y tha t reveale d lymphocyt e infiltration s i n th e alveola r structures whic h di d no t resembl e leukemi a cells .In orde r t o clarif y th e clinica l pictur e an d pathogenesi s o f HTLV-I associate d myelopathy w e hav e studie d th e pulmonar y involvemen t i n eigh t patient s wit h HAM .PATIENTS AND METHO D -Seve n wome n an d on e man , wit h age s tha t range d fro m 3 9 to 6 7 years , wer e admitte d i n thi s study . Th e patient s presente d th e basi c clinica l an d CS F features o f HA M a s ha s bee n describe d elsewher e 5 . CS F an d seru m sample s wer e collecte d from th e patient s lan d teste d fo r HTLV-I wit h a commerciall y availabl e enzym e immunoassay ; the specimen s wer e repeatedl y reactiv e i n thi s screenin g tes t an d furthe r confirme d b y Wester n blot.All patient s wer e submitte d t o pulmonar y evaluatio n b y a certifie d pneumologis t (K.M.) . In eac h patien t wa s performe d hemograma , electrocardiogram , ches t X-ray , arteria l gasometri c studies an d spirometr y a t rest . T o accomplis h th e bronchoalveola r lavag e th e bronchoscop e was wedge d int o a dista l bronchu s an d 30 0 m l o f steril e salin e wa s use d t o recove r th e cell s and epithelia l linin g fluid . Th e bronchoalveola r lavag e wa s carrie d ou t i n a singl e laborator y by th e sam e researche r usin g identica l techniques .•

Molecular and cellular aspects of HTLV-1 associated leukemogenesis in vivo

“…2,3 Furthermore, this virus has been associated with the development of a chronic progressive neuromyelopathy (tropical spastic paraparesis (TSP)/HTLV-1-associated myelopathy (HAM)), 4 and, to a lesser extent, to a variety of inflammatory diseases. [5][6][7][8][9] Among the 15-25 million individuals infected worldwide, approximately 3% to 5% will develop ATLL, depending on as yet unknown cofactors. ATLL harbors different clinical features resulting in a division of the spectrum of the disease into four clinical subtypes 10,11 referred to as acute, lymphoma, chronic and smoldering subtypes.…”

High circulating proviral load with oligoclonal expansion of HTLV-1 bearing T cells in HTLV-1 carriers with strongyloidiasis

“…Furthermore, this virus has been associated, though to a lesser extent, to the development of a variety of in¯ammatory diseases (Eguchi et al, 1992;LaGrenade et al, 1990;Mattos et al, 1993;Mochizuki et al, 1994;Sato et al, 1991;Sherman et al, 1995). Among the 15 ± 25 million individuals infected worldwide, approximately 3 ± 5% will develop ATLL, depending on as yet unknown cofactors.…”

Pulmonary involvement in patients with HTLV-I associated myelopathy

“…As has been previously described, pulmonary involvement in the majority of HAM patients has been proved to be asymptomatic 8,10,14 . In our study 3 patients with HAM presented symptoms of dyspnea and coughing and 2 showed radiological images suggestive of interstitial lung disease.…”

“…In the first paper that identified pulmonary involvement in patients with HAM in South America 8 , we showed that seven of eight patients had lymphocytosis in the bronchoalveolar fluid. Sugimoto et al described T-lymphocytosis in the BAL fluid of 80% of the 15 patients with HAM in Japan 13,14 and Couderc et al 1 reported 76% of T-lymphocyte alveolitis of the 21 patients from Africa.…”

“…It has been shown that patients with HTLV-I associated myelopathy (HAM) are frequently associated with vasculitis, ichtiosis, Sjogren syndrome, and other immunological diseases 3,4,6 . Gessain et al 2 have reported evidence of pulmonary involvement in patients with HAM and subsequently several authors described lymphocytosis in the bronchoalveolar lavage (BAL) fluid of patients with HAM 1,8,13 . The proliferation of peripheral blood mononuclear cells (PBMC), production of interleukin-2 (IL-2), polyclonal Federal University of Bahia (UFBA).…”