Human T-cell lymphotropic virus type I (HTLV-1) associated myelopathy and Sjögren's syndrome representing pulmonary nodular amyloidosis and multiple bullae: report of an autopsy case

2006 ◽  
Vol 448 (6) ◽  
pp. 874-876 ◽  
Author(s):  
Tomoko Seguchi ◽  
Yuka Kyoraku ◽  
Kazuko Saita ◽  
Toshihiko Ihi ◽  
Miyuki Nagai ◽  
...  
Keyword(s):  
T Cell ◽  
Sjögren’S Syndrome ◽  
Virus Type ◽  
Sjögren's Syndrome ◽  
Sjogren’S Syndrome ◽  
Sjogren's Syndrome ◽  
Type I ◽  
Autopsy Case ◽  
Human T Cell ◽  
Sjögren‘S Syndrome

Prevalence of Sjögren's syndrome in Brazilian patients infected with human T-cell lymphotropic virus

2016 ◽  
Vol 46 (7) ◽  
pp. 543-548 ◽  
Author(s):  
Daniela Assis do Vale ◽  
Jorge Casseb ◽  
Augusto César Penalva de Oliveira ◽  
Ivo Bussoloti Filho ◽  
Suzana Cantanhede Orsini Machado de Sousa ◽  
...  
Keyword(s):  
T Cell ◽  
Sjögren’S Syndrome ◽  
Sjögren's Syndrome ◽  
Sjogren’S Syndrome ◽  
Sjogren's Syndrome ◽  
Human T Cell ◽  
Sjögren‘S Syndrome

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

1995 ◽  
Vol 53 (4) ◽  
pp. 777-781 ◽  
Author(s):  
Sônia Regina A. A. Pinheiro ◽  
Marco Aurélio Lana-Peixoto ◽  
Anna B. Carneiro Proietti ◽  
Fernando Oréfice ◽  
Maria V. Lima-Martins ◽  
...  
Keyword(s):  
Sjögren’S Syndrome ◽  
Sjögren's Syndrome ◽  
Spastic Paraparesis ◽  
Clinical Manifestations ◽  
Sjogren’S Syndrome ◽  
Sjogren's Syndrome ◽  
Lower Limbs ◽  
Type I ◽  
Human T Cell ◽  
Sjögren‘S Syndrome

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.

Relationship between Sjögren's syndrome and human T-lymphotropic virus type I infection: Follow-up study of 83 patients

2000 ◽  
Vol 135 (2) ◽  
pp. 139-144 ◽  
Author(s):  
Hideki Nakamura ◽  
Atsushi Kawakami ◽  
Masahiro Tominaga ◽  
Ayumi Hida ◽  
Satoshi Yamasaki ◽  
...  
Keyword(s):  
Sjögren’S Syndrome ◽  
Virus Type ◽  
Sjögren's Syndrome ◽  
Sjogren’S Syndrome ◽  
Sjogren's Syndrome ◽  
Type I ◽  
T Lymphotropic Virus ◽  
Follow Up Study ◽  
Sjögren‘S Syndrome

Detection of HTLV-1 in the Labial Salivary Glands of Patients with Sjögren’s Syndrome: A Distinct Clinical Subgroup?

2012 ◽  
Vol 39 (4) ◽  
pp. 809-815 ◽  
Author(s):  
SUNG-JI LEE ◽  
JI SHIN LEE ◽  
MYUNG-GEUN SHIN ◽  
YUETSU TANAKA ◽  
DONG-JIN PARK ◽  
...  
Keyword(s):  
Salivary Glands ◽  
Sjögren’S Syndrome ◽  
Sjögren's Syndrome ◽  
Minor Salivary Gland ◽  
Sjogren’S Syndrome ◽  
Sjogren's Syndrome ◽  
Type I ◽  
Extraglandular Manifestations ◽  
Human T Cell ◽  
Sjögren‘S Syndrome

Objective.To examine whether patients with Sjögren’s syndrome (SS) can be distinguished based on the expression of human T cell lymphotrophic virus type I (HTLV-1) and, if so, whether the subgroups differ in their clinical features and serological measures.Methods.Polymerase chain reaction (PCR) and nested PCR were used to amplify viral DNA from peripheral blood mononuclear cells (PBMC) in 53 patients with SS, using primers from the HTLV-1 pX, p19, pol, and tax regions. Minor salivary gland biopsy specimens from 33 patients with SS were examined for the presence of HTLV-1 p19 or tax proteins immunohistochemically. The sociodemographic, glandular, and extraglandular manifestations, and laboratory findings including autoantibodies, complement, and immunoglobulin levels, were analyzed.Results.The HTLV-1 tax gene was detected in PBMC samples from 2 of 53 patients (3.8%), whereas the HTLV-1 pX, p19, and pol genes were not expressed. As well, 100% of PBMC samples from 4 family members of patients in whom the tax gene was detected also expressed the tax gene. Immunohistochemical staining for HTLV-1 p19 and tax was seen in 10 out of 33 (30.3%) patients with SS each. Overall, 14 (42.4%) patients expressed HTLV-1 p19 or tax proteins, and they had lower rheumatoid factor and C3 levels (p = 0.015 and p = 0.005, respectively) and higher lymphocyte counts (p = 0.016). The prevalence of glandular and extraglandular manifestations did not differ between the HTLV-1-positive and negative patients.Conclusion.Our findings suggest that HTLV-1 in the salivary glands is involved in the pathogenesis of a subpopulation of SS, and HTLV-1-associated SS might have different immunological patterns than idiopathic SS.

scholarly journals Unique expansion of IL-21+ Tfh and Tph cells under control of ICOS identifies Sjögren’s syndrome with ectopic germinal centres and MALT lymphoma

2020 ◽  
Vol 79 (12) ◽  
pp. 1588-1599 ◽  
Author(s):  
Elena Pontarini ◽  
William James Murray-Brown ◽  
Cristina Croia ◽  
Davide Lucchesi ◽  
James Conway ◽  
...  
Keyword(s):  
T Cell ◽  
Sjögren’S Syndrome ◽  
Sjögren's Syndrome ◽  
Sjogren’S Syndrome ◽  
Sjogren's Syndrome ◽  
Pcr Analysis ◽  
Organ Cultures ◽  
Cytokine Detection ◽  
Costimulatory Pathway ◽  
Sjögren‘S Syndrome

ObjectivesTo explore the relevance of T-follicular-helper (Tfh) and pathogenic peripheral-helper T-cells (Tph) in promoting ectopic lymphoid structures (ELS) and B-cell mucosa-associated lymphoid tissue (MALT) lymphomas (MALT-L) in Sjögren’s syndrome (SS) patients.MethodsSalivary gland (SG) biopsies with matched peripheral blood were collected from four centres across the European Union. Transcriptomic (microarray and quantitative PCR) analysis, FACS T-cell immunophenotyping with intracellular cytokine detection, multicolor immune-fluorescence microscopy and in situ hybridisation were performed to characterise lesional and circulating Tfh and Tph-cells. SG-organ cultures were used to investigate functionally the blockade of T-cell costimulatory pathways on key proinflammatory cytokine production.ResultsTranscriptomic analysis in SG identified Tfh-signature, interleukin-21 (IL-21) and the inducible T-cell co-stimulator (ICOS) costimulatory pathway as the most upregulated genes in ELS+SS patients, with parotid MALT-L displaying a 400-folds increase in IL-21 mRNA. Peripheral CD4+CXC-motif chemokine receptor 5 (CXCR5)+programmed cell death protein 1 (PD1)+ICOS+ Tfh-like cells were significantly expanded in ELS+SS patients, were the main producers of IL-21, and closely correlated with circulating IgG and reduced complement C4. In the SG, lesional CD4+CD45RO+ICOS+PD1+ cells selectively infiltrated ELS+ tissues and were aberrantly expanded in parotid MALT-L. In ELS+SG and MALT-L parotids, conventional CXCR5+CD4+PD1+ICOS+Foxp3- Tfh-cells and a uniquely expanded population of CXCR5-CD4+PD1hiICOS+Foxp3- Tph-cells displayed frequent IL-21/interferon-γ double-production but poor IL-17 expression. Finally, ICOS blockade in ex vivo SG-organ cultures significantly reduced the production of IL-21 and inflammatory cytokines IL-6, IL-8 and tumour necrosis factor-α (TNF-α).ConclusionsOverall, these findings highlight Tfh and Tph-cells, IL-21 and the ICOS costimulatory pathway as key pathogenic players in SS immunopathology and exploitable therapeutic targets in SS.

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