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. 2015 Nov 4:16:335.
doi: 10.1186/s12891-015-0773-1.

Reevaluation for clinical manifestations of HTLV-I-seropositive patients with Sjögren's syndrome

Hideki Nakamura  1 , Toshimasa Shimizu  2 , Yukinori Takagi  3 , Yoshiko Takahashi  4 , Yoshiro Horai  5 , Yoshikazu Nakashima  6 , Shuntaro Sato  7 , Hirokazu Shiraishi  8 , Tatsufumi Nakamura  9 , Junya Fukuoka  10 , Takashi Nakamura  11 , Atsushi Kawakami  12
Affiliations

Reevaluation for clinical manifestations of HTLV-I-seropositive patients with Sjögren's syndrome

Hideki Nakamura et al. BMC Musculoskelet Disord. .

Abstract

Background: The aim of the study was to reassess the prevalence and characteristics of human T lymphotropic virus type I (HTLV-I)-associated Sjögren's syndrome (SS) and SS in HTLV-I-associated myelopathy (HAM) based on the American European Consensus Group (AECG) criteria in HTLV-I endemic area, Nagasaki prefecture.

Methods: The 349 patients who underwent a minor salivary gland biopsy (MSGB) for suspected SS were retrospectively classified by AECG classification criteria and divided with or without anti-HTLV-I antibody.

Results: The HTLV-I data-available 294 patients were investigated. One hundred-seventy patients were classified as SS and 26.5 % were HTLV-I-seropositive. We have included 26 patients with HTLV-I-associated myelopathy (HAM) and 38.5 % were classified as having SS. The prevalences of ANA and anti-SS-A/Ro antibody of HAM + SS were significantly low compared to the HTLV-I asymptomatic carriers (AC) with SS and the HTLV-I-seronegative SS patients, although lacrimal dysfunction tended to be high in HAM + SS and significantly high in AC + SS patients compared with the patients with HTLV-I-seronegative SS. The focus scores of MSGB in the HAM + SS patients were similar to those of the AC + SS patients and the HTLV-I-seronegative patients with SS. Among the MSGB-positive patients, there was a low prevalence of ANA in the HAM + SS patients. Similar results were obtained in case of anti-SS-A/Ro or SS-B/La antibody.

Conclusion: In HTLV-I endemic area, high prevalence of anti-HTLV-I antibody among SS as well as the characteristics of HAM + SS and AC + SS was still determined by AECG classification criteria.

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Figures

Fig. 1
Fig. 1
Patients with suspected SS who underwent a minor salivary gland biopsy (MSGB). All patients who underwent an MSGB were divided into subgroups according to the revised classification criteria determined by the American European Consensus Group (AECG). SS: Sjögren’s syndrome, AC: HTLV-I asymptomatic carrier. Shaded squares indicate the patients with SS
Fig. 2
Fig. 2
Distribution of focus scores (FS) among the three patient groups. The MSGB focus scores were determined by the method of Greenspan et al. [18]. The presence of at least one focus consisting of mononuclear cell (MNC) aggregation was determined as grade 3. Bars indicate the median value of each column. The significance of differences was calculated by Mann-Whitney’s U-test. NS; not significant AC: HTLV-I asymptomatic carrier
Fig. 3
Fig. 3
Prevalence of objective items in SS patients with grades 3 or 4 among the four patient groups. The prevalence of each item was determined by Chisholm & Mason grading [17]. The presence of at least one focus consisting of MNC aggregation was determined as grade 3. The positive rate of four objective items is shown. The significance of differences was assessed using Fisher’s exact probability test. (*p < 0.05 vs. HAM + SS, **p < 0.01 vs. HAM + SS) AC: HTLV-I asymptomatic carrier, ANA: anti-nuclear antibody
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References

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