High prevalence of wild-type transthyretin deposition in patients with idiopathic carpal tunnel syndrome: a common cause of carpal tunnel syndrome in the elderly

Carpal tunnel syndrome is the most common type of entrapment neuropathy. However, the cause of carpal tunnel syndrome remains unclear in most cases. Senile systemic amyloidosis, induced by wild-type transthyretin deposition, is a prevalent aging-related disorder and often accompanied by carpal tunnel syndrome. In this study, we measured the frequency of unrecognized wild-type transthyretin deposition in patients with idiopathic carpal tunnel syndrome. One hundred twenty-three patients with carnal tunnel syndrome, including 100 idiopathic patients, treated by carpal tunnel release surgery were analyzed. Tenosynovial tissues obtained at surgery were analyzed by Congo red and immunohistochemical staining. If staining for transthyretin was positive, the entire transthyretin gene was analyzed by direct DNA sequencing. We also analyzed tenosynovial tissues from 32 autopsy cases as controls. Thirty-four patients (34.0%) with idiopathic carpal tunnel syndrome showed amyloid deposition in the tenosynovial tissue, and all amyloid showed specific immunolabeling with antitransthyretin antibody. Direct DNA sequencing of the entire transthyretin gene did not reveal any mutations, indicating that all amyloid deposits were derived form wild-type transthyretin. Statistical analysis using logistic regression showed that the prevalence of transthyretin deposition in the idiopathic carpal tunnel syndrome group was significantly higher than that in controls (odds ratio, 15.8; 95% confidence interval, 3.3-75.7), and age and male sex were independent risk factors for transthyretin amyloid deposition. Our results demonstrate that wild-type transthyretin deposition is a common cause of carpal tunnel syndrome in elderly men. It is likely that many patients develop carpal tunnel syndrome as an initial symptom of senile systemic amyloidosis.ArticleHUMAN PATHOLOGY. 42(11):1785-1791 (2011)journal articl

Validation of the Japanese Society for Surgery of the Hand version of the Carpal Tunnel Syndrome Instrument

AbstractBackgroundThe Carpal Tunnel Syndrome Instrument (CTSI) is a disease-specific, self-administered questionnaire that consists of a symptom severity scale (SS) and a functional status scale (FS). The CTSI was cross-culturally adapted and developed by the Impairment Evaluation Committee, Japanese Society for Surgery of the Hand (JSSH). The purpose of this study was to test the reliability, validity, and responsiveness of the Japanese version of the CTSI (CTSI-JSSH).MethodsA consecutive series of 87 patients with carpal tunnel syndrome completed the CTSI-JSSH, the JSSH version of the Disability of the Arm, Shoulder, and Hand questionnaire (DASH-JSSH), and the 36-Item Short-Form Health Survey (SF-36). Seventy-two of the patients were reassessed for test–retest reliability 1 or 2 weeks later. Reliability was investigated by the reproducibility and the internal consistency. To analyze the validity, a factor analysis (principal axis factoring) of the CTSI-JSSH and the correlation coefficients between the CTSI-JSSH and DASH-JSSH were obtained. The responsiveness was examined by calculating the standardized response mean (SRM; mean change/SD) and effect size (mean change/SD of baseline value) after carpal tunnel release in 42 patients.ResultsCronbach’s alpha coefficients for the CTSI-JSSH-SS and the CTSI-JSSH-FS were 0.84 and 0.90, respectively, and the intraclass correlation coefficients were 0.82 and 0.83, respectively. The unidimensionality of the CTSI-JSSH-SS was barely confirmed; the unidimensionality of the CTSI-JSSH-FS was confirmed. The correlation coefficients between the CTSI-JSSH-FS and the CTSI-JSSH-SS or DASH-JSSH were 0.58 and 0.80, respectively. The correlation coefficient between the CTSI-JSSH-SS and DASH-JSSH was 0.54. The correlation coefficients between the subscales of SF-36 and the CTSI-JSSH-SS or the CTSI-JSSH-FS ranged from -0.23 to -0.66 and from -0.19 to -0.63, respectively. The SRMs/effect sizes of the CTSI-JSSH-SS and the CTSI-JSSH-FS were -0.85/-0.99 and -0.70/-0.61, which indicated that they were more than moderately sensitive.ConclusionsThe CTSI-JSSH has sufficient reliability, validity, and responsiveness to assess the health status in carpal tunnel syndrome

Validation of the Japanese Society for Surgery of the Hand Version of the Quick Disability of the Arm, Shoulder, and Hand (QuickDASH-JSSH) questionnaire

AbstractBackgroundThe Quick Disability of the Arm, Shoulder, and Hand (QuickDASH) questionnaire is a region-specific, selfadministered questionnaire, which consists of a disability/symptom (QuickDASH-DS) scale, and the same two optional modules, the work (DASH-W) and the sport/music (DASHSM) modules, as the DASH. After the Japanese version of DASH (DASH-JSSH) was cross-culturally adapted and developed, we made the Japanese version of QuickDASH (QuickDASH-JSSH) by extracting 11 out of 30 items of the DASH-JSSH regarding disability/symptoms. The purpose of this study was to test the reliability, validity, and responsiveness of QuickDASH-JSSH.MethodsA series of 72 patients with upper extremity disorders completed the QuickDASH-JSSH, the 36-Item Short-Form Health Survey (SF-36), and the Visual Analog Scale (VAS) for pain. Thirty-eight of the patients were reassessed for test–retest reliability 1 or 2weeks later. Reliability was investigated by the reproducibility and internal consistency. To analyze the validity, a principal component analysis and the correlation coefficients between the QuickDASH-JSSH and the SF-36 were obtained. The responsiveness was examined by calculating the standardized response mean (SRM; mean change/SD) and effect size (mean change/SD of baseline value) after carpal tunnel release of the 17 patients with carpal tunnel syndrome.ResultsCronbach’s alpha coefficient in the QuickDASH-DS was 0.88. The intraclass correlation coefficient (ICC) for the same was 0.82. The unidimensionality of the QuickDASH-DS was confirmed. The correlation coefficients between the QuickDASH-DS and the DASH-DS, DASH-W, or the DASH-SM were 0.92, 0.81, or 0.76, respectively. The correlation coefficients between the QuickDASH-DS score and the subscales of the SF-36 ranged from −0.29 to −0.73. The correlation coefficient between the QuickDASH-DS score and the VAS for pain was 0.52. The SRM/effect size of QuickDASHDS was −0.54/−0.37, which indicated moderate sensitivity.ConclusionThe Japanese version of QuickDASH has equivalent evaluation capacities to the original QuickDASH

High prevalence of wild-type transthyretin deposition in patients with idiopathic carpal tunnel syndrome: a common cause of carpal tunnel syndrome in the elderly

Carpal tunnel syndrome is the most common type of entrapment neuropathy. However, the cause of carpal tunnel syndrome remains unclear in most cases. Senile systemic amyloidosis, induced by wild-type transthyretin deposition, is a prevalent aging-related disorder and often accompanied by carpal tunnel syndrome. In this study, we measured the frequency of unrecognized wild-type transthyretin deposition in patients with idiopathic carpal tunnel syndrome. One hundred twenty-three patients with carnal tunnel syndrome, including 100 idiopathic patients, treated by carpal tunnel release surgery were analyzed. Tenosynovial tissues obtained at surgery were analyzed by Congo red and immunohistochemical staining. If staining for transthyretin was positive, the entire transthyretin gene was analyzed by direct DNA sequencing. We also analyzed tenosynovial tissues from 32 autopsy cases as controls. Thirty-four patients (34.0%) with idiopathic carpal tunnel syndrome showed amyloid deposition in the tenosynovial tissue, and all amyloid showed specific immunolabeling with antitransthyretin antibody. Direct DNA sequencing of the entire transthyretin gene did not reveal any mutations, indicating that all amyloid deposits were derived form wild-type transthyretin. Statistical analysis using logistic regression showed that the prevalence of transthyretin deposition in the idiopathic carpal tunnel syndrome group was significantly higher than that in controls (odds ratio, 15.8; 95% confidence interval, 3.3-75.7), and age and male sex were independent risk factors for transthyretin amyloid deposition. Our results demonstrate that wild-type transthyretin deposition is a common cause of carpal tunnel syndrome in elderly men. It is likely that many patients develop carpal tunnel syndrome as an initial symptom of senile systemic amyloidosis.ArticleHUMAN PATHOLOGY. 42(11):1785-1791 (2011)journal articl

Responsiveness of the Japanese version of the patient-rated wrist evaluation (PRWE-J) and physical impairment measurements in evaluating recovery after treatment of ulnocarpal abutment syndrome

AbstractBackgroundWe evaluated the responsiveness of patient-derived questionnaires and physical findings in evaluating recovery after treatment of ulnocarpal abutment syndrome.MethodsPatients were assessed at their initial visit to our clinic and again 3months after the treatment. At each visit, patients completed a Short Form-36, the Japanese Society for Surgery of the Hand version of Disability of the Arm, Shoulder, and Hand questionnaire (DASH-JSSH), and the Japanese version of patient-rated wrist evaluation (PRWE-J). Grip strength, range of motion, and visual analogue scale for wrist pain were also examined at each visit. Satisfaction with treatment was questioned after 3months using a Likert scale. Standardized response means (SRM) and effect sizes were calculated to evaluate the responsiveness.ResultsThe PRWE-J (SRM, 1.35) was the most responsive questionnaire, followed by the DASH-JSSH (SRM, 0.81) and the Short Form-36 (SRM, −0.38 to −1.19). Of the physical tests, grip strength (SRM, 0.81) was more responsive than range of motion (SRM, 0.01 to −0.29). The visual analogue pain scale (SRM, 1.56) was highly responsive. Changes in the PRWE score were correlated with the satisfaction rating for the treatment.ConclusionsResponsive patient-derived scales can assist in the outcome evaluation of patients with ulnocarpal abutment syndrome