Creation and validation of a bladder dysfunction symptom score for HTLV-1-associated myelopathy/tropical spastic paraparesis | Orphanet Journal of Rare Diseases | Full Text

Creation and validation of a bladder dysfunction symptom score for HTLV-1-associated myelopathy/tropical spastic paraparesis | Orphanet Journal of Rare Diseases | Full Text

Creation and validation of a bladder dysfunction symptom score for HTLV-1-associated myelopathy/tropical spastic paraparesis

• Natsuko Yamakawa, 
• Naoko Yagishita, 
• Tomohiro Matsuo, 
• Junji Yamauchi, 
• Takahiko Ueno, 
• Eisuke Inoue, 
• Ayako Takata, 
• Misako Nagasaka, 
• Natsumi Araya, 
• Daisuke Hasegawa, 
• Ariella Coler-Reilly, 
• Shuntaro Tsutsumi, 
• Tomoo Sato, 
• Abelardo Araujo, 
• Jorge Casseb, 
• Eduardo Gotuzzo, 
• Steven Jacobson, 
• Fabiola Martin, 
• Marzia Puccioni-Sohler, 
• Graham P. Taylor, 
• Yoshihisa Yamano & 
• the Japan Clinical Research Group on HAM/TSP

Orphanet Journal of Rare Diseases volume 15, Article number: 175 (2020) Cite this article

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Abstract

Background

Urinary dysfunction is one of the main features of human T-cell leukemia virus type 1-associated myelopathy/tropical spastic paraparesis (HAM/TSP). However, a comprehensive assessment of the severity is difficult because a standardized assessment measure is unavailable. Therefore, this study aimed to develop a novel symptom score for the assessment of urinary dysfunction in HAM/TSP. We interviewed 449 patients with HAM/TSP using four internationally validated questionnaires for assessment of urinary symptoms (27 question items in total): the International Prostate Symptom Score; the International Consultation on Incontinence Questionnaire-Short Form; the Overactive Bladder Symptom Score; and the Nocturia Quality-of-Life questionnaire. We developed a symptom score based on the data of 322 patients who did not use urinary catheters by selecting question items from questionnaires focused on descriptive statistics, correlation analysis, and exploratory factor analysis. The score distribution, reliability, and validity of the developed score were evaluated.

Results

First, 16 questions related to quality of life, situations, or subjective assessment were omitted from the 27 questions. Exploratory factor analysis revealed that the remaining 11 questions pertained to three factors: frequent urination, urinary incontinence, and voiding symptoms. Three questions, which had similar questions with larger factor loading, were deleted. Finally, we selected eight question items for inclusion in the novel score. The score distribution exhibited no ceiling or floor effect. The Cronbach’s alpha (0.737) demonstrated reliable internal consistency. The new score comprised two subscales with acceptable factorial validity (inter-factor correlation coefficient, 0.322): storage symptoms (frequent urination plus urinary incontinence) and voiding symptoms. The correlation between each item and the subscales suggested acceptable construct validity.

Conclusions

We developed a novel score, the HAM/TSP-Bladder Dysfunction Symptom Score, and demonstrated its reliability and validity. The applicability of this score to patients using catheters should be examined in future research.