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.
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.
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.
Orphanet journal of rare diseases. 2020 Jul 03*** epublish ***
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, Japan Clinical Research Group on HAM/TSP
Department of Rare Diseases Research, Institute of Medical Science, St. Marianna University School of Medicine, Kawasaki, Japan., Department of Urology, Nagasaki University Graduate School of Biochemical Sciences, Nagasaki, Japan., Department of Medical Informatics, St. Marianna University School of Medicine, Kawasaki, Japan., Department of Preventive Medicine, St. Marianna University School of Medicine, Kawasaki, Japan., Department of Oncology, Karmanos Cancer Institute/Wayne State University, Detroit, MI, USA., Department of Advanced Medical Innovation, St. Marianna University Graduate School of Medicine, Kawasaki, Japan., Laboratory for Clinical Research in Neuroinfections, Evandro Chagas National Institute of Infectious Diseases, FIOCRUZ, Rio de Janeiro, Brazil., Institute of Tropical Medicine of Sau Paulo, Sao Paulo, SP, Brazil., Instituto de Medicina Tropical "Alexander von Humbldt", Universidad Peruana Cayetano Heredia, Lima, Peru., Viral immunology Section, Neuroimmunology Branch, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, MD, USA., Faculty of Medicine, University of Queensland, 288 Herston Road, Herston, QLD, 4006, Australia., Escola de Medicina e Cirurgia da Universidade Federal do Estado do Rio de Janeiro/ Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil., Section of Virology, Department of Medicine, Imperial College London, St Mary's Campus, Norfolk Place, London, W2 1PG, UK., Department of Rare Diseases Research, Institute of Medical Science, St. Marianna University School of Medicine, Kawasaki, Japan. .