{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T22:14:42Z","timestamp":1767651282855,"version":"build-2065373602"},"reference-count":54,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2024,2,17]],"date-time":"2024-02-17T00:00:00Z","timestamp":1708128000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Background: DFNA9 is a genetic disease of the inner ear, causing progressive bilateral sensorineural deafness and bilateral vestibulopathy (BV). In this study, DizzyQuest, a mobile vestibular diary, and the MOX accelerometer were combined to assess the daily life functional limitations and physical activity of patients with DFNA9 suffering from BV. These parameters might be appropriate as potential candidacy criteria and outcome measures for new therapeutic interventions for BV. Methods: Fifteen DFNA9 patients with BV and twelve age-matched healthy controls were included. The DizzyQuest was applied for six consecutive days, which assessed the participants\u2019 extent of functional limitations, tiredness, types of activities performed during the day, and type of activity during which the participant felt most limited. The MOX accelerometer was worn during the same six days of DizzyQuest use, measuring the participants intensity and type of physical activity. Mixed-effects linear and logistic regression analyses were performed to compare the DFNA9 patients and control group. Results: DFNA9 patients with BV felt significantly more limited in activities during the day compared to the age-matched controls, especially in social participation (p &lt; 0.005). However, these reported limitations did not cause adjustment in the types of activities and did not reduce the intensity or type of physical activity measured with accelerometry. In addition, no relationships were found between self-reported functional limitations and physical activity. Conclusions: This study demonstrated that self-reported functional limitations are significantly higher among DFNA9 patients with BV. As a result, these limitations might be considered as part of the candidacy criteria or outcome measures for therapeutic interventions. In addition, the intensity or type of physical activity performed during the day need to be addressed more specifically in future research.<\/jats:p>","DOI":"10.3390\/jcm13041131","type":"journal-article","created":{"date-parts":[[2024,2,19]],"date-time":"2024-02-19T03:18:38Z","timestamp":1708312718000},"page":"1131","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["The DizzyQuest Combined with Accelerometry: Daily Physical Activities and Limitations among Patients with Bilateral Vestibulopathy Due to DFNA9"],"prefix":"10.3390","volume":"13","author":[{"given":"Erik","family":"Martin","sequence":"first","affiliation":[{"name":"Division of Balance Disorders, Department of Otorhinolaryngology and Head and Neck Surgery, Maastricht University Medical Center+, 6229 HX Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sofie","family":"de Hoon","sequence":"additional","affiliation":[{"name":"Division of Balance Disorders, Department of Otorhinolaryngology and Head and Neck Surgery, Maastricht University Medical Center+, 6229 HX Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3691-3912","authenticated-orcid":false,"given":"Joost","family":"Stultiens","sequence":"additional","affiliation":[{"name":"Division of Balance Disorders, Department of Otorhinolaryngology and Head and Neck Surgery, Maastricht University Medical Center+, 6229 HX Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Miranda","family":"Janssen","sequence":"additional","affiliation":[{"name":"Division of Balance Disorders, Department of Otorhinolaryngology and Head and Neck Surgery, Maastricht University Medical Center+, 6229 HX Maastricht, The Netherlands"},{"name":"School for Mental Health and Neuroscience (MHENS), Maastricht University, 6229 ER Maastricht, The Netherlands"},{"name":"Department of Methodology and Statistics, Care and Public Health Research Institute (CAPHRI), Maastricht University, 6229 ER Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0961-3167","authenticated-orcid":false,"given":"Hans","family":"Essers","sequence":"additional","affiliation":[{"name":"Department of Nutrition and Movement Sciences, NUTRIM School of Nutrition and Translational Research in Metabolism, Maastricht University, 6229 ER Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8236-8754","authenticated-orcid":false,"given":"Kenneth","family":"Meijer","sequence":"additional","affiliation":[{"name":"Department of Nutrition and Movement Sciences, NUTRIM School of Nutrition and Translational Research in Metabolism, Maastricht University, 6229 ER Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wouter","family":"Bijnens","sequence":"additional","affiliation":[{"name":"Research Engineering (IDEE), Maastricht University, 6229 ER Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maurice","family":"van de Berg","sequence":"additional","affiliation":[{"name":"Division of Balance Disorders, Department of Otorhinolaryngology and Head and Neck Surgery, Maastricht University Medical Center+, 6229 HX Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0074-5814","authenticated-orcid":false,"given":"Nolan","family":"Herssens","sequence":"additional","affiliation":[{"name":"Space Medicine Team (HRE-OM), European Astronaut Centre, European Space Agency, 51147 Cologne, Germany"},{"name":"Faculty of Medicine and Health Sciences, University of Antwerp, 2000 Antwerp, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sebastien","family":"Janssens de Varebeke","sequence":"additional","affiliation":[{"name":"Department of ENT Head & Neck Surgery, Jessa Hospital, 3500 Hasselt, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4101-5279","authenticated-orcid":false,"given":"Ann","family":"Hallemans","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health Sciences, University of Antwerp, 2000 Antwerp, Belgium"},{"name":"Multidisciplinary Motor Centre Antwerp (M2OCEAN), University of Antwerp, 2000 Antwerp, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vincent","family":"Van Rompaey","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health Sciences, University of Antwerp, 2000 Antwerp, Belgium"},{"name":"Department of Otorhinolaryngology & Head and Neck Surgery, Antwerp University Hospital, 2650 Antwerp, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nils","family":"Guinand","sequence":"additional","affiliation":[{"name":"Service of Otorhinolaryngology Head and Neck Surgery, Department of Clinical Neurosciences, Geneva University Hospitals, 1205 Geneva, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3911-8826","authenticated-orcid":false,"given":"Angelica","family":"Perez-Fornos","sequence":"additional","affiliation":[{"name":"Service of Otorhinolaryngology Head and Neck Surgery, Department of Clinical Neurosciences, Geneva University Hospitals, 1205 Geneva, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Josine","family":"Widdershoven","sequence":"additional","affiliation":[{"name":"Division of Balance Disorders, Department of Otorhinolaryngology and Head and Neck Surgery, Maastricht University Medical Center+, 6229 HX Maastricht, The 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