Reliability of functional outcome measures in adults with neurofibromatosis 2

SAGE Open Med. 2022 Aug 19:10:20503121221118996. doi: 10.1177/20503121221118996. eCollection 2022.

Abstract

Objective: To determine inter- and intra-rater reliability of functional performance outcome measures in people with neurofibromatosis 2. To ascertain how closely objective and subjective measures align.

Methods: Twenty-nine people with neurofibromatosis 2 were recorded performing the modified clinical test of sensory integration and balance, four square step test and modified nine-hole peg tests. Three raters scored each measure to determine inter-rater reliability. One rater scored the measures a second time to determine intra-rater reliability. Participants also completed a disease-specific quality of life questionnaire and dynamic visual acuity testing.

Results: Inter-rater and intra-rater reliability scores (intra-class correlation coefficient) were excellent for all tests (intra-class correlation coefficient r ⩾ 0.9). The four square step test correlated with perceived walking challenges and modified clinical test of sensory integration and balance correlated with perceived balance challenges in a neurofibromatosis 2 quality of life patient report outcome measure.

Conclusion: The modified clinical test of sensory integration and balance, four square step test and modified nine-hole peg tests are potentially useful measures for monitoring neurofibromatosis 2.

Keywords: NF2; Neurofibromatosis 2; minimal detectable change; outcome measures; reliability; standard error of measurement.