Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement.
The Revised Hammersmith Scale (RHS) for Spinal Muscular Atrophy (SMA) was designed as a psychometrically robust clinical outcome assessment to assess physical abilities of patients with type 2 and 3 SMA. The reliability properties of the RHS have not yet been reported. A prospective RHS reliability...
| Main Authors: | , , , , , |
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| Format: | Article |
| Language: | English |
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Public Library of Science (PLoS)
2022-01-01
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| Series: | PLoS ONE |
| Online Access: | https://doi.org/10.1371/journal.pone.0278996 |
| _version_ | 1811177214006263808 |
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| author | Danielle Ramsey Gita Ramdharry Mariacristina Scoto Francesco Muntoni Amanda Wallace SMA REACH UK network |
| author_facet | Danielle Ramsey Gita Ramdharry Mariacristina Scoto Francesco Muntoni Amanda Wallace SMA REACH UK network |
| author_sort | Danielle Ramsey |
| collection | DOAJ |
| description | The Revised Hammersmith Scale (RHS) for Spinal Muscular Atrophy (SMA) was designed as a psychometrically robust clinical outcome assessment to assess physical abilities of patients with type 2 and 3 SMA. The reliability properties of the RHS have not yet been reported. A prospective RHS reliability study was undertaken in a UK cohort of experienced neuromuscular paediatric Physiotherapists. Reliability testing was conducted via a virtual survey platform two weeks apart. Through the virtual platform participants scored videos of two RHS assessments, one of a child with SMA 2 and one of a child with SMA 3. Inter and intra-rater reliability was analysed using a type 3 Intraclass Correlation Coefficient (ICC). Intra-rater agreement was further analysed using Bland Altman (BA) Limits of Agreement (LOA) and plots. The acceptable inter and intra-rater variability was set as a change of ± 2 by the international team of expert physiotherapists who developed the RHS. Inter-rater agreement, n = 22 raters, type 3 ICC was 0.989 (95% CI 0.944 to 1.00), 97.7% of scores were within the acceptable limits of ± 2 points. Intra-rater agreement, n = 21 raters, type 3 ICC ranged from 0.922 to 1.0, with 97.6% of scores within the acceptable limits of ± 2 points. The mean SMA 2 intra-rater difference was -0.10 (-0.6 to 0.4), with lower LOA -2.24 and upper LOA +2.04. Intra-rater difference between tests for SMA 3 intra-rater difference was -0.05 (-0.6 to 0.5), with lower LOA -2.48 and upper LOA +2.38. Intra-rater scoring precision fell within BA agreement limits of ±2 points. The results demonstrate that the RHS is highly reliable when used by experienced UK physiotherapists, and variability of test scores regarding inter and intra-rater reliability was confirmed to lie within ±2 points. |
| first_indexed | 2024-04-10T22:57:59Z |
| format | Article |
| id | doaj.art-5dde2486335a4cf3ad0774a744c2c014 |
| institution | Directory Open Access Journal |
| issn | 1932-6203 |
| language | English |
| last_indexed | 2024-04-10T22:57:59Z |
| publishDate | 2022-01-01 |
| publisher | Public Library of Science (PLoS) |
| record_format | Article |
| series | PLoS ONE |
| spelling | doaj.art-5dde2486335a4cf3ad0774a744c2c0142023-01-14T05:31:23ZengPublic Library of Science (PLoS)PLoS ONE1932-62032022-01-011712e027899610.1371/journal.pone.0278996Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement.Danielle RamseyGita RamdharryMariacristina ScotoFrancesco MuntoniAmanda WallaceSMA REACH UK networkThe Revised Hammersmith Scale (RHS) for Spinal Muscular Atrophy (SMA) was designed as a psychometrically robust clinical outcome assessment to assess physical abilities of patients with type 2 and 3 SMA. The reliability properties of the RHS have not yet been reported. A prospective RHS reliability study was undertaken in a UK cohort of experienced neuromuscular paediatric Physiotherapists. Reliability testing was conducted via a virtual survey platform two weeks apart. Through the virtual platform participants scored videos of two RHS assessments, one of a child with SMA 2 and one of a child with SMA 3. Inter and intra-rater reliability was analysed using a type 3 Intraclass Correlation Coefficient (ICC). Intra-rater agreement was further analysed using Bland Altman (BA) Limits of Agreement (LOA) and plots. The acceptable inter and intra-rater variability was set as a change of ± 2 by the international team of expert physiotherapists who developed the RHS. Inter-rater agreement, n = 22 raters, type 3 ICC was 0.989 (95% CI 0.944 to 1.00), 97.7% of scores were within the acceptable limits of ± 2 points. Intra-rater agreement, n = 21 raters, type 3 ICC ranged from 0.922 to 1.0, with 97.6% of scores within the acceptable limits of ± 2 points. The mean SMA 2 intra-rater difference was -0.10 (-0.6 to 0.4), with lower LOA -2.24 and upper LOA +2.04. Intra-rater difference between tests for SMA 3 intra-rater difference was -0.05 (-0.6 to 0.5), with lower LOA -2.48 and upper LOA +2.38. Intra-rater scoring precision fell within BA agreement limits of ±2 points. The results demonstrate that the RHS is highly reliable when used by experienced UK physiotherapists, and variability of test scores regarding inter and intra-rater reliability was confirmed to lie within ±2 points.https://doi.org/10.1371/journal.pone.0278996 |
| spellingShingle | Danielle Ramsey Gita Ramdharry Mariacristina Scoto Francesco Muntoni Amanda Wallace SMA REACH UK network Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement. PLoS ONE |
| title | Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement. |
| title_full | Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement. |
| title_fullStr | Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement. |
| title_full_unstemmed | Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement. |
| title_short | Revised Hammersmith Scale for spinal muscular atrophy: Inter and intra-rater reliability and agreement. |
| title_sort | revised hammersmith scale for spinal muscular atrophy inter and intra rater reliability and agreement |
| url | https://doi.org/10.1371/journal.pone.0278996 |
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