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Letter to the editor
The Goal Attainment Scale Refines Patient-Centered Expectations in Botulinum Toxin Treatment of Cervical Dystonia
Pattamon Panyakaew1*corresp_iconorcid, Piyanat Wongwan1*orcid, Roongroj Bhidayasiri1,2orcid
Journal of Movement Disorders 2024;17(4):462-465.
DOI: https://doi.org/10.14802/jmd.24150
Published online: September 23, 2024

1Chulalongkorn Centre of Excellence for Parkinson’s Disease and Related Disorders, Faculty of Medicine, Chulalongkorn University and King Chulalongkorn Memorial Hospital, Thai Red Cross Society, Bangkok, Thailand

2The Academy of Science, The Royal Society of Thailand, Bangkok, Thailand

Corresponding author: Pattamon Panyakaew, MD, MSc Chulalongkorn Centre of Excellence for Parkinson’s Disease and Related Disorders, Faculty of Medicine, Chulalongkorn University and King Chulalongkorn Memorial Hospital, Thai Red Cross Society, 1873 Rama 4 Road, Bangkok 10330, Thailand / Tel: +662-256-4000 / Fax: +662-256-4630 / E-mail: ppa@chulapd.org
*These authors contributed equally to this work.
• Received: June 30, 2024   • Revised: August 24, 2024   • Accepted: September 20, 2024

Copyright © 2024 The Korean Movement Disorder Society

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Dear Editor,
Cervical dystonia (CD) can significantly impact patients’ quality of life (QoL). It manifests with complex motor and nonmotor symptoms, such as pain, depression, anxiety, sleep disturbances, and cognitive impairments [1]. The current validated clinical rating scales include the Toronto Western Spasmodic Torticollis Scale-2 (TWSTR-2), with domains that evaluate motor symptom severity, pain, disability, and psychiatric comorbidities, and the Cervical Dystonia Impact Profile (CDIP-58), which focuses on QoL measurements. However, these scales do not reflect personalized problems and goals, which highlights the need for comprehensive approaches to tailoring individual therapy [2].
The goal attainment scale (GAS) identifies patient-centered goals and evaluates the accomplishment of essential daily activities through systematically defined expected outcome levels. This standardized assessment computes a GAS T-score to determine intervention effectiveness across individuals with different goals [3]. The GAS can enhance the understanding of patients’ roles in setting realistic goals. While this scale is widely used for neurodegenerative diseases, GAS-involving research on CD is limited [4]. This study aimed to investigate the patient-centered outcomes of botulinum toxin A (BoNT-A) injection treatment by employing the GAS for CD patients. The three most disturbing problems and individualized goal settings were defined and prioritized before BoNT-A treatment, and the expected accomplishments were assessed postinjection.
Patients with isolated CD based on the diagnostic criteria, with or without a tremor, who received prior regular BoNT-A injections were enrolled. The baseline characteristics included the number of prior BoNT-A injections, the BoNT-A equivalent doses (converted to onabotulinum toxin type A units), the CD type (simple CD, with a head deviation in one plane, vs. complex CD, with a head deviation in more than one plane, or a combination of rotations), and the presence of a head tremor. All participants completed the CDIP-58 questionnaire, which could assist in forming an overview of motor and nonmotor CD symptoms before the GAS was established [2].
Before the BoNT-A injection, each participant defined and prioritized their three most disturbing CD-related problems and set the details of their own treatment goals via open-ended queries. For each problem/goal content, the expected level of the BoNT-A accomplishment was quantified via a five-point scale, ranging from -2 to +2 (-2 = the most unfavorable outcome; -1 = mild improvement but not meeting expectations; 0 = the expected level of achievement; +1 = slightly greater than the expected level of achievement; and +2 = the best anticipated successful outcome). The goals were weighted on the basis of the problem’s importance and the difficulty of its achievement, for which the equal weight was set to Wi = 1 in this study. The attainment levels were combined into a single GAS T-score, as shown in the formula below [3]:
GAS T-score = 50 +10Σ(WiXi)(0.7ΣWi2 + 0.3(ΣWi)2),
where Xi is the numerical value achieved for each problem (between -2 and +2). At 6 weeks after the BoNT-A injection, which corresponded to the peak clinical benefit, the participants assessed their goal achievement for each problem, and the results were combined into the overall GAS T-score, with an example shown in Figure 1A. A GAS T-score of 50 indicated the expected accomplishment, while a GAS T-score of >50 or <50 represented better or worse than expected outcomes, respectively.
A total of 22 patients with isolated CD (8 males and 14 females), with a mean of 10.9 cycles (5.6) of BoNT-A injections, were enrolled (Supplementary Table 1 in the online-only Data Supplement). Most patients (16/22, 72.7%) were identified as having complex CD. Head tremor was present in 15 patients (68.2%). A total of 66 problems and goals (3 per patient) were identified. The most disturbing problems from the patient’s perspective were head tremors (10/22, 45.5%), followed by a loss of confidence (9/22, 40.9%) and neck pain (8/22, 36.4%) (Figure 1B). When categorized on the basis of the CDIP-58 dimensions, head and neck symptoms (27.3%) and pain (21.2%) were the most commonly identified problems. Among patients without head tremor, the loss of confidence was the most disabling issue (4/7, 57.1%).
The mean GAS T-score at 6 weeks post-BoNT-A injection was 57.7 (range: 36.4–77.3, standard deviation = 11.0). A GAS T-score >50 was reported by 59.1% (13/22) of the patients (Figure 1C). Subgroup analysis via the Mann‒Whitney U test revealed that patients achieving a GAS T-score >50 had significantly older age of onset compared to those with a GAS T-score ≤50 (p = 0.036) (Supplementary Table 2 in the online-only Data Supplement). A multiple linear regression model with a stepwise method that included multiple variables, including baseline characteristics and CDIP-58 scores, was applied to evaluate the predictive factors for the GAS T-score after BoNT-A injection. The model was statistically significant (R 2 = 0.371, F (1, 20) = 5.602, p = 0.012) and revealed that the CDIP-58 score and the BoNT-A dose were negatively associated with the GAS T-score (standardized coefficients β = -0.428 and -0.404, p = 0.030 and 0.039, respectively) (Supplementary Table 3 in the online-only Data Supplement).
This study demonstrated the usefulness of the GAS to evaluate personalized problems and treatment goals among CD patients receiving regular BoNT-A injections. Our overall BoNT-A satisfaction rate was consistent with that of a recent GAS study (after at least 4 BoNT-A injection cycles) and with that of our published abstract [4,5]. Head tremor, rather than other motor symptoms, was identified as the most disturbing symptom because of the high prevalence of head tremors (68.2%) in our study compared with their varying occurrences (10%–70%, overall 53%) reported previously [6]. However, this finding aligned with previous results showing that head tremors were related to lower initial GAS T-scores [4]. Head tremor is a troublesome CD symptom that is strongly associated with a reduced BoNT-A response, as a minimal tremor severity improvement was observed over a 3-year follow-up [7]. Accurate injection into deep neck muscles via the use of the collis-capitis concept with ultrasound guidance should be considered to treat dystonic head tremors [4]. Visible head tremors, which can be related to abnormal postures, limitations in daily activities, and psychological comorbidities, may diminish self-confidence [1]. Pain has been reported to be associated with a younger age of onset, supporting our finding that a younger age of CD onset was associated with lower satisfactory outcomes [1]. A greater severity of CD, which affects the QoL and requires higher doses of BoNT-A, is a negative predictor of achievement, indicating that more complex CD is likely to be related to less satisfaction [7].
Limitations of this study include a relatively small sample size, which limits the interpretability of the subgroup analysis results. Furthermore, our recruited patients received more than ten BoNT-A injections, which may have introduced bias in goal selection and accomplishment. Finally, we did not measure the TWSTR-2 scale scores since we originally focused on patients’ QoL. Applying the TWSTR-2 scale to validate the GAS results in a larger CD cohort with BoNT-A naive patients would be interesting.
Our study highlights the usefulness of the GAS for comprehensively evaluating individual functional disabilities and BoNT-A treatment expectations in patients with CD. As a patient-centered tool that extends beyond clinical rating scales, the GAS represents a promising methodology to address individual needs and improve the therapeutic benefits and overall satisfaction with BoNT-A injections.
The online-only Data Supplement is available with this article at https://doi.org/10.14802/jmd.24150.
Supplementary Table 1.
Clinical demographics and clinical characteristics of CD patients (n = 22)
jmd-24150-Supplementary-Table-1.pdf
Supplementary Table 2.
Subgroup analysis of clinical characteristics in CD patients with GAS T-score ≤50 (below or equal to expectation) and GAS T-score >50 (above the expectation) 6 weeks after BoNT-A injection
jmd-24150-Supplementary-Table-2.pdf
Supplementary Table 3.
Results from the multiple linear regression model with a stepwise method that analyzed the predictive factors of GAS T-score after BoNT-A injection
jmd-24150-Supplementary-Table-3.pdf

Ethics Statement

All procedures performed in studies involving human participants were by the ethical standards of the institutional and national research committee and with the 1975 Helsinki Declaration and its later amendments or comparable ethical standards. Informed consent was obtained from all the patients included in the study. The local Ethics Committee approved the study (IRB No. 0959/64).

Conflicts of Interest

The authors have no financial conflicts of interest.

Funding Statement

This study is funded by the Thai Red Cross Education and Research Committee and a Center of Excellence grant from Chulalongkorn University, Bangkok, Thailand (GCE 3300160003). Roongroj Bhidayasiri is supported by a Senior Research Scholarship from the Thailand Science Research and Innovation (TSRI) Agency (RTA6280016).

Author Contributions

Conceptualization: all authors. Data curation: Piyanat Wongwan. Formal analysis: Pattamon Panyakaew, Piyanat Wongwan. Funding acquisition: Roongroj Bhidayasiri. Investigation: Pattamon Panyakaew, Piyanat Wongwan. Methodology: all authors. Project administration: Piyanat Wongwan. Resources: Pattamon Panyakaew, Piyanat Wongwan. Software: Pattamon Panyakaew, Piyanat Wongwan. Supervision: Pattamon Panyakaew, Roongroj Bhidayasiri. Validation: Pattamon Panyakaew, Roongroj Bhidayasiri. Writing—original draft: Pattamon Panyakaew, Piyanat Wongwan. Writing—review & editing: Pattamon Panyakaew, Roongroj Bhidayasiri.

None
Figure 1.
Method for GAS-T score calculation, the most troublesome problems and proportion of patients with different GAS-T score categories. A: An example of 3 personal and prioritized goals of treatment (blue box) after BoNT-A injection and calculation of GAS T-score (outcome scores were +1, +2, -2; Wi = 1, GAS T-score = 54.54). B: The most disturbing problems of cervical dystonia patients at baseline. C: The proportion of cervical dystonia patients with different categories of GAS T-score 6 weeks after treatment with BoNT-A injection. GAS, goal attainment scale; BoNT-A; botulinum toxin A.
jmd-24150f1.jpg
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  • 7. Colosimo C, Charles D, Misra VP, Maisonobe P, Om S. How satisfied are cervical dystonia patients after 3 years of botulinum toxin type A treatment? Results from a prospective, long-term observational study. J Neurol 2019;266:3038–3046.ArticlePubMedPMCPDF

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      The Goal Attainment Scale Refines Patient-Centered Expectations in Botulinum Toxin Treatment of Cervical Dystonia
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      Figure 1. Method for GAS-T score calculation, the most troublesome problems and proportion of patients with different GAS-T score categories. A: An example of 3 personal and prioritized goals of treatment (blue box) after BoNT-A injection and calculation of GAS T-score (outcome scores were +1, +2, -2; Wi = 1, GAS T-score = 54.54). B: The most disturbing problems of cervical dystonia patients at baseline. C: The proportion of cervical dystonia patients with different categories of GAS T-score 6 weeks after treatment with BoNT-A injection. GAS, goal attainment scale; BoNT-A; botulinum toxin A.
      The Goal Attainment Scale Refines Patient-Centered Expectations in Botulinum Toxin Treatment of Cervical Dystonia

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