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An up-date on health-related quality of life in myasthenia gravis -results from population based cohorts.

Boldingh MI, Dekker L, Maniaol AH, Brunborg C, Lipka AF, Niks EH, Verschuuren JJ, Tallaksen CM - Health Qual Life Outcomes (2015)

Bottom Line: The mean HRQOL score was lower in patients with bulbar and generalized symptoms (p < 0.001) compared to sex and age adjusted healthy controls, but not in patients with ocular symptoms or patients in remission.Multivariate analysis revealed that female gender, generalized symptoms and use of secondary immunosuppressive drugs at the time of testing were risk factors for reduced HRQOL.Historically, the HRQOL levels have not changed since 2001 and no new clinical predictors could be detected in this exhaustive population-based study.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurology, Oslo University Hospital, Ullevål and Rikshospitalet, Ullevål, Pb. 4950 Nydalen, 0424, Oslo, Norway. marbol@ous-hf.no.

ABSTRACT

Unlabelled: Current available therapies control Myasthenia gravis (MG) reasonably well, but Health Related Quality of life (HRQOL) remains lower than expected. The aim was provide insights in how HRQOL in MG stands across borders and time, compare the scores to general population controls and other chronic disorders and assess the impact of potential predictors for quality of life such as a) clinical characteristics b) antibodies c) thymoma and d) treatment in a population-based cohort.

Methods: We designed a population-based cross-sectional study including 858 patients, 373 from Norway and 485 from the Netherlands. The Short Form Health Survey 36 (SF-36) and a cross-cultural validated questionnaire were used. Data were in addition compared to the general population, other chronic diseases and previous studies.

Results: Mean physical composite score was 59.4 and mental composite score 69.0 with no differences between the countries. The mean HRQOL score was lower in patients with bulbar and generalized symptoms (p < 0.001) compared to sex and age adjusted healthy controls, but not in patients with ocular symptoms or patients in remission. Multivariate analysis revealed that female gender, generalized symptoms and use of secondary immunosuppressive drugs at the time of testing were risk factors for reduced HRQOL.

Conclusions: Remission and absence of generalized symptoms were favorable factors for HRQOL in MG patients. Historically, the HRQOL levels have not changed since 2001 and no new clinical predictors could be detected in this exhaustive population-based study. Further studies should explore the impact of non clinical factors like ethnic variations, socio-economic and hormonal factors on HRQOL.

No MeSH data available.


Related in: MedlinePlus

Overview over HRQOL measured by SF-36 from 2001-2012. Studies providing norm-based scoring are shown. Bars illustrate the distribution of MGFA score within the cohorts, lines illustrate the PCS and MCS. Vertical axis shows SF-36 score 0-100 for lines and distribution of MGFA class within the cohort. Padua et al. reported lower scores in 2002 than we did (p < 0.001), however the cohort consisted of fewer patients in remission (7 %) and 89 % in MGFA class II-IV [1]. Paul et al. 2001 [2], provided not norm-based scoring, but PCS 57.6 (27) and 65.5 (24.8) were not significant different from our study
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Fig5: Overview over HRQOL measured by SF-36 from 2001-2012. Studies providing norm-based scoring are shown. Bars illustrate the distribution of MGFA score within the cohorts, lines illustrate the PCS and MCS. Vertical axis shows SF-36 score 0-100 for lines and distribution of MGFA class within the cohort. Padua et al. reported lower scores in 2002 than we did (p < 0.001), however the cohort consisted of fewer patients in remission (7 %) and 89 % in MGFA class II-IV [1]. Paul et al. 2001 [2], provided not norm-based scoring, but PCS 57.6 (27) and 65.5 (24.8) were not significant different from our study

Mentions: The reduced quality of life was determined by lowered physical capacities, but psychological wellbeing was affected in roughly half of the patients as well. There was no differences in HRQOL between MG patients in Norway and the Netherlands. Pooling the data could therefore be done without affecting the validity of the results and confirms the reliability of the results. Put into an historical context, the HRQOL has not changed much for MG patients over the last 10-15 years and we found the same levels as reported in 2001 [2, 5–10] (Fig. 5). The reduced results of Padua et al. [1] may be explained by a low percentage in clinical remission and many patients with generalized symptoms compared to our cohort. The result of “the more generalized disease or active” disease, the poorer HRQOL” is in accordance with other reports [1, 3, 7, 8, 29]. Leonardi et al. [4] reported that patients in remission scored similar to Italian general population, supporting our findings from the Dutch and Norwegian MG patients. We did not find any harmful impact of antibodies or thymectomy in accordance with other studies [1, 8, 30]. Our findings of normal levels of HRQOL in ocular MG is supported by another study of 91 ocular Italian MG patients [11]. In contrast, a Japanese study reported QOL impairment of 123 ocular MG patients in those who not responded to therapy [13]. One explanation of these contradictory results may be patients selection, since ocular MG includes several symptoms (ptosis, diplopia, complete ophthalmoplegia), with potentially different grades of disability. Additionnally, the use of another questionnaire and cultural factors may account for the difference.Fig. 5


An up-date on health-related quality of life in myasthenia gravis -results from population based cohorts.

Boldingh MI, Dekker L, Maniaol AH, Brunborg C, Lipka AF, Niks EH, Verschuuren JJ, Tallaksen CM - Health Qual Life Outcomes (2015)

Overview over HRQOL measured by SF-36 from 2001-2012. Studies providing norm-based scoring are shown. Bars illustrate the distribution of MGFA score within the cohorts, lines illustrate the PCS and MCS. Vertical axis shows SF-36 score 0-100 for lines and distribution of MGFA class within the cohort. Padua et al. reported lower scores in 2002 than we did (p < 0.001), however the cohort consisted of fewer patients in remission (7 %) and 89 % in MGFA class II-IV [1]. Paul et al. 2001 [2], provided not norm-based scoring, but PCS 57.6 (27) and 65.5 (24.8) were not significant different from our study
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4522107&req=5

Fig5: Overview over HRQOL measured by SF-36 from 2001-2012. Studies providing norm-based scoring are shown. Bars illustrate the distribution of MGFA score within the cohorts, lines illustrate the PCS and MCS. Vertical axis shows SF-36 score 0-100 for lines and distribution of MGFA class within the cohort. Padua et al. reported lower scores in 2002 than we did (p < 0.001), however the cohort consisted of fewer patients in remission (7 %) and 89 % in MGFA class II-IV [1]. Paul et al. 2001 [2], provided not norm-based scoring, but PCS 57.6 (27) and 65.5 (24.8) were not significant different from our study
Mentions: The reduced quality of life was determined by lowered physical capacities, but psychological wellbeing was affected in roughly half of the patients as well. There was no differences in HRQOL between MG patients in Norway and the Netherlands. Pooling the data could therefore be done without affecting the validity of the results and confirms the reliability of the results. Put into an historical context, the HRQOL has not changed much for MG patients over the last 10-15 years and we found the same levels as reported in 2001 [2, 5–10] (Fig. 5). The reduced results of Padua et al. [1] may be explained by a low percentage in clinical remission and many patients with generalized symptoms compared to our cohort. The result of “the more generalized disease or active” disease, the poorer HRQOL” is in accordance with other reports [1, 3, 7, 8, 29]. Leonardi et al. [4] reported that patients in remission scored similar to Italian general population, supporting our findings from the Dutch and Norwegian MG patients. We did not find any harmful impact of antibodies or thymectomy in accordance with other studies [1, 8, 30]. Our findings of normal levels of HRQOL in ocular MG is supported by another study of 91 ocular Italian MG patients [11]. In contrast, a Japanese study reported QOL impairment of 123 ocular MG patients in those who not responded to therapy [13]. One explanation of these contradictory results may be patients selection, since ocular MG includes several symptoms (ptosis, diplopia, complete ophthalmoplegia), with potentially different grades of disability. Additionnally, the use of another questionnaire and cultural factors may account for the difference.Fig. 5

Bottom Line: The mean HRQOL score was lower in patients with bulbar and generalized symptoms (p < 0.001) compared to sex and age adjusted healthy controls, but not in patients with ocular symptoms or patients in remission.Multivariate analysis revealed that female gender, generalized symptoms and use of secondary immunosuppressive drugs at the time of testing were risk factors for reduced HRQOL.Historically, the HRQOL levels have not changed since 2001 and no new clinical predictors could be detected in this exhaustive population-based study.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurology, Oslo University Hospital, Ullevål and Rikshospitalet, Ullevål, Pb. 4950 Nydalen, 0424, Oslo, Norway. marbol@ous-hf.no.

ABSTRACT

Unlabelled: Current available therapies control Myasthenia gravis (MG) reasonably well, but Health Related Quality of life (HRQOL) remains lower than expected. The aim was provide insights in how HRQOL in MG stands across borders and time, compare the scores to general population controls and other chronic disorders and assess the impact of potential predictors for quality of life such as a) clinical characteristics b) antibodies c) thymoma and d) treatment in a population-based cohort.

Methods: We designed a population-based cross-sectional study including 858 patients, 373 from Norway and 485 from the Netherlands. The Short Form Health Survey 36 (SF-36) and a cross-cultural validated questionnaire were used. Data were in addition compared to the general population, other chronic diseases and previous studies.

Results: Mean physical composite score was 59.4 and mental composite score 69.0 with no differences between the countries. The mean HRQOL score was lower in patients with bulbar and generalized symptoms (p < 0.001) compared to sex and age adjusted healthy controls, but not in patients with ocular symptoms or patients in remission. Multivariate analysis revealed that female gender, generalized symptoms and use of secondary immunosuppressive drugs at the time of testing were risk factors for reduced HRQOL.

Conclusions: Remission and absence of generalized symptoms were favorable factors for HRQOL in MG patients. Historically, the HRQOL levels have not changed since 2001 and no new clinical predictors could be detected in this exhaustive population-based study. Further studies should explore the impact of non clinical factors like ethnic variations, socio-economic and hormonal factors on HRQOL.

No MeSH data available.


Related in: MedlinePlus