J Clin Neurol.  2019 Jul;15(3):376-385. 10.3988/jcn.2019.15.3.376.

Examining the Impact of Refractory Myasthenia Gravis on Healthcare Resource Utilization in the United States: Analysis of a Myasthenia Gravis Foundation of America Patient Registry Sample

Affiliations
  • 1Department of Health Care Organization and Policy, University of Alabama at Birmingham, Birmingham, AL, USA.
  • 2Alexion Pharmaceuticals, Inc., Boston, MA, USA.
  • 3Department of Biostatistics, University of Alabama at Birmingham, Birmingham, AL, USA. caban@uab.edu

Abstract

BACKGROUND AND PURPOSE
Patients with refractory myasthenia gravis (MG) experience ongoing disease burden that might be reflected in their healthcare utilization. Here we examine the impact of refractory MG on healthcare utilization.
METHODS
The 825 included participants were aged 18-64 years, enrolled in the Myasthenia Gravis Foundation of America Patient Registry between July 2013 and February 2018, and had been diagnosed with MG ≥2 years previously.
RESULTS
Participants comprised 76 (9.2%) with refractory MG and 749 (90.8%) with nonrefractory MG. During the 6 months before enrollment, participants with refractory MG were significantly more likely than those with nonrefractory MG to have experienced at least one exacerbation [67.1% vs. 52.0%, respectively, p=0.01; odds ratio (OR)=1.882, 95% confidence interval (CI)=1.141-3.104], visited an emergency room at least once [43.4% vs. 27.1%, p<0.01; OR=2.065, 95% CI=1.276-3.343], been hospitalized overnight at least once (32.9% vs. 20.5%, p=0.01; OR=1.900, 95% CI=1.140-3.165), ever been admitted to an intensive care unit (ICU) (61.8% vs. 33.4%, p<0.01; OR=3.233, 95% CI=1.985-5.266), or ever required a feeding tube (21.1% vs. 9.1%, p<0.01; OR=2.671, 95% CI=1.457-4.896). A total of 75.8% younger females with refractory disease (<51 years, n=33) experienced at least one exacerbation, 69.7% had been admitted to an ICU, and 30.3% had required a feeding tube. For older females with refractory disease (≥51 years, n=33), 60.6%, 54.6%, and 6.1% experienced these outcomes, respectively (between-group differences were not significant).
CONCLUSIONS
Refractory MG is associated with higher disease burden and healthcare utilization than nonrefractory MG.

Keyword

myasthenia gravis; refractory disease; disease exacerbations; healthcare resource utilization

MeSH Terms

Americas*
Delivery of Health Care*
Emergency Service, Hospital
Female
Humans
Intensive Care Units
Myasthenia Gravis*
Odds Ratio
United States*

Figure

  • Fig. 1 Criteria used for classifying refractory MG. MG-ADL: myasthenia gravis activities of daily living scale.

  • Fig. 2 Flow chart showing selection process for the study sample. MG-ADL: myasthenia gravis activities of daily living scale.

  • Fig. 3 Exacerbations in and healthcare resource utilization by participants with refractory MG and nonrefractory MG (A) and numbers of exacerbations and ER visits during the 6 months before enrollment in participants with refractory and nonrefractory MG (B). Frequencies of study variables were compared between participants with refractory and nonrefractory MG using χ2 tests for categorical variables and the number of exacerbations, and Fisher's exact test for the number of ER visits. p values are for comparisons between the refractory- and nonrefractory-MG groups, and were calculated after excluding data from participants with unknown/missing data. *During the 6 months before enrollment. ER: emergency room, ICU: intensive care unit, MG: myasthenia gravis.

  • Fig. 4 Unadjusted ordinal logistic model examining the impact of refractory MG on exacerbations and healthcare resource utilization. *During the 6 months before enrollment. Analyses were based on fitting logistic models for binary outcomes except for when analyzing the numbers of exacerbations and ER visits, for which proportional-odds models for ordinal outcomes were fitted. CI: confidence interval, ER: emergency room, ICU: intensive care unit, MG: myasthenia gravis, OR: odds ratio.

  • Fig. 5 Exacerbations in and healthcare resource utilization by younger and older female participants with refractory MG (A) and numbers of exacerbations and ER visits during the 6 months before enrollment in younger and older female participants with refractory MG (B). Frequencies of study variables were compared between participants with refractory and nonrefractory MG using χ2 tests for categorical variables and the number of exacerbations, and using Fisher's exact test for the number of ER visits. p values are for comparisons between the refractory- and nonrefractory-MG groups, and were calculated after excluding data from participants with unknown/missing data. *During the 6 months before enrollment. ER: emergency room, ICU: intensive care unit, MG: myasthenia gravis.


Reference

1. Heldal AT, Owe JF, Gilhus NE, Romi F. Seropositive myasthenia gravis: a nationwide epidemiologic study. Neurology. 2009; 73:150–151. PMID: 19597135.
Article
2. Fang F, Sveinsson O, Thormar G, Granqvist M, Askling J, Lundberg IE, et al. The autoimmune spectrum of myasthenia gravis: a Swedish population-based study. J Intern Med. 2015; 277:594–604. PMID: 25251578.
Article
3. Cetin H, Fülöp G, Zach H, Auff E, Zimprich F. Epidemiology of myasthenia gravis in Austria: rising prevalence in an ageing society. Wien Klin Wochenschr. 2012; 124:763–768. PMID: 23129486.
Article
4. Park SY, Lee JY, Lim NG, Hong YH. Incidence and prevalence of myasthenia gravis in Korea: a population-based study using the National Health Insurance claims database. J Clin Neurol. 2016; 12:340–344. PMID: 27165426.
Article
5. Murai H, Yamashita N, Watanabe M, Nomura Y, Motomura M, Yoshikawa H, et al. Characteristics of myasthenia gravis according to onset-age: Japanese nationwide survey. J Neurol Sci. 2011; 305:97–102. PMID: 21440910.
Article
6. Grob D, Brunner N, Namba T, Pagala M. Lifetime course of myasthenia gravis. Muscle Nerve. 2008; 37:141–149. PMID: 18059039.
Article
7. Robertson NP, Deans J, Compston DA. Myasthenia gravis: a population based epidemiological study in Cambridgeshire, England. J Neurol Neurosurg Psychiatry. 1998; 65:492–496. PMID: 9771771.
Article
8. Melzer N, Ruck T, Fuhr P, Gold R, Hohlfeld R, Marx A, et al. Clinical features, pathogenesis, and treatment of myasthenia gravis: a supplement to the Guidelines of the German Neurological Society. J Neurol. 2016; 263:1473–1494. PMID: 26886206.
Article
9. Zieda A, Ravina K, Glazere I, Pelcere L, Naudina MS, Liepina L, et al. A nationwide epidemiological study of myasthenia gravis in Latvia. Eur J Neurol. 2018; 25:519–526. PMID: 29194859.
Article
10. Muppidi S, Wolfe GI, Conaway M, Burns TM. MG Composite and MG-QoL15 Study Group. MG-ADL: still a relevant outcome measure. Muscle Nerve. 2011; 44:727–731. PMID: 22006686.
Article
11. Hoffmann S, Ramm J, Grittner U, Kohler S, Siedler J, Meisel A. Fatigue in myasthenia gravis: risk factors and impact on quality of life. Brain Behav. 2016; 6:e00538. PMID: 27781147.
Article
12. Paul RH, Cohen RA, Goldstein JM, Gilchrist JM. Fatigue and its impact on patients with myasthenia gravis. Muscle Nerve. 2000; 23:1402–1406. PMID: 10951443.
Article
13. Silvestri NJ, Wolfe GI. Treatment-refractory myasthenia gravis. J Clin Neuromuscul Dis. 2014; 15:167–178. PMID: 24872217.
Article
14. Wendell LC, Levine JM. Myasthenic crisis. Neurohospitalist. 2011; 1:16–22. PMID: 23983833.
Article
15. Conti-Fine BM, Milani M, Kaminski HJ. Myasthenia gravis: past, present, and future. J Clin Invest. 2006; 116:2843–2854. PMID: 17080188.
Article
16. Sanders DB, Wolfe GI, Benatar M, Evoli A, Gilhus NE, Illa I, et al. International consensus guidance for management of myasthenia gravis: executive summary. Neurology. 2016; 87:419–425. PMID: 27358333.
17. Suh J, Goldstein JM, Nowak RJ. Clinical characteristics of refractory myasthenia gravis patients. Yale J Biol Med. 2013; 86:255–260. PMID: 23766745.
18. Mantegazza R, Antozzi C. When myasthenia gravis is deemed refractory: clinical signposts and treatment strategies. Ther Adv Neurol Disord. 2018; 11:1756285617749134. PMID: 29403543.
Article
19. Boldingh MI, Dekker L, Maniaol AH, Brunborg C, Lipka AF, Niks EH, et al. An up-date on health-related quality of life in myasthenia gravis-results from population based cohorts. Health Qual Life Outcomes. 2015; 13:115. PMID: 26232146.
Article
20. Utsugisawa K, Suzuki S, Nagane Y, Masuda M, Murai H, Imai T, et al. Health-related quality-of-life and treatment targets in myasthenia gravis. Muscle Nerve. 2014; 50:493–500. PMID: 24536040.
Article
21. Engel-Nitz NM, Boscoe A, Wolbeck R, Johnson J, Silvestri NJ. Burden of illness in patients with treatment refractory myasthenia gravis. Muscle Nerve. 2018; 58:99–105.
Article
22. Murai H, Hasebe M, Murata T, Utsugisawa K. Clinical burden and healthcare resource utilization associated with myasthenia gravis: assessments from a Japanese claims database. Clin Exp Neuroimmunol. 2019; 10:61–68.
Article
23. Wolfe GI, Herbelin L, Nations SP, Foster B, Bryan WW, Barohn RJ. Myasthenia gravis activities of daily living profile. Neurology. 1999; 52:1487–1489. PMID: 10227640.
Article
24. Howard JF Jr, Utsugisawa K, Benatar M, Murai H, Barohn RJ, Illa I, et al. Safety and efficacy of eculizumab in anti-acetylcholine receptor antibody-positive refractory generalised myasthenia gravis (REGAIN): a phase 3, randomised, double-blind, placebo-controlled, multicentre study. Lancet Neurol. 2017; 16:976–986. PMID: 29066163.
25. Lee I, Kaminski HJ, Xin H, Cutter G. Gender and quality of life in myasthenia gravis patients from the myasthenia gravis foundation of America registry. Muscle Nerve. 2018; 58:90–98.
Article
26. Gilhus NE, Nacu A, Andersen JB, Owe JF. Myasthenia gravis and risks for comorbidity. Eur J Neurol. 2015; 22:17–23. PMID: 25354676.
Article
27. Diaz BC, Flores-Gavilán P, García-Ramos G, Lorenzana-Mendoza NA. Myasthenia gravis and its comorbidities. J Neurol Neurophysiol. 2015; 6:1–5. PMID: 26753104.
Article
28. Tanovska N, Novotni G, Sazdova-Burneska S, Kuzmanovski I, Boshkovski B, Kondov G, et al. Myasthenia gravis and associated diseases. Open Access Maced J Med Sci. 2018; 6:472–478. PMID: 29610603.
Article
29. Guptill JT, Marano A, Krueger A, Sanders DB. Cost analysis of myasthenia gravis from a large U.S. insurance database. Muscle Nerve. 2011; 44:907–911. PMID: 22102461.
Article
30. Bertakis KD, Azari R, Helms LJ, Callahan EJ, Robbins JA. Gender differences in the utilization of health care services. J Fam Pract. 2000; 49:147–152. PMID: 10718692.
Full Text Links
  • JCN
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr