What is the evidence behind Soliris (eculizumab) for myasthenia gravis?

Comment by InpharmD Researcher

Eculizumab (Soliris®) was found to be safe and effective for treatment of acetylcholine receptor antibody-positive myasthenia gravis (AChR-Ab+ MG) in the phase III REGAIN trial (Table 1) and in an open-label extension of REGAIN (Table 2). Meta-analyses have also found eculizumab to be superior in reducing qualitative myasthenia gravis scores compared to alternative treatments. International Consensus Guidance for Management of Myasthenia Gravis recommends eculizumab be considered in the treatment of severe, refractory, AChR-Ab+ MG; however, future research is needed to assess the optimal duration of eculizumab therapy and its efficacy in other myasthenia gravis populations and stages.

Background

According to a 2020 update of the International Consensus Guidance for Management of Myasthenia Gravis, eculizumab, a humanized monoclonal antibody against the terminal C5 complement molecule, should be considered in the treatment of severe, refractory, acetylcholine receptor positive myasthenia gravis (AChR-Ab+ MG). The role of eculizumab in the treatment of MG is suggested to evolve over time, but until further data become available to allow comparisons of cost and efficacy with other treatments, eculizumab should be considered after trials of other immunotherapies have been unsuccessful in meeting treatment goals. Recommendations of the Advisory Committee on Immunization Practices or other local guidelines regarding immunization against meningococcal meningitis should be followed prior to treatment with eculizumab. Vaccination against Neisseria meningitidis (both meningococcal conjugate Men ACWY and serogroup B or MenB) is required at least 2 weeks before starting treatment with eculizumab. Antibiotic coverage, for at least 4 weeks after immunization, is recommended if eculizumab is started before the 2-week period after vaccination. It is recommended that future research include assessment of the duration of eculizumab therapy necessary to achieve and maintain treatment goals, its efficacy in other MG populations (i.e., MG with thymoma and seronegative MG), and in other stages of disease (i.e., MG crises, exacerbations, and early therapy in non-refractory AChR-Ab+ MG). [1], [2]

This guideline update also provides a review of data regarding the use of eculizumab for treatment of MG. Through its prevention of the formation of the membrane attack complex, eculizumab reduces damage caused by complement-fixing acetylcholine receptor antibodies. In a phase II crossover randomized controlled trial (RCT) of 14 patients with refractory generalized AChR-Ab+ MG, 6/7 (86%) of eculizumab-treated patients achieved the primary end-point of a 2 point reduction in the quantitative MG score (QMG score) at the end of the first treatment period, compared to 57% with placebo. Additionally, a repeated measures mixed model of data from all visits found significant differences in the QMGS favoring eculizumab. Eculizumab treatment was also well tolerated. [1]

While there was no difference in the primary outcome measure of change in the MG-activity of daily living (MG-ADL) score from baseline to week 26 between eculizumab and placebo in a phase III, international, multicenter RCT of patients with generalized nonthymomatous AChR-Ab+ MG (REGAIN trial); the QMG score change on worst-rank analysis of covariance, and all prespecified secondary endpoints, and multiple sensitivity analyses showed a significant benefit for eculizumab (see Table 1). Participants who completed the 26-week REGAIN study were followed in an open-label extension (OLE) within 2 weeks of completing REGAIN (see Table 2). A preplanned interim analysis of the OLE at 22.7 months of median follow-up found a reduction in MG exacerbations by 75% compared with the year before REGAIN, and 56% of patients achieved minimal manifestation status or pharmacologic remission. Eculizumab was well tolerated despite one case of meningococcal meningitis occurring that was successfully treated. [1]

A 2019 network meta-analysis compared and ranked immunotherapies including immunosuppressant agents or monoclonal antibodies for MG. A total of 14 studies (N= 808 MG patients) were included for analysis. In a traditional pairwise analysis, eculizumab had a significant mean reduction of QMGS versus placebo of -0.8 (95% confidence interval [CI] -1.37 to -0.23). In the surface under the cumulative ranking curve (SUCRA) analysis, eculizumab was ranked second with a significant reduction in QMGS versus placebo of -0.75 (95% CI -1.33 to -0.3). Cyclosporine A was ranked first with a significant reduction in QMGS versus placebo of -1.18 (95% CI -1.81 to -0.59). After controlling for the intervention periods, eculizumab reached a significant reduction in QMGS of -1.5 (95% CI -2.81 to -0.18) versus placebo. Eculizumab and belimumab were ranked the most tolerable therapies causing the least counts of adverse events. This analysis is limited due to the overall quality of included studies being moderate to low; however, the authors suggest eculizumab represented the most effective therapeutic alternative to improve QMGS while maintaining good tolerability. Eculizumab may be recommended for refractory MG patients. [2]

Another recent meta-analysis aimed to evaluate drug efficacy and identify associated factors that affect QMGS and MG-ADLs in patients with MG. A total of 13 trials (N= 673) that evaluated immunosuppressants and targeted therapies (i.e., eculizumab) were eligible for analysis. A pharmacodynamic model found eculizumab to have the highest efficacy in reducing QMGs scores (3.66 points); however, efgartigimod had the highest efficacy in reducing MG-ADLs scores (1.97 points). [3]

Finally, a 2019 network meta-analysis compared and ranked immunotherapies including immunosuppressant agents or monoclonal antibodies for MG. A total of 14 studies (N= 808 MG patients) were included for analysis. In a traditional pairwise analysis, eculizumab had a significant mean reduction of QMGS versus placebo of -0.8 (95% CI -1.37 to -0.23). In the surface under the cumulative ranking curve (SUCRA) analysis, eculizumab was ranked second with a significant reduction in QMGS versus placebo of -0.75 (95% CI -1.33 to -0.3). Cyclosporine A was ranked first with a significant reduction in QMGS versus placebo of -1.18 (95% CI -1.81 to -0.59). After controlling for the intervention periods, eculizumab reached a significant reduction in QMGS of -1.5 (95% CI -2.81 to -0.18) versus placebo. Eculizumab and belimumab were ranked the most tolerable therapies causing the least counts of adverse events. This analysis is limited due to the overall quality of included studies being moderate to low; however, the authors suggest eculizumab represented the most effective therapeutic alternative to improve QMGS while maintaining good tolerability. Eculizumab may be recommended for refractory MG patients. [4]

References: [1] Narayanaswami P, Sanders DB, Wolfe G, et al. International Consensus Guidance for Management of Myasthenia Gravis: 2020 Update. Neurology. 2021;96(3):114-122. doi:10.1212/WNL.0000000000011124
[2] Jiao L, Li H, Guo S. Eculizumab treatment for myasthenia gravis subgroups: 2021 update. J Neuroimmunol. 2022;362:577767. doi:10.1016/j.jneuroim.2021.577767
[3] Chen R, Zhang N, Gao L, et al. Quantitative evaluation of drug efficacy in the treatment of myasthenia gravis. Expert Opin Investig Drugs. 2021;30(12):1231-1240. doi:10.1080/13543784.2021.2010704
[4] Wang L, Huan X, Xi JY, et al. Immunosuppressive and monoclonal antibody treatment for myasthenia gravis: A network meta-analysis. CNS Neurosci Ther. 2019;25(5):647-658. doi:10.1111/cns.13110
Relevant Prescribing Information

Clinical Studies: The efficacy of Soliris for the treatment of gMG was established in a 26-week randomized, double-blind, parallel-group, placebo-controlled, multi-center trial (NCT01997229). [5]

References: [5] Eculizumab (Soliris) [prescribing information]. Boston, MA: Alexion Pharmaceuticals Inc.; 2021
Literature Review

A search of the published medical literature revealed 4 studies investigating the researchable question:

What is the evidence behind Soliris (eculizumab) for myasthenia gravis?

Level of evidence

A - Multiple high-quality studies with consistent results  Read more→



Please see Tables 1-4 for your response.


 

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

Design

Prospective, randomized, double-blind, placebo-controlled, multicenter, phase 3 trial

N= 125

Objective

To assess the efficacy and safety of eculizumab in patients with anti-acetylcholine receptor antibody-positive refractory generalized myasthenia gravis

Study Groups

Eculizumab (n= 62)

Placebo (n= 63)

Inclusion Criteria

Age ≥ 18 years, confirmed generalized myasthenia gravis, positive for anti-acetylcholine receptor antibodies, impaired activities of daily living defined by Myasthenia Gravis-Activities of Daily Living (MG-ADL) score 6 or higher, class II-IV disease according to Myasthenia Gravis Foundation of America (MGFA) classification system, received treatment with two or more immunosuppressive therapy or at least one immunosuppressive therapy with intravenous (IV) immunoglobulin or plasma exchange given at least four times per year, for 12 months without symptom control

Exclusion Criteria

History of thymic neoplasms, thymectomy within 12 months prior to screening, ocular myasthenia gravis, myasthenic crisis, use of IV immunoglobulin or plasma exchange within 4 weeks prior to randomization, rituximab use within 6 months prior to screening

Methods

Patients were randomized (1:1) to receive either intravenous eculizumab 900 mg induction dose on day 1 and weeks 1, 2, and 3; 1,200 mg at week 4; and maintenance dosing 1,200 mg every second week thereafter or a matching placebo. Treatment lasted up to 26 weeks. Patients on previous maintenance medications (e.g. cholinesterase inhibitors, corticosteroids, other immunosuppressive treatments) were allowed to continue medication so long as they were maintained. Rescue medications were allowed under protocol-defined conditions at the investigator's discretion. 

After treatment, patients were allowed to enroll in an ongoing extension, open-label study.

Duration

Up to 38 weeks; 8-week follow-up for patients who discontinued prematurely 

Outcome Measures

Primary: Change in MG-ADL total score from baseline to week 26 measured by the worst-rank ANCOVA score

Secondary: Change in qualitative (QMG) total score; responder analysis of MG-ADL and Myasthenia Gravis Composite (MGC) score; change in MGC total score; change in 15-item Myasthenia Gravis Quality of Life (MG-QOL15) total score

Safety: Hospital admission, discontinuation due to adverse events, exacerbations of myasthenia gravis, rescue therapy needed, common adverse events

Baseline Characteristics

 

Eculizumab (n= 62)

Placebo (n= 63)

   

Age, years

47.5  46.9    

Female

66% 65%    

White

85% 67%    

Body mass index, kg/m2

31.4 30.5    

Myasthenia gravis duration, years

9.9 9.2    

MG-ADL score

QMG score

MGC score

MG-QOL15 score

10.5

17.3

20.4

33.6

9.9

16.9

18.9

30.7

   

MGFA classification by randomization stratification

Class IIa or IIIa

Class IVa

Class IIb or IIIb

Class IVb

 

48%

6%

40%

5%

 

51%

3%

41%

5%

   

Previous use of immunosuppressive treatments

≥ 3

≥ 2

 

50%

98%

 

54%

98%

   

Results

Endpoint

Eculizumab (n= 62)

Placebo (n= 63)

Difference (95% confidence interval [CI])

p-Value

Prespecified worst-rank ANCOVA score

MG-ADL (primary outcome)

QMG

MCG

MG-QOL15

 

56.6

54.7

57.3

55.5

 

68.3

70.7

67.7

69.7

 

-11.7 (-24.3 to 0.96)

-16.0 (-28.5 to -3.4)

-10.5 (-23.1 to 2.1)

-14.3 (27.0 to -1.6)

 

0.0698

0.0129

0.1026

0.0281

Admissions to hospital

15% 29%    

Discontinuation due to adverse events

6% 0    

Myasthenia gravis exacerbations

10% 24%    

Rescue therapy during the 26-week treatment period

High-dose corticosteroids

Plasmapheresis or plasma exchange

Intravenous immunoglobulin

Other

10%

0

5%

6%

2%

19%

8%

6%

10%

3%

   

Most common adverse events (> 10% in either group)

Headache

Upper respiratory tract infection

Nasopharyngitis

Nausea

Diarrhea

Myasthenia gravis

 

16%

16%

15%

13%

13%

10%

 

19%

19%

16%

14%

13%

17%

   

Adverse Events

Serious adverse events: 15% in the eculizumab group and 29% in the placebo group. Most frequent serious adverse event is infection (3% in the eculizumab group vs. 10% in the placebo group).

No deaths or meningococcal infections were reported.

Study Author Conclusions

The change in the MG-ADL score was not statistically significant between eculizumab and placebo, as measured by the worst-rank analysis. Eculizumab was well tolerated. The use of a worst-rank analytical approach proved to be an important limitation of this study since the secondary and sensitivity analyses results were inconsistent with the primary endpoint result; further research into the role of complement is needed.

InpharmD Researcher Critique

The worst-rank analytical approach automatically assigns all patients who discontinued study into the poor-outcome group, regardless of the reason for discontinuation. While a bold method to demonstrate the benefits of an experimental agent, the authors noted this led to inconsistencies in secondary analyses which presents possible disparities in the presented results. The differences between eculizumab and placebo might be different had the authors defined treatment failure differently.



References:
[1] Howard JF Jr, Utsugisawa K, Benatar M, 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 [published correction appears in Lancet Neurol. 2017 Dec;16(12 ):954]. Lancet Neurol. 2017;16(12):976-986. doi:10.1016/S1474-4422(17)30369-1

 

Concomitant Immunosuppressive Therapy Use in Eculizumab-Treated Adults With Generalized Myasthenia Gravis During the REGAIN Open-Label Extension Study

Design

Open-label extension of the REGAIN study

N= 117

Objective

To examine changes in the use of immunosuppressive therapies (ISTs), including prednisone and related corticosteroids (PRED), azathioprine (AZA), and mycophenolate mofetil (MMF), in patients receiving eculizumab during the open-label extension (OLE) of the REGAIN study

Study Groups

Study participants (N= 117)

PRED (n= 90)

AZA (n= 39)

MMF (n= 30)

Inclusion Criteria

Age ≥ 18 years, refractory anti-acetylcholine receptor (AChR) antibody-positive plus generalized myasthenia gravis (gMG), MG Activities of Daily Living (MG-ADL) total score ≥ 6, refractory disease, received two or more ISTs for at least 1 year or one or more ISTs with intravenous immunoglobulin or plasma exchange treatment at least four times in 1 year, without symptom control

 +Exclusion Criteria

History of thymic neoplasms, thymectomy within 12 months prior to screening, ocular myasthenia gravis, myasthenic crisis, use of IV immunoglobulin or plasma exchange within 4 weeks prior to randomization, rituximab use within 6 months prior to screening

Methods

Patients who completed REGAIN could be enrolled into the OLE within 2 weeks to receive open-label eculizumab for up to a max of 4 years. The extended treatment consisted of eculizumab 1,200 mg every 2 weeks for up to 4 years after a 4-week blinded induction period. Patients on previous IST must continue and be maintained on their regimen, with modifications permitted per the investigator's discretion.

Duration

Initial enrollment: April 30, 2014

Completed January 2019

Follow-up: 4 years

Outcome Measures

Changes in the use of IST, changes in MG-ADL and QMG scores from baseline to OLE last assessment, treatment-emergent adverse events

Baseline Characteristics

 

PRED (n= 90)

AZA (n= 39)

MMF (n= 30)

Age, years

48.3 46.7 49.4

Female

62.2% 64.1% 70%

White

74.4% 87.2% 86.7%

Duration of myasthenia gravis (MG), years

9.87 9.67 10.21

Myasthenia Gravis Foundation of America classification

IIa or IIIa

IVa

IIb or IIIb

IVb

 

52.2%

3.3%

40%

4.4%

 

53.8%

7.7%

33.3%

5.1%

 

60%

3.3%

33.3%

3.3%

Prior IST use

2 ISTs

3 ISTs

≥ 4 ISTs

 

46.7%

30%

22.2%

 

82.1%

12.8%

5.1%

 

30%

46.7%

20%

Prior intravenous immunoglobulin use

77.8%

74.4%

80%

Prior plasma exchange use

43.3%

43.6%

56.7%

Results

Endpoint

Study participants (N= 117)

 

 

Change in IST use

Decrease in daily dose of 1 IST

Increase in daily dose of 1 IST

Stopped an existing IST

Started a new IST

Increase in daily dose of > 1 IST

Decrease in daily dose of > 1 IST

76.9%

63.2%

43.6%

43.6%

31.6%

2.6%

1.7%

   

Stoppage or decrease in dose of > 1 IST

MG symptoms improved

MG symptoms worsened

New indication other than MG for IST use

Side effects - Intolerant to existing IST

Other

71.8%

48.7%

2.6%

6%

15.4%

40.2%

   

Start or increase in dose of > 1 IST

MG symptoms worsened

New indication other than MG for ISt use

Other

60.7%

37.6%

10.3%

36.8%

   

Median duration of eculizumab treatment, days (range)

972 (1 to 1,372)

   
 

PRED (n= 90)

AZA (n= 39) MMF (n= 30)

Change in MG-ADL score from baseline to OLE last assessment

Decreased and/or stopped IST

No change in IST

Increased and/or stopped IST

 

-4.7

-2.3

-0.7

 

-3.4

-4.7

0.3

 

-5.1

-2.5

-5.3

Change in QMG total score from baseline to OLE last assessment

Decreased and/or stopped IST

No change in IST

Increased and/or stopped IST

 

-5.6

-1.5

0.2

 

-3.8

-5.1

-2.7

 

-4.9

-1.6

-7.9

Treatment-emergent adverse events occurring in > 15% of all patients

Headache

Nasopharyngitis

Diarrhea

Myasthenia gravis

Upper respiratory tract infection

Arthralgia

Cough

Influenza

Nausea

Urinary tract infection

Pain in extremity

 

37.8%

37.8%

18.9%

25.6%

23.3%

20%

14.4%

16.7%

17.8%

10%

15.6%

 

43.6%

20.5%

35.9%

25.6%

38.5%

25.6%

20.5%

23.1%

23.1%

10.3%

20.5%

 

30%

30%

23.3%

40%

20%

16.7%

16.7%

20%

20%

20%

10%

Adverse Events

One meningococcal infection occurred which was resolved with antibiotics.

Three deaths were reported.

Study Author Conclusions

Use of ISTs by patients with previously refractory gMG decreased during eculizumab treatment in the REGAIN OLE. Clinical improvements with eculizumab were maintained by patients in all groups, including those who decreased and/or stopped concomitant ISTs.

InpharmD Researcher Critique

There was no protocol regarding the adjustment of IST during the OLE period. As an international study that took place in various continents, the management of myasthenia gravis can vary greatly between patients. No formal analysis was performed, as the goal of the study is to observe long-term results.



References:
[1] Nowak RJ, Muppidi S, Beydoun SR, O'Brien FL, Yountz M, Howard JF Jr. Concomitant Immunosuppressive Therapy Use in Eculizumab-Treated Adults With Generalized Myasthenia Gravis During the REGAIN Open-Label Extension Study. Front Neurol. 2020;11:556104. Published 2020 Nov 24. doi:10.3389/fneur.2020.556104

 

Long-term efficacy of eculizumab in refractory generalized myasthenia gravis: responder analyses

Design

Retrospective analysis of responders from REGAIN and its open-label extension (OLE)

N= 98

Objective

To analyze response profiles in adult patients with anti-acetylcholine receptor antibody-positive refractory generalized myasthenia gravis (gMG) treated with eculizumab in the REGAIN study or its OLE

Study Groups

Responders (N= 98)

Inclusion Criteria

Participant in REGAIN or its OLE

Exclusion Criteria

Participants who did not participate in REGAIN or its OLE

Methods

Response profiles of participants in REGAIN and its OLE were explored according to myasthenia gravis-activities of daily living (MG-ADL) and qualitative MG scores recorded during the studies. Response was defined as a ≥ 3 point reduction in the MG-ADL total score or a ≥ 5 point reduction in the QMG total score from the start of eculizumab baseline.

Early responders were defined as patients in whom a response was seen on or before the week 12 assessment; late responders were defined as those in whom a response was seen after Week 12 through to the final assessment in the OLE; non-responders were defined as patients in whom no response was observed up to and including their last assessment in the OLE.

Duration

REGAIN: 26 weeks

OLE: 208 weeks

Outcome Measures

Percentage of MG-ADL and QMG early, late, and non-responders; time to MG-ADL and QMG response; change from baseline in MG-ADL and QMG total score over time for early and late responders

Baseline Characteristics

See Tables 1 and 2 for REGAIN trial and its OLE

Results

Endpoint

Responders (N= 98)

MG-ADL response

Early

Late

Non-response

 

66 (67.3%)

17 (17.3%)

15 (15.3%)

QMG response

Early

Late

Non-response


55 (56.1%)

15 (15.3%)

28 (28.6%)

LSM change from baseline in MG-ADL at week 130 (95% CI)

Early responders

Late responders


-61.9% (-69.9% to -53.9%)

-47.5% (-59% to -36%)

LSM change from baseline in QMG at week 130 (95% CI)

Early responders

Late responders


-40.8% (-48.3% to -33.4%)

-55.5% (-68.4% to -42.7%)

Among the early responders according to MG‐ADL score, significant improvement in the corresponding total score (LSM percentage change from baseline −27.3%; 95% CI −34.6% to −20%) was observed by Week 1. Early responders as assessed by QMG scores also achieved a statistically significant improvement in the corresponding total score (LSM percentage change from baseline −20.8%; 95% CI −26.2% to −15.5%) by Week 1.

LSM, least-squares mean; CI, confidence interval

Adverse Events

Common Adverse Events: No clear differences were observed in the adverse‐event incidences for early and late responders using either the MG‐ADL or QMG response definitions.

Serious Adverse Events: 59.7 per 100 person-years for MG-ADL early responders and 45.9 per 100 person-years for MG-ADL late responders; 52.3 per 100 person-years for QMG early responders and 62.8 per 100 person-years for QMG late responders

Percentage that Discontinued due to Adverse Events: N/A

Study Author Conclusions

The findings suggest that, although most patients with refractory gMG will achieve clinical response by Week 12 of eculizumab treatment, first responses can be observed with longer‐term treatment.

InpharmD Researcher Critique

Response was defined by the achievement of a qualifying score at a single time point during treatment, which does not incorporate durability of response. Additionally, some variability was reported in the number of assessments per patient over time and small patient numbers in some analysis groups, which was related to a reduction in frequency of assessments conducted later in the trial. This may have limited ability to analyze patient fluctuations in response stability over time. The open-label design and lack of placebo comparator in the OLE could have potentially led to higher responder rates over time.



References:
[1] Howard JF Jr, Karam C, Yountz M, O'Brien FL, Mozaffar T; REGAIN Study Group. Long-term efficacy of eculizumab in refractory generalized myasthenia gravis: responder analyses. Ann Clin Transl Neurol. 2021;8(7):1398-1407. doi:10.1002/acn3.51376

 

Post-intervention Status in Patients With Refractory Myasthenia Gravis Treated With Eculizumab During REGAIN and Its Open-Label Extension

Design

Analysis of the tertiary endpoint of Myasthenia Gravis Foundation of America (MGFA) post-intervention status and safety data during REGAIN (phase 3, randomized, placebo-controlled study) and the open-label extension study 

N= 117

Objective

To evaluate whether eculizumab helps patients with anti-acetylcholine receptor-positive (AChR+) refractory generalized myasthenia gravis (gMG) achieve the MGFA post-intervention status of minimal manifestations (MM) in an assessment of patients’ status throughout REGAIN (Safety and Efficacy of Eculizumab in AChR+ Refractory Generalized Myasthenia Gravis) and its open-label extension

Study Groups

REGAIN study groups:

Placebo/eculizumab (n= 61)

Eculizumab/eculizumab (n= 56)

Inclusion Criteria

Completed the REGAIN randomized controlled trial; continued into the open-label extension

Exclusion Criteria

Patients who were not assessed for MGFA post-intervention status at a particular time point were not included in the analysis for that time point

Methods

Patients were included in the open-label study within 2 weeks of completing REGAIN study. Inclusion and exclusion criteria of the patients (for REGAIN and open-label study are provided in Tables 1 and 2, respectively). MGFA post-intervention status following administration of eculizumab or placebo during REGAIN, including achievement of MM, was assessed at weeks 4, 12, and 26 of REGAIN and weeks 26, 40, 52, 78, 104, and 130 of the open-label study and was based on comparison with patients’ disease state before REGAIN. 

Patients were assessed for the MGFA post-intervention status of improved, unchanged, worse, MM, and pharmacologic remission at defined time points during REGAIN and through week 130 of the open-label study. Patients with improved status included patients who achieved MM or pharmacological remission (PR). In patients with improved status, MM was achieved if they had no symptoms indicating functional limitations from MG but had some weakness on examination of some muscles.

Duration

REGAIN: 26 weeks

Open-label extension: 208 weeks

Outcome Measures

Change in MGFA status by treatment group, change in MGFA status by eculizumab treatment duration

Baseline Characteristics

See Tables 1 and 2 for REGAIN trial and its open-label extension

Results

Endpoint

Placebo/eculizumab (n= 61)

Eculizumab/eculizumab (n= 56)  

Change in MGFA by treatment group

Improved  MM  Unchanged  Worse  Improved  MM 

Unchanged 

Worse 

REGAIN

Week 4

Week 12

Week 26

 

15/61 (24.6%)

22/61 (36.1%)

25/60 (41.7%)

 

5/61 (8.2%)

7/61 (11.5%)

8/60 (13.3%)

 

41/61 (67.2%)

35/61 (57.4%)

30/60 (50.0%)

 

5/61 (8.2%)

4/61 (6.6%) 

5/60 (8.3%)

 

30/55 (54.5%)

28/53 (52.8%)

34/56 (60.7%) 

 

10/55 (18.2%)

11/53 (20.8%)

14/56 (25.0%) 

 

25/55 (45.5%)

24/53 (45.3%)

21/56 (37.5%) 

 

0/55 (0.0%) 

1/53 (1.9%)

1/56 (1.8%)

Open-label

Week 26

Week 40

Week 52

Week 78

Week 104

Week 130

Last assessment

 

40/56 (71.4%)

46/54 (85.2%)

44/54 (81.5%)

42/48 (87.5%)

34/37 (91.9%) 

33/35 (94.3%)

47/57 (82.5%)

 

27/56 (48.2%)

34/54 (63.0%)

31/54 (57.4%)

29/48 (60.4%)

22/37 (59.5%)

22/35 (62.9%)

31/57 (54.4%)

 

15/56 (26.8%)

7/54 (13.0%)

10/54 (18.5%)

5/48 (10.4%)

2/37 (5.4%)

2/35 (5.7%)

9/57 (15.8%)

 

1/56 (1.8%)

1/54 (1.9%)

0/54 (0.0%)

1/48 (2.1%)

1/37 (2.7%)

0/35 (0.0%)

1/57 (1.8%) 

 

36/48 (75.0%)

37/49 (75.5%)

41/48 (85.4%)

37/47 (78.7%)

33/40 (82.5%)

28/35 (80.0%)

40/56 (71.4%) 

 

22/48 (45.8%)

21/49 (42.9%)

22/48 (45.8%)

22/47 (46.8%)

21/40 (52.5%)

18/35 (51.4%)

23/56 (41.1%)

 

12/48 (25.0%)

10/49 (20.4%)

6/48 (12.5%)

9/47 (19.1%)

6/40 (15.0%)

5/35 (14.3%)

13/56 (23.2%)

0/48 (0.0%)

2/49 (4.1%)

1/48 (2.1%)

1/47 (2.1%)

1/40 (2.5%)

2/35 (5.7%)

3/56 (5.4%) 

Duration of eculizumab treatment, week

Improved MM Unchanged  Worse        

26

52

78

104

130

Last assessment

74/112 (66.1%)

80/102 (78.4%)

83/96 (86.5%)

71/84 (84.5%)

66/75 (88.0%)

87/113 (77.0%) 

41/112 (36.6%)

53/102 (52.0%)

51/96 (53.1%)

44/84 (52.4%)

43/75 (57.3%)

54/113 (47.8%)

36/112 (32.1%)

22/102 (21.6%)

11/96 (11.5%)

11/84 (13.1%)

8/75 (10.7%)

22/113 (19.5%) 

2/112 (1.8%)

0/102 (0.0%)

2/96 (2.1%)

2/84 (2.4%)

1/75 (1.3%)

4/113 (3.5%)  

       

At the time of study completion, patients had participated for different periods of time.

The numbers of patients at each time point reflect this and also account for discontinuations and for patients who did not complete the assessment at the specified time point.

Adverse Events

Common Adverse Events: headache (44.4%), nasopharyngitis (38.5%), diarrhea (28.2%), upper respiratory tract infection (26.5%), nausea (23.1%)

Serious Adverse Events: worsening of MG (15.4%), MG crisis (3.4%)

Percentage that Discontinued due to Adverse Events: During REGAIN and the open-label study, 11 patients discontinued because of an adverse event, and 3 deaths occurred.

Study Author Conclusions

The results of this analysis confirm the rapid and sustained clinical response to eculizumab that was observed during REGAIN and the open-label study. Over 50% of patients who were considered to have refractory disease achieved the consensus treatment goal of MM or better after 1 year of eculizumab treatment. These findings further support the long-term effectiveness of eculizu- mab for use in patients with AChR+ refractory gMG.

InpharmD Researcher Critique

This analysis is limited by the open-label design and lack of a placebo comparator in the extension study; however, since over 90% of patients who enrolled in REGAIN continued into the open-label study, selection bias in the open-label study population is remote.



References:
[1] Mantegazza R, Wolfe GI, Muppidi S, et al. Post-intervention Status in Patients With Refractory Myasthenia Gravis Treated With Eculizumab During REGAIN and Its Open-Label Extension. Neurology. 2021;96(4):e610-e618. doi:10.1212/WNL.0000000000011207