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Neurology Clinical Trial Database

SID-GBS

Second intravenous immunoglobulin dose in patients with Guillain-Barré syndrome with poor prognosis: a double-blind, randomised, placebo-controlled trial

Year of Publication: 2021

Authors: Christa Walgaard, Bart C Jacobs, Hester F Lingsma, ..., et al.; on behalf of the Dutch GBS Study Group

Journal: Lancet Neurology

Citation: Lancet Neurol 2021;20:275-283

Link: https://doi.org/10.1016/S1474-4422(20)30494-4


Clinical Question

Does a second course of intravenous immunoglobulin (SID) improve outcomes in patients with Guillain-Barré syndrome who have a predicted poor prognosis after standard IVIg treatment?

Bottom Line

A second IVIg course in GBS patients with poor prognosis does not provide clinical benefit and is associated with significantly more serious adverse events, including thromboembolic complications. All 4 deaths occurred in the SID group. This trial provides strong evidence against routine use of a second IVIg course in GBS patients with poor prognosis.

Major Points

  • First randomized, placebo-controlled, double-blind trial of second IVIg course in GBS with poor prognosis
  • Primary endpoint not met: No significant difference in GBS disability score at 4 weeks (adjusted OR 1.4; 95% CI 0.6-3.3; P=0.45)
  • No benefit in any secondary outcomes including disability at weeks 8, 12, 26, MRC sumscore, ONLS, or duration of ventilation
  • Serious adverse events significantly higher with SID (51% vs 23%; OR 3.54; P=0.005)
  • Thromboembolic events more common with SID: pulmonary embolism (2 vs 0), coronary ischemia (1 vs 1), asystole (2 vs 0)
  • All 4 deaths occurred in SID group (at 13-24 weeks post-randomization)
  • SID maintained higher serum IgG levels longer (34 vs 17 g/L at 2 weeks) but this did not improve outcomes
  • 85% of patients were still deteriorating at day 7-9 despite standard IVIg, indicating severe disease
  • Prespecified subgroup analyses showed no benefit in any subgroup including those with highest mEGOS or demyelinating subtype
  • Results argue against current practice where ~25% of severe GBS patients receive second IVIg course

Design

Study Type: Phase 3, randomized, double-blind, placebo-controlled trial

Randomization: 1

Blinding: Double-blind; IVIg bags concealed with aluminum foil, opaque connecting lines; patients, outcome adjudicators, monitors, and steering committee masked

Enrollment Period: February 16, 2010 to June 5, 2018

Follow-up Duration: 26 weeks

Centers: 59

Countries: Netherlands

Sample Size: 93

Analysis: Modified intention-to-treat; proportional odds regression with prespecified covariate adjustment for age, preceding diarrhoea, and MRC sumscore at randomization; interim monitoring after 36 randomizations


Inclusion Criteria

  • Age ≥12 years
  • Diagnosis of Guillain-Barré syndrome
  • Indication for IVIg treatment according to treating neurologist
  • Poor prognosis: modified Erasmus GBS Outcome Score (mEGOS) ≥6 at day 7-9 after start of standard IVIg

Exclusion Criteria

  • Good prognosis (mEGOS <6)
  • Known pre-existing vascular risk factors (contraindication for randomization)
  • Full exclusion criteria in appendix

Arms

FieldSecond IVIg Dose (SID)Control
InterventionNanogam 50 mg/mL (Sanquin) 2 g/kg (8 mL/kg) over 5 days, administered 7-9 days after start of standard IVIgAlbumin 4% (Albuman 40 g/L, Sanquin) 8 mL/kg over 5 days, administered 7-9 days after start of standard IVIg
Duration5 days5 days

Outcomes

OutcomeTypeControlInterventionHR / OR / RRP-value
GBS disability score at 4 weeks after start of standard IVIg (7-point scale: 0=no symptoms to 6=death)Primary0.45
GBS disability score at 8 weeks | SID: Median 4 (IQR 3-4); Placebo: Median 4 (IQR 2-4); Adjusted OR: 1.5 (0.7-3.3)Secondary
GBS disability score at 12 weeks | SID: Median 3 (IQR 2-3); Placebo: Median 3 (IQR 2-3); Adjusted OR: 2.1 (0.9-4.6)Secondary
GBS disability score at 26 weeks | SID: Median 2 (IQR 1-4); Placebo: Median 2 (IQR 1-3); Adjusted OR: 1.0 (0.5-2.2)Secondary
Improvement ≥1 point GBS disability at 4 weeks | SID: 18 (37%); Placebo: 12 (27%); Adjusted OR: 1.8 (0.6-5.3)Secondary
Improvement ≥1 point GBS disability at 26 weeks | SID: 40 (82%); Placebo: 41 (93%); Adjusted OR: 0.4 (0.1-2.6)Secondary
Mean MRC sumscore at 4 weeks | SID: 32 (26-37); Placebo: 30 (25-36); β coefficient: 1.3 (95% CI -1.6 to 4.1)Secondary
Mean MRC sumscore at 26 weeks | SID: 46 (41-52); Placebo: 51 (47-55); β coefficient: -2.0 (95% CI -4.8 to 0.8)Secondary
ONLS score at 4 weeks | SID: 10 (8-12); Placebo: 10 (7-12); Adjusted OR: 1.2 (0.5-2.6)Secondary
Mechanical ventilation required | SID: 30 (61%); Placebo: 25 (57%); Adjusted OR: 1.3 (0.5-3.3)Secondary
Duration of mechanical ventilation - median (IQR) | SID: 26 (12-58) days; Placebo: 43 (9-80) daysSecondary
Duration of hospital admission - median (IQR) | SID: 39 (21-67) days; Placebo: 30 (21-73) daysSecondary
Treatment-related fluctuation | SID: 3 (6%); Placebo: 5 (11%); Adjusted OR: 0.6 (0.1-2.7)Secondary
Serum IgG at 2 weeks - median (IQR) | SID: 34 (30-43) g/L; Placebo: 17 (16-20) g/LSecondary
Any serious adverse event during studyAdverseSID: 25 (51%); Placebo: 10 (23%); OR: 3.54 (95% CI 1.44-8.72)0.005
Any SAE in first 30 daysAdverseSID: 17 (35%); Placebo: 7 (16%)
DeathsAdverseSID: 4 (8%); Placebo: 0 (0%); Details: All 4 deaths in SID group: 59M asystole at 16 weeks (possibly related to acute coronary syndrome 4 days post-SID); 82F withdrawal of ventilation at 13 weeks; 72F cardiac death at 21 weeks; 81F withdrawal of ventilation at 24 weeks
Coronary ischaemiaAdverseSID: 1 (2%); Placebo: 1 (2%)
AsystoleAdverseSID: 2 (4%); Placebo: 0 (0%)
Pulmonary embolismAdverseSID: 2 (4%); Placebo: 0 (0%)
Transient ischemic attackAdverseSID: 1 (2%); Placebo: 0 (0%)
Radial artery thrombosisAdverseSID: 1 (2%); Placebo: 0 (0%)
PneumoniaAdverseSID: 12 (24%); Placebo: 7 (16%)
Renal insufficiencyAdverseSID: 1 (2%); Placebo: 1 (2%)
Hemolytic anemiaAdverseSID: 0 (0%); Placebo: 0 (0%)

Subgroup Analysis

Prespecified subgroup analyses showed no benefit of SID in any subgroup including: age <60 or ≥60, MRC 0-12 or >12, mEGOS 10-12 or 6-9, mechanical ventilation yes/no, demyelinating vs axonal subtype, Campylobacter positive/negative, anti-GM1/GD1a positive/negative, high vs low delta IgG.


Criticisms

  • Relatively small sample size with wide confidence intervals
  • Baseline imbalance in prognostic factors (SID group older, more preceding diarrhoea)
  • Long enrollment period (>8 years) though treatment standards unchanged
  • Selected only poor prognosis patients; results may not apply to all GBS
  • 4 patients excluded post-randomization (2 alternative diagnoses, 2 withdrew)
  • Cannot exclude very small beneficial effect below detection threshold
  • Acute-onset CIDP diagnosed in 4 patients during follow-up (known confounder)
  • Pre-existing vascular risk factors were exclusion criterion limiting generalizability
  • Single country (Netherlands) study

Funding

Prinses Beatrix Spierfonds and Sanquin Plasma Products

Based on: SID-GBS (Lancet Neurology, 2021)

Authors: Christa Walgaard, Bart C Jacobs, Hester F Lingsma, ..., et al.; on behalf of the Dutch GBS Study Group

Citation: Lancet Neurol 2021;20:275-283

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