Trial Outcomes & Findings for Use of Rituximab in Opsoclonus-Myoclonus in Children With Neuroblastoma (NCT NCT00202930)

NCT ID: NCT00202930

Last Updated: 2021-01-22

Results Overview

To evaluate the feasibility of using 4 weekly Rituximab infusions in the treatment of children with neuroblastoma associated opsoclonus-myoclonus syndrome (OMS). The first infusion involved a slow up titration from an initial rate of 0.5 mg/kg/hour to a maximum of 400 mg/hour. If well tolerated, the subsequent infusions were administered at a starting rate of 1 mg/kg/hour to a maximum of 100 mg/hour for the first hour. In the absence of adverse reaction doses were escalated by 1 mg/kg/hour (maximum 100 mg increase per hour) every 30 minutes to a maximum rate of 400 mg/hour. If hypersensitivity or infusion related events occurred, the infusion was temporarily interrupted and resumed at 50% of the previous rate if reaction resolves. The number of participants for whom the study dosing was feasible is reported.

Recruitment status

TERMINATED

Study phase

PHASE2

Target enrollment

4 participants

Primary outcome timeframe

4 weeks

Results posted on

2021-01-22

Participant Flow

Participant milestones

Participant milestones
Measure
Rituximab
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
Overall Study
STARTED
4
Overall Study
COMPLETED
4
Overall Study
NOT COMPLETED
0

Reasons for withdrawal

Withdrawal data not reported

Baseline Characteristics

Use of Rituximab in Opsoclonus-Myoclonus in Children With Neuroblastoma

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Rituximab
n=4 Participants
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
Age, Categorical
<=18 years
4 Participants
n=99 Participants
Age, Categorical
Between 18 and 65 years
0 Participants
n=99 Participants
Age, Categorical
>=65 years
0 Participants
n=99 Participants
Sex: Female, Male
Female
3 Participants
n=99 Participants
Sex: Female, Male
Male
1 Participants
n=99 Participants
Region of Enrollment
United States
4 participants
n=99 Participants
OMS Evaluation Scale of Motor-Performance.
NA units on a scale
n=99 Participants

PRIMARY outcome

Timeframe: 4 weeks

To evaluate the feasibility of using 4 weekly Rituximab infusions in the treatment of children with neuroblastoma associated opsoclonus-myoclonus syndrome (OMS). The first infusion involved a slow up titration from an initial rate of 0.5 mg/kg/hour to a maximum of 400 mg/hour. If well tolerated, the subsequent infusions were administered at a starting rate of 1 mg/kg/hour to a maximum of 100 mg/hour for the first hour. In the absence of adverse reaction doses were escalated by 1 mg/kg/hour (maximum 100 mg increase per hour) every 30 minutes to a maximum rate of 400 mg/hour. If hypersensitivity or infusion related events occurred, the infusion was temporarily interrupted and resumed at 50% of the previous rate if reaction resolves. The number of participants for whom the study dosing was feasible is reported.

Outcome measures

Outcome measures
Measure
Rituximab
n=4 Participants
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
Feasibility of Using 4 Weekly Rituximab Infusions
4 Participants

PRIMARY outcome

Timeframe: Individuals were followed for adverse events until day 270

The trial was to be terminated early for any grade 3 or 4 toxicity that did not resolve in 2 of the first 5 patients treated, or for any infusion related death. The number of participants who experienced these events \[grade 3 or 4 toxicity that did not resolve in 2 of the first 5 patients treated, or for any infusion related death\] is reported.

Outcome measures

Outcome measures
Measure
Rituximab
n=4 Participants
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
Toxicity of Rituximab
0 Participants

SECONDARY outcome

Timeframe: Baseline, following the four infusions, at months 3, 6, 12, 18 and 24 following the first infusion.

OMS Evaluation Scale of Motor-Performance This scoring system utilizes a scale of 0 to 3, with 0 being unaffected by OMS, and 3 representing those with severe impairment. OMS testing will be scored by two independent scorers who have special expertise in the evaluation and management of children with neurologic disorders. The mean of these scores will be obtained, and a standard error of the mean reported.

Outcome measures

Outcome measures
Measure
Rituximab
n=4 Participants
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
OMS Evaluation Scale of Motor-Performance
NA score on a scale
The raw data for OMS scoring is at the NIH in the offices of Dr. Nina Schor, MD PhD. She was the responsible neurology investigator in this study. The trial closed early due to low accrual and did not enroll adequate numbers to conduct detailed statistical analysis of the raw data. Due to the pandemic and discontinuation of in person activities, a decision was made by the NIH to undertake extensive renovations of the facility. Dr. Schor is unable to access them due to construction.

SECONDARY outcome

Timeframe: Baseline; 3, 6, 9 months post treatment

Population: These studies were unable to be performed due to withdraw of funding from the study sponsor. 0 of 4 patients participated.

Blood samples to be evaluated for the occurrence of antibody formation and potential effect on pharmacokinetic profiles.

Outcome measures

Outcome data not reported

SECONDARY outcome

Timeframe: 2 years

B cell depletion was measured through analysis of serum IgG levels. The number of participants who experienced B cell depletion is reported.

Outcome measures

Outcome measures
Measure
Rituximab
n=4 Participants
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
Peripheral B Cell Depletion
0 Participants

Adverse Events

Rituximab

Serious events: 1 serious events
Other events: 0 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
Rituximab
n=4 participants at risk
Participants received 4 weekly doses of IV rituxan at 375 mg/m\^2 on days 1, 8, 15 and 22
General disorders
Febrile illness with upper respiratory symptoms and dehydration
25.0%
1/4 • Number of events 1 • In addition to outlined in-office evaluations, telephone contact to inquire regarding hospitalizations and adverse event questioning will be performed at days 60, 120 and 270 due to the length of time between scheduled testing.

Other adverse events

Adverse event data not reported

Additional Information

Jean M. Tersak, M.D.

UPMC Children's Hospital of Pittsburgh

Phone: 412-692-8570

Results disclosure agreements

  • Principal investigator is a sponsor employee
  • Publication restrictions are in place