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Trial Evaluating Efficacy and Safety of Dasiglucagon in Children With Congenital Hyperinsulinism

Phase 2
Completed
Conditions
Congenital Hyperinsulinism
Interventions
Drug: Placebo
Registration Number
NCT04172441
Lead Sponsor
Zealand Pharma
Brief Summary

The objective of the trial is to evaluate the efficacy of dasiglucagon in reducing glucose requirements in children with persistent congenital hyperinsulinism (CHI) requiring continuous intravenous (IV) glucose administration to prevent/manage hypoglycemia.

Detailed Description

Not available

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
12
Inclusion Criteria
  • CHI diagnosis established based on the following:

    1. Hyperinsulinemia: plasma insulin above the limit of detection of the assay documented during an event of hypoglycemia, and/or
    2. Hypofattyacidemia: plasma free fatty acid <1.7 mmol/L, and/or
    3. Hypoketonemia: Beta-hydroxybutyrate <1.8 mmol/L, and/or
    4. Glycemic response: an increase in plasma glucose (PG) of >30 mg/dL (1.7 mmol/L) after 1 mg IV or intramuscular (IM) glucagon administration
  • Male or female, age ≥7 days and <12 months at screening

  • Body weight of ≥2.0 kg (4.4 lbs.)

  • Continuous IV glucose requirement to prevent hypoglycemia

Exclusion Criteria
  • Is suspected of having a transient form of CHI (e.g., transient hyperinsulinism due to maternal diabetes or perinatal stress)
  • Was born preterm below 34 weeks of gestational age
  • Presence of hypertension or hypotension, including circulatory instability requiring supportive medication or presence of pheochromocytoma
  • Known or suspected presence of severe brain damage
  • Evidence of metabolic, endocrine, or syndromic causes of hypoglycemia not due to hyperinsulinism
  • Use of systemic corticosteroids, e.g., hydrocortisone >20 mg/m^2 body surface area or equivalent within 5 days before screening
  • Prior use of lanreotide, sirolimus (mechanistic target of rapamycin [mTOR] inhibitors), anti-inflammatory biological agents, or other immune modulating agents. Prior use of octreotide is allowed after a minimum of 48 hour washout before randomization.
  • Any clinically significant abnormality identified on echocardiogram that in the opinion of the investigator would affect the subject's ability to participate in the trial
  • Any recognized clotting or bleeding disorder
  • The use of prescription or non-prescription medications known to cause QT prolongation

Study & Design

Study Type
INTERVENTIONAL
Study Design
CROSSOVER
Arm && Interventions
GroupInterventionDescription
dasiglucagon first then placeboPlacebo48 hours of dasiglucagon subcutaneous (sc) infusion starting at 10 µg/hour with crossover to 48 hours placebo sc infusion (part 1) followed by 21 days of dasiglucagon sc infusion (part 2).
placebo first then dasiglucagonPlacebo48 hours of placebo sc infusion with crossover to 48 hours dasiglucagon sc infusion starting at 10 µg/hour (part 1) followed by 21 days of dasiglucagon sc infusion (part 2).
dasiglucagon first then placebodasiglucagon48 hours of dasiglucagon subcutaneous (sc) infusion starting at 10 µg/hour with crossover to 48 hours placebo sc infusion (part 1) followed by 21 days of dasiglucagon sc infusion (part 2).
placebo first then dasiglucagondasiglucagon48 hours of placebo sc infusion with crossover to 48 hours dasiglucagon sc infusion starting at 10 µg/hour (part 1) followed by 21 days of dasiglucagon sc infusion (part 2).
Primary Outcome Measures
NameTimeMethod
Mean Intravenous Glucose Infusion RateHours 36-48 after initiation of trial drug (Part 1)

Mean intravenous (IV) glucose infusion rate (GIR) in the last 12 hours of each treatment period during Part 1, the crossover part of the trial (dasiglucagon or placebo administration).

Secondary Outcome Measures
NameTimeMethod
Carbohydrates AdministeredDays 5 to 25

Total amount (g) of carbohydrates administered (regardless of the route) per day.

Mean Intravenous Glucose Infusion Rate48 hours after initiation of trial drug (Part 1)

Mean IV GIR for each 48-hour treatment period during Part 1, the crossover part of the trial (dasiglucagon or placebo administration).

Mean Intravenous Glucose Infusion Rate Below 10 mg/kg/MinuteHours 36-48 after initiation of trial drug (Part 1)

Mean IV GIR below 10 mg/kg/minute in the last 12 hours of each treatment period during Part 1, the crossover part of the trial (dasiglucagon or placebo administration) (yes/no)

Time to Complete Weaning Off Intravenous GlucoseDays 5 to 25 (Part 2)

Time in days to complete weaning off IV glucose administration during Part 2, defined as the first point in time when the patient had been off IV glucose administration for at least 12 hours.

Hypoglycemia Event Rate in Part 2Days 5 to 25

Hypoglycemia event rate, defined as number of hypoglycemic events when PG was \<70 mg/dL (or 3.9 mmol/L), as detected by self-monitored plasma glucose.

Clinically Significant Hypoglycemia Events in Part 2Days 5 to 25

Clinically significant hypoglycemia event rate, defined as number of events when PG was \<54 mg/dL (3.0 mmol/L), as detected by self-monitored plasma glucose.

Time to Actual Hospital DischargeDays 5 to 25

Time in days to actual hospital discharge defined as the time from first exposure to the study drug in Part 2 to discharge from hospital.

Time to Pancreatic SurgeryDays 5 to 25

Time (days) to pancreatic surgery (sub-total or total pancreatectomy with a cutoff of ≥95%).

Carbohydrates Administered IntravenouslyDays 5 to 25

Amount (g) of carbohydrates administered via IV glucose infusion or bolus or total parenteral nutrition. This secondary endpoint was intended to account only for carbohydrates administered via IV glucose infusion or bolus. It was not possible to differentiate between carbohydrates administered via IV glucose infusion or bolus and carbohydrates administered as being, or not being, part of total parenteral nutrition from the collected data. This endpoint was expanded to include both.

Carbohydrates Administered ParenterallyDays 5 to 25

Amount (g) of carbohydrates administered as part of total parenteral nutrition.

Carbohydrates Administered OrallyDays 5 to 25

Amount (g) of carbohydrates administered via oral route.

Carbohydrates Administered Via Gastric FeedDays 5 to 25

Amount (g) of carbohydrates administered via nasogastric tube or gastrostomy.

Time in Range in Part 2Days 5 to 25

Percent time in range (PG between 70 to 180 mg/dL \[3.9-10.0 mmol/L\]) as measured by continuous glucose monitoring.

Time in Hypoglycemia in Part 2Days 5 to 25

Percent time in hypoglycemia (when PG was \<70 mg/dL \[or 3.9 mmol/L\]) as measured by continuous glucose monitoring.

Time in Clinically Significant Hypoglycemia in Part 2Days 5 to 25

Percent time in clinically significant hypoglycemia (when PG was \<54 mg/dL \[or 3.0 mmol/L\]) as measured by continuous glucose monitoring.

Hypoglycemia Episodes in Part 2Days 5 to 25

Rate of hypoglycemia episodes, defined as number of episodes per week when PG was \<70 mg/dL (3.9 mmol/L) for 15 minutes or more, as measured by continuous glucose monitoring.

Clinically Significant Hypoglycemia Episodes in Part 2Days 5 to 25

Rate of clinically significant hypoglycemia episodes, defined as number of episodes per week when was \<54 mg/dL (3.0 mmol/L) for 15 minutes or more, as measured by continuous glucose monitoring.

Extent of Hypoglycemia in Part 2Days 5 to 25

Extent of hypoglycemia (defined as the area over the glucose curve \[AOCglucose\] below 70 mg/dL \[3.9 mmol/L\]) as measured by continuous glucose monitoring.

Extent of Clinically Significant Hypoglycemia in Part 2Days 5 to 25

Extent of clinically significant hypoglycemia (defined as the area over the glucose curve \[AOCglucose\] below 54 mg/dL \[3.0 mmol/L\]) as measured by continuous glucose monitoring, divided by the total duration in hours of continuous glucose monitoring.

Time in Hyperglycemia in Part 2Days 5 to 25

Percent time in hyperglycemia (when PG was \>180 mg/dL \[10.0 mmol/L\]), as measured by continuous glucose monitoring.

Trial Locations

Locations (6)

University Children's Hospital

🇩🇪

Düsseldorf, Germany

Children's Hospital of Philadelphia

🇺🇸

Philadelphia, Pennsylvania, United States

Cook Children's Medical Center

🇺🇸

Fort Worth, Texas, United States

Hadassah Medical Center

🇮🇱

Jerusalem, Israel

University Hospital, Magdeburg

🇩🇪

Magdeburg, Germany

Manchester University NHS Foundation Trust

🇬🇧

Manchester, United Kingdom

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