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Combined chelation therapy with daily oral deferiprone and twice-weekly subcutaneous infusion of desferrioxamine in children with β-thalassemia: 3-year experience.

Acta haematologica
January 1, 2015
Duantida Songdej et al. (7 authors)
Clinical TrialJournal ArticleResearch Support, Non-U.S. Gov'tHuman StudyClinical
Study Details

Study Goal

The researchers aimed to evaluate the safety and efficacy of combined oral deferiprone (DFP) and subcutaneous desferrioxamine (DFO) therapy in reducing ferritin levels in transfusion-dependent β-thalassemia children.

Results Summary

The combination therapy significantly reduced median ferritin levels from 2,954.6 to 936.6 ng/ml in responders, with adverse events including elevated alanine aminotransferase (n = 5) and other minor side effects. Nonresponders had higher median age and longer transfusion duration.

Population

Transfusion-dependent β-thalassemia children (ages 3-18 years), including β-thalassemia major and β-thalassemia hemoglobin E patients.

Effective Dosage

50-100 mg/kg/day oral DFP combined with 40 mg/kg/dose s.c. DFO twice weekly.

Duration

Up to 36 months (28 patients completed follow-up).

Interactions

None mentioned

Extracted Claims (5)
InterventionDirectionEndpointPopulationDosageImpactClaim #
combined treatment with oral and subcutaneous iron chelators (50-100 mg/kg/day of oral deferiprone (DFP) combined with 40 mg/kg/dose s.c. desferrioxamine (DFO) twice weekly)
no change
chelation therapy
transfusion-dependent β-thalassemia children
-
is a safe and effective alternative to chelation monotherapy
#1
combined treatment with oral and subcutaneous iron chelators (50-100 mg/kg/day of oral deferiprone (DFP) combined with 40 mg/kg/dose s.c. desferrioxamine (DFO) twice weekly)
decrease
median ferritin levels
responders (among transfusion-dependent β-thalassemia children)
from 2,954.6 to 936.6 ng/ml
exhibited a significant fall
#2
combined treatment with oral and subcutaneous iron chelators (50-100 mg/kg/day of oral deferiprone (DFP) combined with 40 mg/kg/dose s.c. desferrioxamine (DFO) twice weekly)
decrease
serum ferritin
responders (among transfusion-dependent β-thalassemia children)
6 months
Time to a significant decrease
#3
100 mg/kg/day of DFP
decrease
ferritin
nonresponders (among transfusion-dependent β-thalassemia children)
<15%
ferritin declined <15%
#4
combined treatment with oral and subcutaneous iron chelators
increase
adverse events
13 patients (among transfusion-dependent β-thalassemia children)
16 episodes
occurred
#5
Abstract

OBJECTIVE: To study the efficacy of combined treatment with oral and subcutaneous iron chelators. MATERIAL AND METHODS: 50-100 mg/kg/day of oral deferiprone (DFP) combined with 40 mg/kg/dose s.c. desferrioxamine (DFO) twice weekly were given to transfusion-dependent β-thalassemia children. RESULTS: Enrolled patients (9 with β-thalassemia major and 33 with β-thalassemia hemoglobin E), ranging from 3 to 18 years in age, were divided into 3 groups; group 1 ferritin ≥1,000-2,500 ng/ml (n = 10), group 2 ferritin >2,500-4,000 ng/ml (n = 23) and group 3 ferritin >4,000 ng/ml (n = 9). Of the 42 patients, 28 reached the 36-month follow-up. Ten patients whose ferritin declined <15% while receiving 100 mg/kg/day of DFP were considered nonresponders. The median age and previous transfusion duration before enrollment were significantly higher in nonresponders than responders (p = 0.04 and 0.003, respectively). The responders exhibited a significant fall in median ferritin levels from 2,954.6 to 936.6 ng/ml (p < 0.001). Time to a significant decrease in serum ferritin among responders was 6 months. In 13 patients, 16 episodes of adverse events occurred: hemophagocytosis with cytopenia (n = 1), neutropenia (n = 2), thrombocytopenia (n = 2), elevated alanine aminotransferase (n = 5), elevated serum creatinine (n = 1), proteinuria (n = 1) and gastrointestinal discomfort (n = 4). CONCLUSION: Combination therapy with daily oral DFP and subcutaneous DFO twice weekly is a safe and effective alternative to chelation monotherapy in β-thalassemia children.

Medical Subject Headings (MeSH)
Administration, OralAdolescentAlanine TransaminaseChildChild, PreschoolCreatinineDeferiproneDeferoxamineDrug Therapy, CombinationFemaleFerritinsHemoglobin EHumansInfusions, SubcutaneousMaleNeutropeniaPyridonesSiderophoresThrombocytopeniabeta-Thalassemia
Study Links
Quality Scores
Safety70
Efficacy85/10
Quality75/10
Citation Metrics
Total Citations15
Citations/Year1.5
Relative Citation Ratio0.83
NIH Percentile43.3%
Research Impact Scores
APT Score0.50
Weight Score1.64
Normalized Score0.77
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