A phase I trial and pharmacokinetic study of 9-cis-retinoic acid (ALRT1057) in pediatric patients with refractory cancer: A joint Pediatric Oncology Branch, National Cancer Institute, and Children's Cancer Group Study

Peter C. Adamson, Brigitte C. Widemann, Robert F. Murphy, Andrea F. Gillespie, Frank M. Balis, Gregory H. Reaman, Nita L. Seibel

Research output: Contribution to journalArticlepeer-review

18 Scopus citations

Abstract

Purpose: To determine the maximum tolerated dose and describe the toxicities of 9-cis-retinoic acid (9cRA, ALRT1057) administered p.o. tid in pediatric patients with refractory cancer and to study the pharmacokinetics of 9cRA and determine whether systemic drug exposure changes with chronic dosing. Patients and Methods: Children with refractory cancer (stratified by age, ≤12 and > 12 years) were treated with p.o. 9cRA for 28 consecutive days. The starting dose was 50 mg/m2/day divided into 3 doses with planned escalations to 65, 85, and 110 mg/m2/day. Pharmacokinetic sampling was performed on days 1 and 29 of the first cycle. Results: Of the 37 patients entered, 18 patients ≤12 years of age and 11 patients >12 years of age were evaluable for toxicity. In patients >12 years of age, dose-limiting headache occurred in 2/2 patients at the 110 mg/m2/day dose level; 1/8 patients at 85 mg/m2/day developed dose-limiting pseudotumor cerebri. In patients ≤12 years of age, 3/5 patients at the starting dose level of 50 mg/m2/day developed dose-limiting pseudotumor cerebri; and 0/6 patients experienced dose-limiting toxicity at 35 mg/m2/day. Reversible non-dose-limiting hepatotoxicity was observed in 15 patients across all of the dose levels. There was considerable interpatient variability in 9cRA plasma concentrations. Peak plasma concentrations of 9cRA occurred at a median of 1.5 h after a p.o. dose, and the harmonic-mean terminal half-life was 43 min. By day 29 of 9cRA administration, the plasma 9cRA area under the curve declined by an average of 65% from day 1 values. Conclusions: The dose-limiting toxicity of 9cRA in pediatric patients was neurotoxicity, primarily pseudotumor cerebri. Younger children tolerate significantly lower doses of 9cRA than older children. Similar to all-trans-retinoic acid, the pharmacokinetics of 9cRA demonstrated a wide degree of interpatient variability and decreased over time when administered on a daily basis. The recommended Phase II dose of 9cRA in patients ≤12 and >12 years of age is 35 and 85 mg/m2/day, respectively.

Original languageEnglish (US)
Pages (from-to)3034-3039
Number of pages6
JournalClinical Cancer Research
Volume7
Issue number10
StatePublished - 2001
Externally publishedYes

ASJC Scopus subject areas

  • Oncology
  • Cancer Research

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