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Maintenance therapy with dose-adjusted 6-mercaptopurine in idiopathic pulmonary hemosiderosis
Xue-Qun Luo1, Zhi-Yong Ke1, Li-Bin Huang1
1Department of Pediatric, The First Affiliated Hospital of Sun Yat-Sen University, Zhongshan Er Lu, Guangzhou 510080, China.
Insights
Idiopathic pulmonary hemosiderosis (IPH) in children can be challenging to diagnose and treat. Dose-adjusted 6-mercaptopurine (6MP) maintenance therapy may lead to steroid-free remission, with relative leukopenia as a predictor of response.
Area of Science:
- Pediatric Pulmonology
- Clinical Immunology
- Pharmacology
Background:
- Idiopathic pulmonary hemosiderosis (IPH) presents diagnostic and therapeutic challenges in children.
- Delayed diagnosis is frequent due to the absence of the classical IPH triad in many pediatric cases.
Purpose of the Study:
- To review IPH diagnosis in children.
- To evaluate the efficacy of maintenance therapy with dose-adjusted 6-mercaptopurine (6MP) in pediatric IPH patients.
Main Methods:
- Fifteen children with IPH were enrolled in the study.
- Initial treatment involved prednisone (2 mg/kg/day) followed by a taper.
- Maintenance therapy with 6MP (60 mg/m²/day) was administered for 3 years, with dose adjustments based on leukopenia.
Main Results:
- All patients responded to initial prednisone treatment.
- Patients with relative leukopenia on 6MP maintenance had significantly lower recurrence rates (1/8) compared to those without (5/7, P < 0.05).
- Four of five patients who recurred became recurrence-free after upward dose adjustment of 6MP to maintain relative leukopenia.
Conclusions:
- Children with IPH can achieve long-term, steroid-free remission with 6MP maintenance therapy.
- Relative leukopenia during 6MP therapy may serve as a predictive marker for clinical response in pediatric IPH.
Abstract:
There are challenges for diagnosis and treatment of idiopathic pulmonary hemosiderosis (IPH). This clinical trial was to review the diagnosis and evaluate the efficacy of maintenance therapy with dose-adjusted 6-mercaptopurine (6MP) in IPH children. Fifteen children were enrolled. Prednisone was administered at 2 mg/kg/day for 4 weeks in acute phase of the disease followed by taper. 6MP was also started at 60 mg/m(2)/day simultaneously and continued for 3 years in outpatient. The delay in diagnosis of IPH is common and probably due to a lack of classical triad of IPH in most children. All the patients exhibited response to the initial treatment. Only one of eight patients with relative leukopenia on 6MP maintenance recurred while 5 of 7 others recurred (P < 0.05) during median 4.5-year follow-up. Of the latter five patients who recurred, 4 remained recurrence-free after adjusting the dose of 6MP upwards to keep relative leucopenia. It suggests that children with IPH could achieve steroid-free long term remission on 6MP maintenance therapy, and relative leukopenia on 6MP might be a simple maker of predicting clinical response in most IPH children.
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