Dramatic pain relief and resolution of bone inflammation following pamidronate in 9 pediatric patients with persistent chronic recurrent multifocal osteomyelitis (CRMO)

被引:120
作者
Miettunen, Paivi M. H. [1 ,2 ,3 ]
Wei, Xingchang [2 ,4 ]
Kaura, Deepak [2 ,4 ]
Abou Reslan, Walid [2 ,4 ]
Aguirre, Alberto Nettel [1 ,2 ,5 ]
Kellner, James D. [1 ,2 ,6 ]
机构
[1] Alberta Childrens Prov Gen Hosp, Dept Pediat, Calgary, AB T3B 6A8, Canada
[2] Univ Calgary, Calgary, AB T3B 6A8, Canada
[3] Alberta Childrens Prov Gen Hosp, Div Pediat Rheumatol, Calgary, AB T3B 6A8, Canada
[4] Alberta Childrens Prov Gen Hosp, Dept Diagnost Imaging, Calgary, AB T3B 6A8, Canada
[5] Alberta Childrens Prov Gen Hosp, Dept Community Hlth Sci, Calgary, AB T3B 6A8, Canada
[6] Alberta Childrens Prov Gen Hosp, Div Pediat Infect Dis, Calgary, AB T3B 6A8, Canada
关键词
INTRAVENOUS PAMIDRONATE; SAPHO SYNDROME; OSTEOGENESIS IMPERFECTA; CHILDREN; OSTEONECROSIS; OSTEITIS; THERAPY; DISEASE;
D O I
10.1186/1546-0096-7-2
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Background: Chronic recurrent multifocal osteomyelitis (CRMO) is an inflammatory, noninfectious osteopathy that affects predominantly patients <= 18 years of age. There is no uniformly effective treatment. Our objective is to describe clinical, magnetic resonance imaging (MRI), and bone resorption response to intravenous pamidronate in pediatric CRMO. Methods: We report our prospectively documented experience with all CRMO patients treated with pamidronate between 2003 and 2008 at a tertiary pediatric centre. Pamidronate was administered as intravenous cycles. The dose of pamidronate varied among subjects but was given as monthly to every 3 monthly cycles depending on the distance the patient lived from the infusion center. Maximum cumulative dose was <= 11.5 mg/kg/year. Pamidronate treatment was continued until resolution of MRI documented bone inflammation. Visual analog scale for pain (VAS) and bone resorption marker urine N-telopeptide/urine creatinine (uNTX/uCr) were measured at baseline, preceding each subsequent pamidronate treatment, at final follow-up, and/or at time of MRI confirmed CRMO flare. MRI of the affected site(s) was obtained at baseline, preceding every 2(nd) treatment, and with suspected CRMO recurrence. Results: Nine patients (5 F: 4 M) were treated, with a median (range) age at treatment of 12.9 (4.5-16.3) years, and median (range) duration of symptoms of 18 (6-36) months. VAS decreased from 10/10 to 0-3/10 by the end of first 3-day treatment for all patients. The mean (range) time to complete MRI resolution of bone inflammation was 6.0 (2-12) months. The mean (confidence interval (CI)) baseline uNTX/uCr was 738.83 (CI 464.25, 1013.42) nmol/mmol/creatinine and the mean (CI) decrease from baseline to pamidronate discontinuation was 522.17 (CI 299.77, 744.56) nmol/mmol/creatinine. Median (range) of follow-up was 31.4 (24-54) months. Four patients had MRI confirmed CRMO recurrence, which responded to one pamidronate re-treatment. The mean (range) uNTX/uCr change as a monthly rate from the time of pamidronate discontinuation to flare was 9.41 (1.38-19.85) nmol/mmol/creatinine compared to -29.88 (-96.83-2.01) nmol/mmol/creatinine for patients who did not flare by the time of final follow-up. Conclusion: Pamidronate resulted in resolution of pain and MRI documented inflammation in all patients. No patient flared while his/her uNTX/uCr remained suppressed. We propose that pamidronate is an effective second-line therapy in persistent CRMO.
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页数:14
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