TITLE

Intravenous pamidronate treatment of infants with severe osteogenesis imperfecta

AUTHOR(S)
Åström, Eva; Jorulf, Hâkan; Söderhäll, Stefan
PUB. DATE
April 2007
SOURCE
Archives of Disease in Childhood;Apr2007, Vol. 92 Issue 4, p332
SOURCE TYPE
Academic Journal
DOC. TYPE
Article
ABSTRACT
Objective: Children with the severe forms of osteogenesis imperfecta have in several studies been treated with intravenous ipamidronate, but there are only few reports of the effect of early treatment. Aim: To evaluate the effect of treatment started in infancy. Methods: In a prospective observational study, with a historic control group, intravenous disodium pamidronate (APD) was given as monthly infusions to 11 children with osteogenesis imperfecta aged 3–13 (median 3.6) months, who had severe osteogenesis imperfecta with congenital bowing of the femora and vertebral compression fractures. Results: During treatment of children aged between 3 and 6 (median 4.5) years, dual-energy x ray absorptiometry measurements of the lumbar spine showed a gradual increase in bone density. Bone metabolism parameters in serum (alkaline phosphatase, osteocalcin, procollagen 1 carboxy-terminal peptide, collagen 1 teleopeptide) and in urine (deoxypyridinoline) indicated a decrease in bone turnover. An improvement of mobility was seen and at the latest recording, at the age of 3.3–6.5 (median 4.8) years, the children could all walk. Vertebral remodelling was seen, with increased vertebral height, and no child developed scoliosis, kyphosis or basilar impression. All children required femoral intramedullar rods for fractures, and five needed tibial rodding for extreme curvatures that prevented functional standing and walking. No adverse effects were seen on growth, fracture healing or blood chemistry. Conclusions: APD is an efficient symptomatic treatment for infants with severe osteogenesis imperfecta, but additional orthopaedic surgery is often needed. Early treatment may prevent scoliosis and basilar impression. Long-term follow-up is important.
ACCESSION #
25020985

 

Related Articles

  • Treatment of symptomatic osteoporosis in children: a comparison of two pamidronate dosage regimens. Martinez-Soto, Tania; Pacaud, Danièle; Stephure, David; Trussell, Rebecca; Huang, Carol // Journal of Pediatric Endocrinology & Metabolism;Jun2011, Vol. 24 Issue 5/6, p271 

    Bisphosphonate treatment for bone fragility has expanded beyond children with osteogenesis imperfecta (OI) to those with other causes of low bone mass. However, clinical efficacy and optimal dosing in non-OI patients has not been established. We conducted a retrospective descriptive study of...

  • Intravenous pamidronate treatment in osteogenesis imperfecta. Fujiwara, I.; Ogawa, E.; Igarashi, Y.; Ohba, M.; Asanuma, A. // European Journal of Pediatrics;1998, Vol. 157 Issue 3, p261 

    Presents two case reports which indicated effectiveness of oral pamidronate, a bisphosphonate which potentially inhibits bone resorption, in osteogenesis imperfecta (OI). Concern that the efficacy was unclear because the patients were pubertal and sex hormone changes might have contributed to...

  • Intermittent Oral Disodium Pamidronate in Established Osteoporosis: A 2 Year Double-Masked Placebo-Controlled Study of Efficacy and Safety. Ryan, P. J.; Blake, G. M.; Davie, M.; Haddaway, M.; Gibson, T.; Fogelman, I. // Osteoporosis International;Feb2000, Vol. 11 Issue 2, p171 

    : The effect of oral pamidronate on bone mineral density and its adverse effect profile was investigated by a double-masked placebo-controlled study of 122 patients aged 55–75 years with established vertebral osteoporosis. Patients on active therapy received disodium pamidronate 300...

  • Beneficial effect of long term intravenous bisphosphonate treatment of osteogenesis imperfecta. Åström, E.; Söderhäll, S. // Archives of Disease in Childhood;May2002, Vol. 86 Issue 5, p356 

    Aim: To find an effective symptomatic treatment for osteogenesis imperfecta (OI). Methods: In a prospective observational study disodium pamidronate (APD) was given as monthly intravenous infusions to 28 children and adolescents (aged 0.6-18 years) with severe OI or a milder Form of the disease,...

  • Aredia Dry Powder.  // Royal Society of Medicine: Medicines;2002, p86 

    The article presents information on Aredia Dry Powder, a proprietary, prescription-only preparation of disodium pamidronate. It is used to treat bone disorders, and is available in a form for intravenous infusion.

  • Bisphosphonate-Induced Osteopetrosis. Whyte, Michael P.; Wenkert, Deborah; Clements, Karen L.; McAlister, William H.; Mumm, Steven // New England Journal of Medicine;7/31/2003, Vol. 349 Issue 5, p457 

    The article discusses the case of drug-induced osteopetrosis in a male pediatric patient. It was found that osteopetrosis developed in the patient, coinciding with the administration of pamidronate. Bone densitometry revealed a supranormal whole-body value. According to the article, untoward...

  • Evaluation of a Modified Pamidronate Protocol for the Treatment of Osteogenesis Imperfecta. Palomo, Telma; Andrade, Maria; Peters, Barbara; Reis, Fernanda; Carvalhaes, João; Glorieux, Francis; Rauch, Frank; Lazaretti-Castro, Marise; Andrade, Maria C; Peters, Barbara S E; Reis, Fernanda A; Carvalhaes, João Tomás A; Glorieux, Francis H // Calcified Tissue International;Jan2016, Vol. 98 Issue 1, p42 

    Intravenous pamidronate is widely used to treat children with osteogenesis imperfecta (OI). In a well-studied protocol ('standard protocol'), pamidronate is given at a daily dose of 1 mg per kg body weight over 4 h on 3 successive days; infusion cycles are repeated every 4 months. Here, we...

  • Modeling the benefits of pamidronate in children with osteogenesis imperfecta. Lindsay, Robert // Journal of Clinical Investigation;11/1/2002, Vol. 110 Issue 9, p1239 

    Discusses the utility of pamidronate in children with osteogenesis imperfecta (OI). Brone growth and metabolism in OI; Effects of biphosphonates; Risks and benefits of pamidronate.

  • Cyclic Pamidronate Therapy in Children with Osteogenesis Imperfecta. Salehpour, S.; Tavakkoli, S. // Journal of Pediatric Endocrinology & Metabolism;Jan/Feb2010, Vol. 23 Issue 1/2, p73 

    The article discusses the findings of a study which determined the efficacy of cyclic pamidronate treatment in improving bone mineralization of children diagnosed with osteogenesis imperfecta (OI). For this research, 64 children ranging from 21 months to 10 years old were treated with...

Share

Read the Article

Courtesy of THE LIBRARY OF VIRGINIA

Sorry, but this item is not currently available from your library.

Try another library?
Sign out of this library

Other Topics