Trial reportJournal of inherited metabolic disease2002
Pamidronate treatment improves bone mineral density in children with Menkes disease.
Trial report in Journal of inherited metabolic disease, 2002. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers, 1 of them a synthesis that pooled it.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
15 citing papers in PubMed, 1 synthesis or guideline pooled it, 61 citations in OpenAlex.
- Bisphosphonate therapy for children and adolescents with secondary osteoporosis.The Cochrane database of systematic reviews · 2007Pooled it
- Review Article on Molecular Basis of Zinc and Copper Interactions in Cancer Physiology.Biological trace element research · 2025Review
- Cuproptosis and its potential role in musculoskeletal disease.Frontiers in cell and developmental biology · 2025Review
- Design and application of metal organic frameworks for heavy metals adsorption in water: a review.RSC advances · 2024Review
- Bone fragility in patients affected by congenital diseases non skeletal in origin.Orphanet journal of rare diseases · 2021Review
- The Role of Zinc and Copper in Gynecological Malignancies.Nutrients · 2020Review
- A critical review on recent developments in MOF adsorbents for the elimination of toxic heavy metals from aqueous solutions.Environmental science and pollution research international · 2020Review
- Impaired osteogenesis in Menkes disease-derived induced pluripotent stem cells.Stem cell research & therapy · 2015Article
- Urinary concentrations of toxic and essential trace elements among rural residents in Hainan Island, China.International journal of environmental research and public health · 2014Article
- The ever-expanding conundrum of primary osteoporosis: aetiopathogenesis, diagnosis, and treatment.Italian journal of pediatrics · 2014Review
- Copper deficit as a potential pathogenic factor of reduced bone mineral density and severe tooth wear.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2014Observational
- Copper status in patients with thalassemia major in zahedan, iran.International journal of hematology-oncology and stem cell research · 2013Article
- Somatic mosaicism in Menkes disease suggests choroid plexus-mediated copper transport to the developing brain.American journal of medical genetics. Part A · 2010Article
- Bisphosphonate treatment of bone disease.Archives of disease in childhood · 2005Review
- Intravenous bisphosphonate therapy for osteoporosis: where do we stand?Current osteoporosis reports · 2004Review
Corrections and comments
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Authors and funding
3 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Menkes disease is a severe multisystem disorder due to defective bioavailability and transport of copper at the cellular level. Deficient activity of lysyl oxidase, a copper-dependent enzyme, causes defective collagen cross-linking leading to osteoporosis and pathological fractures in these children. The objective of the study was to evaluate the changes in bone mineral density following pamidronate treatment in children with Menkes disease. The study design was an open observational study of three children with Menkes disease and significant osteoporosis with or without pathological fractures, all of whom received pamidronate treatment for 1 year. There were 34-55% and 16-36% increases in lumbar spine bone mineral content and areal bone mineral density, respectively, following 1 year of treatment with pamidronate. There were no further fractures in two of the three children treated. No adverse effects of pamidronate treatment were noted. Pamidronate treatment was associated with an increase in bone mineral density and may be an effective treatment modality for the management of osteoporosis in children with Menkes disease.
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Registered trials
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