ArticleThe Journal of pediatrics2013
Effect of age at treatment on cognitive performance in patients with cystinosis.
Article in The Journal of pediatrics, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 1 of them a synthesis that pooled it.
What it found
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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
18 citing papers in PubMed, 1 synthesis or guideline pooled it, 49 citations in OpenAlex.
- Diagnosis and management of cystinosis: systematic review for a clinical practice guideline.Orphanet journal of rare diseases · 2025Pooled it
- A behavioral and electrophysiological investigation of conflict monitoring in cystinosis (CTNS gene mutations) using the flanker paradigm.Frontiers in nephrology · 2026Article
- Extrarenal complications of cystinosis.Pediatric nephrology (Berlin, Germany) · 2024Review
- Event-related potential (ERP) evidence for visual processing differences in children and adults with cystinosis (CTNS gene mutations).Orphanet journal of rare diseases · 2023Article
- Multisystem involvement, defective lysosomes and impaired autophagy in a novel rat model of nephropathic cystinosis.Human molecular genetics · 2022Article
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- Neuropsychological and neuroanatomical phenotype in 17 patients with cystinosis.Orphanet journal of rare diseases · 2020Article
- Hierarchical processing of visual stimuli in nephropathic cystinosis.Journal of inherited metabolic disease · 2019Article
- Effects of long-term cysteamine treatment in patients with cystinosis.Pediatric nephrology (Berlin, Germany) · 2019Review
- Therapeutic Applications of Cysteamine and Cystamine in Neurodegenerative and Neuropsychiatric Diseases.Frontiers in neurology · 2019Review
- [Cystinosis : Diagnosis, cystine-depleting therapy, and transition].Der Internist · 2018Review
- Nephropathic Cystinosis: Symptoms, Treatment, and Perspectives of a Systemic Disease.Frontiers in pediatrics · 2018Article
- The renal Fanconi syndrome in cystinosis: pathogenic insights and therapeutic perspectives.Nature reviews. Nephrology · 2017Review
- Intracranial Hypertension in Cystinosis Is a Challenge: Experience in a Children's Hospital.JIMD reports · 2017Article
- Chiari I Malformation in Nephropathic Cystinosis.The Journal of pediatrics · 2015Article
Corrections and comments
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Authors and funding
2 authors at 1 institution in 1 country.
Funding
Abstract
objectiveTo determine whether early treatment with cysteamine affects cognitive functioning in patients with nephropathic cystinosis. STUDY
designForty-six subjects aged 3-18 years with cystinosis underwent cognitive testing to determine intelligence, visual-spatial abilities, and visual-motor skills. An age-matched control group (n = 85; age 2-22 years) underwent the same tests. Age at diagnosis and age at initiation of treatment with cysteamine were recorded at the time of testing.
resultsPatients with cystinosis treated at or after age 2 years (late-treatment group) scored significantly lower on verbal, performance, and full-scale IQ measures, as well as on a test of visual-spatial skills, compared with patients treated before age 2 years (early-treatment group) and controls. Both groups of subjects with cystinosis demonstrated impaired visual-motor skills compared with controls, with no difference between the early-treatment and late-treatment groups.
conclusionEarly treatment with cysteamine appears to improve intellectual function in patients with nephropathic cystinosis. However, the lack of improvement in visual-motor function with early cysteamine treatment suggests possibly different mechanisms underlying visual-motor performance compared with other areas of cognition in this disorder.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.