Previous studies have shown that individuals with cystinosis may exhibit difficulty with visuospatial tasks. Global and local (hierarchical) processing are specific types of visuospatial tasks mediated by the right and left parietal lobes respectively. The study objective was to determine whether individuals with cystinosis and carriers of the cystinosin gene mutation show deficits in global and/or local processing. The study included 48 children (32 controls, 16 cystinosis) and 56 adults (25 controls, 25 carriers, and 6 cystinosis). Participants were instructed to replicate 10 hierarchical stimuli to assess global-local processing. The primary outcome measure was mean global and local performance on the hierarchical stimuli task between subgroups. Error subtypes were included to further assess each image. Compared to the control subgroups, adult and child cystinosis patients as well adult carriers demonstrated significant deficits in the global processing of a hierarchical stimulus against a background of unimpaired local processing. Child cystinosis patients performed significantly more poorly than controls on all error subtypes except local shape distortion. Adult cystinosis patients and carriers made significantly more global shape distortion errors than the controls. Our study shows that the cognitive profile documented in cystinosis patients and carriers includes significant difficulties with the global processing of an image. Results of the carrier population are important since they suggest that the cognitive impairments observed in patients cannot be attributed to symptomatic manifestations of the disease. Instead, the global processing deficits observed provide insight into the potential role of the cystinosin gene mutation on neurodevelopmental differences seen in these individuals.
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http://dx.doi.org/10.1002/jimd.12062 | DOI Listing |
Drugs Context
November 2024
Chiesi España S.A.U., Barcelona, Spain.
Background: Few studies have assessed patient-reported experience measures in nephropathic cystinosis. This study uses patient reports focused on the impact of cystinosis, cysteamine treatment-associated problems, and therapeutic adherence and suggests potential actions for improvement.
Methods: In March 2022, six patients with nephropathic cystinosis treated with cysteamine, aged between 12 and 40 years as well as two caregivers, underwent standardized online interviews.
Purpose: To evaluate ocular treatment adherence and its impact on clinical outcomes in patients with cystinosis in southwestern Ontario, where the disease incidence is higher due to a founder effect in the Old Order Amish population.
Methods: This was a retrospective case series of patients with ocular cystinosis seen at Victoria Hospital and the Ivey Eye Institute at St Joseph's Health Care in London, Ontario, Canada, from 2008 to 2023. The authors investigated the demographics, ocular manifestations, and visual outcomes and characteristics in pediatric patients with ocular cystinosis.
Clin Ophthalmol
November 2024
Department of Ophthalmology and Visual Science, Graduate School of Biomedical and Health Sciences, Hiroshima University, 1-2-3 Kasumi, Minami-ku, Hiroshima, 734-8551, Japan.
Purpose: Corneal cystine accumulation results in photophobia and affects patients' quality of life. We assessed the efficacy and safety of cysteamine 0.55% solution in Japanese cystinosis patients with corneal cystine crystals for 52 weeks.
View Article and Find Full Text PDFMedicina (Kaunas)
October 2024
Faculty of Biology, Medicine and Health, The University of Manchester, Manchester M1 7HR, UK.
Inherited metabolic disorders (IMDs), primarily cystinosis, Fabry disease, and methylmalonic acidemia (MMA), are genetic conditions that typically result in multi-organ disease manifestations. Kidney function progressively deteriorates in many cases, with patients eventually reaching end-stage kidney disease (ESKD) and requiring renal replacement therapy. Kidney transplantation has been deemed the optimal renal replacement therapy option to achieve long-term survival in patients with IMD.
View Article and Find Full Text PDFEur J Pediatr
November 2024
Pathophysiology, Diagnosis and Treatments of Bone Diseases, INSERM UMR1033, Lyon, France.
Unlabelled: Cystinosis metabolic bone disease (CMBD) is an emerging concept in infantile nephropathic cystinosis, patients presenting with bone pains, fractures, and deformations during teenage or early adulthood. The underlying mechanisms remain unclear. Our aim was to explore the pro-inflammatory profile of osteoclastic lineage in cystinotic patients.
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