Allogeneic hematopoietic stem cell transplantation (HSCT) is the effective mean of immune restoration in severe combined immunodefiency (SCID). Usually, HSCT without cytoreductive conditioning is attempted. Nevertheless, conditioning procedures are still preferred in a subset of patients. Herein, we describe the immunological outcome in a cohort of conditioned and unconditioned patients, from diagnosis, through transplantation, to follow-up. This retrospective study was conducted on 17 patients with SCID (10 conditioned, 7 unconditioned) who later underwent HSCT. Immune reconstitution was assessed in the post-transplant year by quantification of T cell receptor excision circles (TRECs) and kappa-deleting recombination excision circles (KRECs), among additional laboratory and clinical evaluations. Unconditioned patients were diagnosed and transplanted earlier. TREC and KREC quantification showed a gradual increase in both groups, with higher levels in the conditioned group. Engraftment percentages differed drastically between groups, favoring the conditioned group. Unconditioned patients were significantly more dependent on intravenous immunoglobulins (IVIGs). One patient from each group succumbed to disease complications. Conditioning demonstrated superior laboratorial outcomes. Patients with unique characteristics (i.e., consanguinity, Bacillus Calmette-Guérin vaccination, impaired access to IVIG) may require personalized considerations. The effort to implement secondary prevention of SCID with newborn screening should continue.
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Cells
December 2024
Oujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision, and Brain Health), Institute of Mental Health and Drug Discovery, School of Mental Health, Wenzhou Medical University, Ouhai District, Wenzhou 325000, China.
Background: glucocorticoids may play an important role in the formation of fear memory, which is relevant to the neurobiology of post-traumatic stress disorder (PTSD). In our previous study, we showed the glucocorticoid receptor (GR) forms a protein complex with FKBP51, which prevents translocation of GR into the nucleus to affect gene expression; this complex is elevated in PTSD patients and by fear-conditioned learning in mice, and disrupting this complex blocks the storage and retrieval of fear-conditioned memories. The timing of release of glucocorticoid relative to the formation of a traumatic memory could be important in this process, and remains poorly understood.
View Article and Find Full Text PDFNeurobiol Dis
January 2025
Université Paris-Saclay, CNRS, Institut des Neurosciences Paris Saclay, 91400 Saclay, France. Electronic address:
Duchenne muscular dystrophy (DMD) is associated with a range of cognitive and behavioral problems. Brain-related comorbidities show clinical heterogeneity depending on the position of the mutation within the multi-promoter dystrophin (DMD) gene, likely due to the differential impact of mutations on the expression of distinct brain dystrophins. A deficiency of the full-length brain dystrophin, Dp427, has been associated with enhanced stress reactivity, characterized by abnormal fear responses in both patients and mdx mouse model.
View Article and Find Full Text PDFNutrients
December 2024
Department of Physical Medicine and Rehabilitation, Faculty of Medicine, University of Debrecen, 4026 Debrecen, Hungary.
Background: Obesity poses an enormous public health and economic burden worldwide. Visceral fat accumulation is associated with various metabolic and cardiovascular consequences, resulting in an increased prevalence of atherosclerotic conditions. We aimed to examine the impact of low-and moderate-intensity aerobic training on several anthropometric and cardiorespiratory parameters and markers of atherosclerosis, including inflammation, serum levels of lipoproteins and adipokines of extremely obese patients in poor condition.
View Article and Find Full Text PDFJACC Cardiovasc Interv
November 2024
Interventional Cardiology, Hospital Clínico San Carlos, IdISSC, Madrid, Spain; Faculty of Medicine, Universidad Complutense de Madrid, Madrid, Spain; Cardiology, Hospital Universitario de Torrejón, Madrid, Spain; Faculty of Biomedical and Health Sciences, Universidad Europea de Madrid, Madrid, Spain. Electronic address:
J Cancer Policy
December 2024
Santa Maria della Misericordia University Hospital, ASUFC, Piazzale Santa Maria della Misericordia 15, Udine 33100, Italy.
Objective: Precision Oncology requires deep changes in organizational settings but little evidence has been identified about the best strategy to guarantee the delivery of this innovation to patients. In the Italian health care system, high heterogeneity could jeopardize equal access opportunity for patients. Following a consensus method, we aim to define shared solutions to address these issues in clinical practice.
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