In this paper, a third form of Alder anomaly is described. In addition to the morphological changes which are characteristic of this anomaly, the peroxidase of the neutrophil polymorphonuclear leukocytes (NPNL) is abnormal in its properties. It is resistant to the fixation step in the May-Grünwald Giemsa procedure and is similar, therefore, to the eosinophil peroxidase. In the phenotype observed, 2 genetic changes are probably involved: one is a change in the regulation of NPNL granulation, the other is a mutation in the myeloperoxidase structural gene. In the light of these new findings, 3 forms of the anomaly are now known: the original Alder anomaly, in which the abnormal granulation, particularly in the granulocytes, is not linked with any hereditary disorder; the Alder-Reilly form, in which in addition to the abnormal granulation the leukocytes also contain inclusion bodies, the phenomenon sometimes being associated with mucopolysaccharidosis (gargoylism), and the third form of anomaly described here, where-apart from the abnormal granulation-the myeloperoxidase behaves differently, resembling that of eosinophils in its resistance to methanol fixation.
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http://dx.doi.org/10.1159/000206110 | DOI Listing |
Genet Med Open
July 2024
Department of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.
Purpose: is one of the most frequently mutated genes in intellectual disability cohorts. Thus, far few adult-aged patients with -related disorder have been described, which limits our understanding of the disease's natural history and our ability to counsel patients and their families.
Methods: Data on patients aged 18+ years with -related disorder were collected through an online questionnaire completed by clinicians and parents.
Pediatr Surg Int
November 2024
Institute of Systems Molecular and Integrative Biology, University of Liverpool, Liverpool, UK.
This article highlights the evolution, birth and legacy of the world's first neonatal surgical unit established at Alder Hey Children's Hospital Liverpool in 1953. Peter Paul Rickham, a creative pioneering and innovative surgeon, is credited here as a major driving force that helped shape and progress the modern day development of neonatal surgery. Rickham's vision was realised by studying neonatal surgical disorders and the mortality rate of congenital anomalies in Mersey Region while working as a young senior registrar with Isabella Forshall.
View Article and Find Full Text PDFSci Total Environ
December 2024
Department of Earth and Environmental Sciences, University of Milano-Bicocca, Milan, Italy; National Biodiversity Future Center (NBFC), Palermo, Italy.
To our knowledge, there has not been a review article summarizing the current evidence with regard to perinatal risk factors, and our aim is to perform a systematic review and meta-analysis of the evidence of perinatal risk factors in single suture craniosynostosis to inform our practice and identify any need for further research in this area. Our target population was pediatric single-suture craniosynostosis patients, and the intervention was perinatal risk factors. The comparison group was an age and sex-matched control group without craniosynostosis and the outcome we investigated was presence of single suture craniosynostosis.
View Article and Find Full Text PDFHGG Adv
January 2025
Amsterdam UMC, Department of Human Genetics, University of Amsterdam, Amsterdam, the Netherlands; Amsterdam Reproduction & Development Research Institute, Amsterdam, the Netherlands. Electronic address:
Neurodevelopmental disorder with or without autism or seizures (NEDAUS) is a neurodevelopmental disorder characterized by global developmental delay, speech delay, seizures, autistic features, and/or behavior abnormalities. It is caused by CUL3 (Cullin-3 ubiquitin ligase) haploinsufficiency. We collected clinical and molecular data from 26 individuals carrying pathogenic variants and variants of uncertain significance (VUS) in the CUL3 gene, including 20 previously unreported cases.
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