More than 60% of prelingual deafness is genetic in origin, and of these up to 95% are monogenic autosomal recessive traits. Causal mutations have been identified in 1 of 38 different genes in a subset of patients with nonsyndromic autosomal recessive deafness. In this study, we screened 49 unrelated Turkish families with at least three affected children born to consanguineous parents. Probands from all families were negative for mutations in the GJB2 gene, two large deletions in the GJB6 gene, and the 1555A>G substitution in the mitochondrial DNA MTRNR1 gene. Each family was subsequently screened via autozygosity mapping with genomewide single-nucleotide polymorphism arrays. If the phenotype cosegregated with a haplotype flanking one of the 38 genes, mutation analysis of the gene was performed. We identified 22 different autozygous mutations in 11 genes, other than GJB2, in 26 of 49 families, which overall explains deafness in 62% of families. Relative frequencies of genes following GJB2 were MYO15A (9.9%), TMIE (6.6%), TMC1 (6.6%), OTOF (5.0%), CDH23 (3.3%), MYO7A (3.3%), SLC26A4 (1.7%), PCDH15 (1.7%), LRTOMT (1.7%), SERPINB6 (1.7%), and TMPRSS3 (1.7%). Nineteen of 22 mutations are reported for the first time in this study. Unknown rare genes for deafness appear to be present in the remaining 23 families.
Download full-text PDF |
Source |
---|---|
http://dx.doi.org/10.1089/gtmb.2010.0120 | DOI Listing |
AJOG Glob Rep
February 2025
Department of Obstetrics and Gynecology, Northwestern University Feinberg School of Medicine, Chicago, IL (Steinberg, Young, Strom, Andebrhan, Perry, Barry, Holder, Roque, and Yee).
Background: In obstetrics and gynecology (OBGYN) research, gender disparities permeate through leadership, funding, promotion, mentorship, publishing, compensation, and publicity. Few studies have investigated OBGYN clinical trial leadership as it relates to investigator gender. Thus, we undertook an investigation of principal investigator (PI) gender and clinical trial success.
View Article and Find Full Text PDFCureus
December 2024
Surgical Oncology, Mahamana Pandit Madan Mohan Malaviya Cancer Centre, Homi Bhabha Cancer Hospital, Tata Memorial Centre, Varanasi, IND.
Thrombocytopenia is a common complication in patients with solid tumors, particularly renal cell carcinoma (RCC), arising from mechanisms such as chemotherapy, direct tumor invasion, and paraneoplastic syndromes. Managing thrombocytopenia in advanced cancer presents significant challenges, often limiting therapeutic options and impacting patient outcomes. This case report describes a 62-year-old man with metastatic RCC complicated by persistent thrombocytopenia, unresponsive to both conventional treatments and novel therapies.
View Article and Find Full Text PDFOrphanet J Rare Dis
January 2025
Paediatric Orthopaedics, Deformity Reconstruction and Foot Surgery, General Orthopaedics and Tumour Orthopaedics, Muenster University Hospital, Albert-Schweitzer-Campus 1, Muenster, 48149, Germany.
Background: Sprengel deformity is a rare congenital malformation of the scapula defined by malposition during embryonic development. Affected individuals have limited range of motion of the shoulder and torticollis. Surgical reconstruction is an option to treat patients with severe deformity and functional impairment.
View Article and Find Full Text PDFAddiction
January 2025
Family and Community Medicine, University of California San Francisco, San Francisco, CA, USA.
Background And Aims: Opioid smoking is becoming more common in the United States. The aim of this analysis was to estimate relative mortality risk among those who primarily smoke opioids compared with those who inject.
Design: Retrospective propensity score-matched cohort analysis.
Int J Drug Policy
January 2025
Centre de recherche du Centre Hospitalier de l'Université de Montréal (CRCHUM), 900 rue St-Denis, Montréal, Québec H2X 0A9, Canada; Department of Emergency and Family Medicine, Université de Montréal, 2900 blvd Edouard Montpetit, Montréal, Québec H3T 1J4, Canada; National Drug and Alcohol Research Centre, UNSW Sydney, Anzac Parade, Kensington NSW 2052, Australia. Electronic address:
Background: Supervised injection sites (SIS) offer a hygienic environment in which people can inject drugs under observation; as such, these harm reduction services have been on the forefront of the overdose epidemic. We sought to understand factors predictive of an overdose requiring an emergency response intervention at SIS in Montréal, Canada.
Methods: We used administrative data from all four Montréal SIS from 1 March 2018 - 31 October 2022 to first calculate the rate of onsite overdose requiring intervention (e.
Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!