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Article ; Online: Finding a window for gene therapy for hereditary deafness.

Ivanchenko, Maryna V / Corey, David P

Proceedings of the National Academy of Sciences of the United States of America

2023 Volume 120, Issue 39, Page(s) e2311864120

MeSH term(s)	Animals ; Mice ; Transcriptional Activation ; Hearing Loss ; Deafness/genetics ; Deafness/therapy ; Hearing Loss, Sensorineural ; Genetic Therapy ; Anion Transport Proteins
Chemical Substances	Spns2 protein, mouse ; Anion Transport Proteins
Language	English
Publishing date	2023-09-13
Publishing country	United States
Document type	Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't ; Comment
ZDB-ID	209104-5
ISSN	1091-6490 ; 0027-8424
ISSN (online)	1091-6490
ISSN	0027-8424
DOI	10.1073/pnas.2311864120
Database	MEDical Literature Analysis and Retrieval System OnLINE

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Zs.A 1148: Show issues

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Article ; Online: Viral vectors for gene delivery to the inner ear.

Maguire, Casey A / Corey, David P

Hearing research

2020 Volume 394, Page(s) 107927

Abstract: Gene therapy using virus vectors to treat hereditary diseases has made remarkable progress in the past decade. There are FDA-approved products for ex-vivo gene therapy for diseases such as immunodeficiencies (e.g., SCID), and in vivo gene therapy for a ... ...

Abstract	Gene therapy using virus vectors to treat hereditary diseases has made remarkable progress in the past decade. There are FDA-approved products for ex-vivo gene therapy for diseases such as immunodeficiencies (e.g., SCID), and in vivo gene therapy for a rare blindness and neuro-muscular disease. Gene therapy for hereditary hearing loss has picked up pace in the past five years due to progress in understanding disease gene function as well as the development of better technologies such as adeno-associated virus (AAV) vectors, to deliver nucleic acid to target cells in the inner ear. This review has two major goals. One is to review the state of the art for investigators already working in preclinical cochlear gene therapy. The other is to present the language of vectorology and important considerations for designing and using AAV vectors to inner ear neurobiologists who might use AAV vectors in the cochlea for either therapeutic or basic biological applications.
MeSH term(s)	Dependovirus/genetics ; Ear, Inner ; Gene Transfer Techniques ; Genetic Therapy ; Genetic Vectors
Language	English
Publishing date	2020-02-23
Publishing country	Netherlands
Document type	Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't ; Review
ZDB-ID	282629-x
ISSN	1878-5891 ; 0378-5955
ISSN (online)	1878-5891
ISSN	0378-5955
DOI	10.1016/j.heares.2020.107927
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Zs.A 1467: Show issues

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Article: Electron Microscopy Techniques for Investigating Structure and Composition of Hair-Cell Stereociliary Bundles.

Ivanchenko, Maryna V / Indzhykulian, Artur A / Corey, David P

Frontiers in cell and developmental biology

2021 Volume 9, Page(s) 744248

Abstract: Hair cells-the sensory cells of the vertebrate inner ear-bear at their apical surfaces a bundle of actin-filled protrusions called stereocilia, which mediate the cells' mechanosensitivity. Hereditary deafness is often associated with morphological ... ...

Abstract	Hair cells-the sensory cells of the vertebrate inner ear-bear at their apical surfaces a bundle of actin-filled protrusions called stereocilia, which mediate the cells' mechanosensitivity. Hereditary deafness is often associated with morphological disorganization of stereocilia bundles, with the absence or mislocalization within stereocilia of specific proteins. Thus, stereocilia bundles are closely examined to understand most animal models of hereditary hearing loss. Because stereocilia have a diameter less than a wavelength of light, light microscopy is not adequate to reveal subtle changes in morphology or protein localization. Instead, electron microscopy (EM) has proven essential for understanding stereocilia bundle development, maintenance, normal function, and dysfunction in disease. Here we review a set of EM imaging techniques commonly used to study stereocilia, including optimal sample preparation and best imaging practices. These include conventional and immunogold transmission electron microscopy (TEM) and scanning electron microscopy (SEM), as well as focused-ion-beam scanning electron microscopy (FIB-SEM), which enables 3-D serial reconstruction of resin-embedded biological structures at a resolution of a few nanometers. Parameters for optimal sample preparation, fixation, immunogold labeling, metal coating and imaging are discussed. Special attention is given to protein localization in stereocilia using immunogold labeling. Finally, we describe the advantages and limitations of these EM techniques and their suitability for different types of studies.
Language	English
Publishing date	2021-10-22
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2737824-X
ISSN	2296-634X
ISSN	2296-634X
DOI	10.3389/fcell.2021.744248
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Article: Fully resorbable poly-4-hydroxybutyrate (P4HB) mesh for soft tissue repair and reconstruction: A scoping review.

Deeken, Corey R / Chen, David C / Lopez-Cano, Manuel / Martin, David P / Badhwar, Amit

Frontiers in surgery

2023 Volume 10, Page(s) 1157661

Abstract: Background: Poly-4-hydroxybutyrate (P4HB) is a fully resorbable, biologically-produced polymer with a strength and flexibility comparable to permanent synthetic polymers. The objective was to identify/summarize all peer-reviewed publications involving ... ...

Abstract	Background: Poly-4-hydroxybutyrate (P4HB) is a fully resorbable, biologically-produced polymer with a strength and flexibility comparable to permanent synthetic polymers. The objective was to identify/summarize all peer-reviewed publications involving P4HB mesh. Methods: A scoping review was conducted within PubMed and included articles published through October 2022. Results: A total of Conclusion: P4HB mesh provides long-term hernia repair strength and exhibits promising clinical outcomes beyond its resorption period. Future studies should include randomized controlled trials comparing P4HB to other biomaterials, as well as optimal patient selection, operative technique, long-term outcomes, minimization of potential mesh-related complications, and potential contraindications/complications for P4HB in hernia/abdominal wall reconstruction.
Language	English
Publishing date	2023-04-12
Publishing country	Switzerland
Document type	Journal Article ; Review
ZDB-ID	2773823-1
ISSN	2296-875X
ISSN	2296-875X
DOI	10.3389/fsurg.2023.1157661
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Article: The auditory midbrain mediates tactile vibration sensing.

Huey, Erica L / Turecek, Josef / Delisle, Michelle M / Mazor, Ofer / Romero, Gabriel E / Dua, Malvika / Sarafis, Zoe K / Hobble, Alexis / Booth, Kevin T / Goodrich, Lisa V / Corey, David P / Ginty, David D

bioRxiv : the preprint server for biology

2024

Abstract: Vibrations are ubiquitous in nature, shaping behavior across the animal kingdom. For mammals, mechanical vibrations acting on the body are detected by mechanoreceptors of the skin and deep tissues and processed by the somatosensory system, while sound ... ...

Abstract	Vibrations are ubiquitous in nature, shaping behavior across the animal kingdom. For mammals, mechanical vibrations acting on the body are detected by mechanoreceptors of the skin and deep tissues and processed by the somatosensory system, while sound waves traveling through air are captured by the cochlea and encoded in the auditory system. Here, we report that mechanical vibrations detected by the body's Pacinian corpuscle neurons, which are unique in their ability to entrain to high frequency (40-1000 Hz) environmental vibrations, are prominently encoded by neurons in the lateral cortex of the inferior colliculus (LCIC) of the midbrain. Remarkably, most LCIC neurons receive convergent Pacinian and auditory input and respond more strongly to coincident tactile-auditory stimulation than to either modality alone. Moreover, the LCIC is required for behavioral responses to high frequency mechanical vibrations. Thus, environmental vibrations captured by Pacinian corpuscles are encoded in the auditory midbrain to mediate behavior.
Language	English
Publishing date	2024-04-06
Publishing country	United States
Document type	Preprint
DOI	10.1101/2024.03.08.584077
Database	MEDical Literature Analysis and Retrieval System OnLINE

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Article ; Online: A chemo-mechano-biological modeling framework for cartilage evolving in health, disease, injury, and treatment.

Rahman, Muhammed Masudur / Watton, Paul N / Neu, Corey P / Pierce, David M

Computer methods and programs in biomedicine

2023 Volume 231, Page(s) 107419

Abstract: Background and objective: Osteoarthritis (OA) is a pervasive and debilitating disease, wherein degeneration of cartilage features prominently. Despite extensive research, we do not yet understand the cause or progression of OA. Studies show biochemical, ...

Abstract	Background and objective: Osteoarthritis (OA) is a pervasive and debilitating disease, wherein degeneration of cartilage features prominently. Despite extensive research, we do not yet understand the cause or progression of OA. Studies show biochemical, mechanical, and biological factors affect cartilage health. Mechanical loads influence synthesis of biochemical constituents which build and/or break down cartilage, and which in turn affect mechanical loads. OA-associated biochemical profiles activate cellular activity that disrupts homeostasis. To understand the complex interplay among mechanical stimuli, biochemical signaling, and cartilage function requires integrating vast research on experimental mechanics and mechanobiology-a task approachable only with computational models. At present, mechanical models of cartilage generally lack chemo-biological effects, and biochemical models lack coupled mechanics, let alone interactions over time. Methods: We establish a first-of-its kind virtual cartilage: a modeling framework that considers time-dependent, chemo-mechano-biologically induced turnover of key constituents resulting from biochemical, mechanical, and/or biological activity. We include the "minimally essential" yet complex chemical and mechanobiological mechanisms. Our 3-D framework integrates a constitutive model for the mechanics of cartilage with a novel model of homeostatic adaptation by chondrocytes to pathological mechanical stimuli, and a new application of anisotropic growth (loss) to simulate degradation clinically observed as cartilage thinning. Results: Using a single set of representative parameters, our simulations of immobilizing and overloading successfully captured loss of cartilage quantified experimentally. Simulations of immobilizing, overloading, and injuring cartilage predicted dose-dependent recovery of cartilage when treated with suramin, a proposed therapeutic for OA. The modeling framework prompted us to add growth factors to the suramin treatment, which predicted even better recovery. Conclusions: Our flexible framework is a first step toward computational investigations of how cartilage and chondrocytes mechanically and biochemically evolve in degeneration of OA and respond to pharmacological therapies. Our framework will enable future studies to link physical activity and resulting mechanical stimuli to progression of OA and loss of cartilage function, facilitating new fundamental understanding of the complex progression of OA and elucidating new perspectives on causes, treatments, and possible preventions.
MeSH term(s)	Humans ; Cartilage, Articular/pathology ; Suramin/pharmacology ; Models, Biological ; Osteoarthritis/metabolism ; Osteoarthritis/pathology ; Chondrocytes/pathology ; Chondrocytes/physiology
Chemical Substances	Suramin (6032D45BEM)
Language	English
Publishing date	2023-02-18
Publishing country	Ireland
Document type	Journal Article
ZDB-ID	632564-6
ISSN	1872-7565 ; 0169-2607
ISSN (online)	1872-7565
ISSN	0169-2607
DOI	10.1016/j.cmpb.2023.107419
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Zs.B 521: Show issues

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Article ; Online: Modulation of Gene Expression in the Eye with Antisense Oligonucleotides.

Hu, Jiaxin / Gong, Xin / Fan, Yan / Aguilar, Selina / Rigo, Frank / Prakash, Thahza P / Corey, David R / Mootha, V Vinod

Nucleic acid therapeutics

2023 Volume 33, Issue 6, Page(s) 339–347

Abstract: One advantage of antisense oligonucleotides (ASOs) for drug development is their long-lasting gene knockdown after ... ...

Abstract	One advantage of antisense oligonucleotides (ASOs) for drug development is their long-lasting gene knockdown after administration
MeSH term(s)	Mice ; Animals ; Oligonucleotides, Antisense/genetics ; Oligonucleotides, Antisense/pharmacology ; Retina ; Endothelium, Corneal ; Gene Expression
Chemical Substances	Oligonucleotides, Antisense
Language	English
Publishing date	2023-11-02
Publishing country	United States
Document type	Journal Article
ZDB-ID	2639888-6
ISSN	2159-3345 ; 2159-3337
ISSN (online)	2159-3345
ISSN	2159-3337
DOI	10.1089/nat.2023.0044
Database	MEDical Literature Analysis and Retrieval System OnLINE

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Book: Sensory transduction

Corey, David P.

Marine Biological Laboratory, Woods Hole, Massachusetts, 5 - 8 September 1991

(... annual symposium / Society of General Physiologists ; 45 ; Society of General Physiologists series ; 47)

1992

Institution	Marine Biological Laboratory
Author's details	ed. by David P. Corey
Series title	... annual symposium / Society of General Physiologists ; 45 Society of General Physiologists series ; 47 Annual meeting / Society of General Physiologists
Collection	Annual meeting / Society of General Physiologists
Keywords	Perception / congresses ; Sensation / congresses ; Sinnesphysiologie ; Sensorik ; Erregungsleitung
Subject	Reizleitung ; Nervenleitung ; Sensorisches System
Language	English
Size	VII, 424 S. : Ill., graph. Darst.
Publisher	Rockefeller Univ. Pr
Publishing place	New York
Publishing country	United States
Document type	Book
HBZ-ID	HT004261698
ISBN	0-87470-051-5 ; 978-0-87470-051-0
Database	Catalogue ZB MED Medicine, Health

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Article ; Online: Concussion Symptoms and Neurocognitive Performance of Children and Adolescents on Antidepressants.

Daley, Mary M / Howell, David R / Lanois, Corey J / Berkner, Paul D / Mannix, Rebekah C / Oldham, Jessie R / Meehan, William P

Medicine and science in sports and exercise

2024

Abstract: Introduction/purpose: There is a well-established association between pre-existing depression/anxiety and greater post-concussion symptom burden, but the potential impact of antidepressant medications has not been fully explored. The primary objective ... ...

Abstract	Introduction/purpose: There is a well-established association between pre-existing depression/anxiety and greater post-concussion symptom burden, but the potential impact of antidepressant medications has not been fully explored. The primary objective of this study was to compare pre-injury/baseline and post-injury concussion symptom scores and neurocognitive performance of athletes on antidepressant medications, both with healthy controls and with those with depression/anxiety not on antidepressants. Methods: This is a cross-sectional study using data collected from 49,270 junior and high school athletes from computerized neurocognitive assessments (Immediate Post-Concussion Assessment and Cognitive Test [ImPACT]) administered between 2009 and 2018 held by the Massachusetts Concussion Management Coalition. The main outcome measures were symptom scores and neurocognitive performance measures, all of which were assessed both at baseline and post-injury. Statistical analysis included analysis of variance and Tukey pairwise comparisons for continuous variables and Fisher's exact test for categorical variables. Multivariate regression models were used to adjust for potential confounding variables. Results: Both at baseline and post-injury, athletes with depression/anxiety had mean total symptom scores that were more than double that of healthy controls regardless of antidepressant use. While there were no significant differences in neurocognitive performance at baseline, depression/anxiety was associated with small but significant decreases in post-injury visual memory and visual motor scores. Conclusions: Both at baseline and after sustaining a concussion, young athletes with depression/anxiety experience significantly greater symptom burden compared with healthy controls regardless of antidepressant use.
Language	English
Publishing date	2024-01-18
Publishing country	United States
Document type	Journal Article
ZDB-ID	603994-7
ISSN	1530-0315 ; 0195-9131 ; 0025-7990
ISSN (online)	1530-0315
ISSN	0195-9131 ; 0025-7990
DOI	10.1249/MSS.0000000000003383
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Zs.A 869: Show issues

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Article ; Online: Rescue of hearing by adenine base editing in a humanized mouse model of Usher syndrome type 1F.

Peters, Cole W / Hanlon, Killian S / Ivanchenko, Maryna V / Zinn, Eric / Linarte, Elizabeth F / Li, Yaqiao / Levy, Jonathan M / Liu, David R / Kleinstiver, Benjamin P / Indzhykulian, Artur A / Corey, David P

Molecular therapy : the journal of the American Society of Gene Therapy

2023 Volume 31, Issue 8, Page(s) 2439–2453

Abstract: Usher syndrome type 1F (USH1F), characterized by congenital lack of hearing and balance and progressive loss of vision, is caused by mutations in the PCDH15 gene. In the Ashkenazi population, a recessive truncation mutation accounts for a large ... ...

Abstract	Usher syndrome type 1F (USH1F), characterized by congenital lack of hearing and balance and progressive loss of vision, is caused by mutations in the PCDH15 gene. In the Ashkenazi population, a recessive truncation mutation accounts for a large proportion of USH1F cases. The truncation is caused by a single C→T mutation, which converts an arginine codon to a stop (R245X). To test the potential for base editors to revert this mutation, we developed a humanized Pcdh15
MeSH term(s)	Mice ; Animals ; Usher Syndromes/genetics ; Usher Syndromes/therapy ; Gene Editing ; Mutation ; Hearing/genetics ; Cadherins/genetics
Chemical Substances	Cadherins
Language	English
Publishing date	2023-06-12
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't ; Research Support, N.I.H., Extramural
ZDB-ID	2010592-7
ISSN	1525-0024 ; 1525-0016
ISSN (online)	1525-0024
ISSN	1525-0016
DOI	10.1016/j.ymthe.2023.06.007
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