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Author E Bryan Crenshaw

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Perspective on the Development of a Large-Scale Clinical Data Repository for Pediatric Hearing Research.

Jeffrey W Pennington, Byron Ruth, Jeffrey M Miller, Joy Peterson, Baichen Xu, Aaron J Masino, Ian Krantz, Juliana Manganella, Tamar Gomes, Derek Stiles, Margaret Kenna, Linda J Hood, John Germiller, E Bryan Crenshaw,

The use of "big data" for pediatric hearing research requires new approaches to both data collection and research methods. The widespread deployment of electronic health record systems creates new opportunities and corresponding challenges in the secondary use of large volumes of audiological and medical data. Opportunities include cost-effective hypothesis generation, ... Read more >>

Ear Hear (Ear and hearing)
[2020, 41(2):231-238]

Cited: 0 times

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Analyses with double knockouts of the Bmpr1a and Bmpr1b genes demonstrate that BMP signaling is involved in the formation of precerebellar mossy fiber nuclei derived from the rhombic lip.

Lihua Qin, Kyung J Ahn, Lara Wine Lee, Charles de Charleroy, E Bryan Crenshaw,

Bone morphogenetic proteins (BMPs) have been hypothesized to specify distinct dorsal neural fates. During neural development, BMPs are expressed in the roof plate and adjacent neuroepithelium. Because several hindbrain nuclei that form the proprioceptive/vestibular/auditory sensory network originate from the rhombic lip, near the roof plate, BMP signaling may regulate the ... Read more >>

PLoS One (PloS one)
[2019, 14(12):e0226602]

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Outcomes of evaluation and testing of 660 individuals with hearing loss in a pediatric genetics of hearing loss clinic.

Devanshi Mehta, Sarah E Noon, Emily Schwartz, Alisha Wilkens, Emma C Bedoukian, Irene Scarano, E Bryan Crenshaw, Ian D Krantz,

Hearing loss is a relatively common condition in children, occurring in approximately 2 out of every 1,000 births with approximately 50% of reported diagnoses having a primary genetic etiology. Given the prevalence and genetic component of hearing loss, coupled with a trend toward early diagnosis with the institution of universal ... Read more >>

Am J Med Genet A (American journal of medical genetics. Part A)
[2016, 170(10):2523-2530]

Cited: 16 times

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Temporal bone radiology report classification using open source machine learning and natural langue processing libraries.

Aaron J Masino, Robert W Grundmeier, Jeffrey W Pennington, John A Germiller, E Bryan Crenshaw,

<h4>Background</h4>Radiology reports are a rich resource for biomedical research. Prior to utilization, trained experts must manually review reports to identify discrete outcomes. The Audiological and Genetic Database (AudGenDB) is a public, de-identified research database that contains over 16,000 radiology reports. Because the reports are unlabeled, it is difficult to select ... Read more >>

BMC Med Inform Decis Mak (BMC medical informatics and decision making)
[2016, 16:65]

Cited: 6 times

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Pou3f4-mediated regulation of ephrin-b2 controls temporal bone development in the mouse.

Steven Raft, Thomas M Coate, Matthew W Kelley, E Bryan Crenshaw, Doris K Wu,

The temporal bone encases conductive and sensorineural elements of the ear. Mutations of POU3F4 are associated with unique temporal bone abnormalities and X-linked mixed deafness (DFNX2/DFN3). However, the target genes and developmental processes controlled by POU3F4 transcription factor activity have remained largely uncharacterized. Ephrin-B2 (Efnb2) is a signaling molecule with ... Read more >>

PLoS One (PloS one)
[2014, 9(10):e109043]

Cited: 7 times

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Harvest: an open platform for developing web-based biomedical data discovery and reporting applications.

Jeffrey W Pennington, Byron Ruth, Michael J Italia, Jeffrey Miller, Stacey Wrazien, Jennifer G Loutrel, E Bryan Crenshaw, Peter S White,

Biomedical researchers share a common challenge of making complex data understandable and accessible as they seek inherent relationships between attributes in disparate data types. Data discovery in this context is limited by a lack of query systems that efficiently show relationships between individual variables, but without the need to navigate ... Read more >>

J Am Med Inform Assoc (Journal of the American Medical Informatics Association : JAMIA)
[2014, 21(2):379-383]

Cited: 11 times

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Harvest: a web-based biomedical data discovery and reporting application development platform.

Michael J Italia, Jeffrey W Pennington, Byron Ruth, Stacey Wrazien, Jennifer G Loutrel, E Bryan Crenshaw, Jeffrey Miller, Peter S White,

Biomedical researchers share a common challenge of making complex data understandable and accessible. This need is increasingly acute as investigators seek opportunities for discovery amidst an exponential growth in the volume and complexity of laboratory and clinical data. To address this need, we developed Harvest, an open source framework that ... Read more >>

AMIA Jt Summits Transl Sci Proc (AMIA Joint Summits on Translational Science proceedings. AMIA Joint Summits on Translational Science)
[2013, 2013:82]

Cited: 0 times

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Bcl11a is required for neuronal morphogenesis and sensory circuit formation in dorsal spinal cord development.

Anita John, Heike Brylka, Christoph Wiegreffe, Ruth Simon, Pentao Liu, René Jüttner, E Bryan Crenshaw, Frank P Luyten, Nancy A Jenkins, Neal G Copeland, Carmen Birchmeier, Stefan Britsch,

Dorsal spinal cord neurons receive and integrate somatosensory information provided by neurons located in dorsal root ganglia. Here we demonstrate that dorsal spinal neurons require the Krüppel-C(2)H(2) zinc-finger transcription factor Bcl11a for terminal differentiation and morphogenesis. The disrupted differentiation of dorsal spinal neurons observed in Bcl11a mutant mice interferes with ... Read more >>

Development (Development (Cambridge, England))
[2012, 139(10):1831-1841]

Cited: 35 times

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Otic mesenchyme cells regulate spiral ganglion axon fasciculation through a Pou3f4/EphA4 signaling pathway.

Thomas M Coate, Steven Raft, Xiumei Zhao, Aimee K Ryan, E Bryan Crenshaw, Matthew W Kelley,

Peripheral axons from auditory spiral ganglion neurons (SGNs) form an elaborate series of radially and spirally oriented projections that interpret complex aspects of the auditory environment. However, the developmental processes that shape these axon tracts are largely unknown. Radial bundles are comprised of dense SGN fascicles that project through otic ... Read more >>

Neuron (Neuron)
[2012, 73(1):49-63]

Cited: 48 times

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The closely related transcription factors Sox4 and Sox11 function as survival factors during spinal cord development.

Daniela C Thein, Johannes M Thalhammer, Anna C Hartwig, E Bryan Crenshaw, Veronique Lefebvre, Michael Wegner, Elisabeth Sock,

Development of the mouse CNS was reported to be normal in the absence of either Sox4 or its close relative Sox11 despite strong and widespread expression of both transcription factors. In this study, we show that combined absence of both Sox proteins in the mouse leads to severe hypoplasia of ... Read more >>

J Neurochem (Journal of neurochemistry)
[2010, 115(1):131-141]

Cited: 48 times

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Wnt signaling is sufficient to perturb oligodendrocyte maturation.

Keith Feigenson, Mary Reid, Jill See, E Bryan Crenshaw, Judith B Grinspan,

The development of oligodendrocytes, the myelinating cells of the central nervous system, is temporally and spatially controlled by local signaling factors acting as inducers or inhibitors. Dorsal spinal cord tissue has been shown to contain inhibitors of oligodendrogliogenesis, although their identity is not completely known. We have studied the actions ... Read more >>

Mol Cell Neurosci (Molecular and cellular neurosciences)
[2009, 42(3):255-265]

Cited: 96 times

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Otic mesenchyme expression of Cre recombinase directed by the inner ear enhancer of the Brn4/Pou3f4 gene.

Kyung J Ahn, Frank Passero, E Bryan Crenshaw,

Brn4/Pou3f4 is a POU-domain transcription factor expressed in the otic mesenchyme that is required for the normal development of the inner ear. In this report, we describe the isolation of an otic mesenchyme enhancer in the Brn4 gene. Subsequently, this enhancer was used to drive the expression of Cre recombinase ... Read more >>

Genesis (Genesis (New York, N.Y. : 2000))
[2009, 47(3):137-141]

Cited: 16 times

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Cooperative function of Tbx1 and Brn4 in the periotic mesenchyme is necessary for cochlea formation.

Evan M Braunstein, E Bryan Crenshaw, Bernice E Morrow, Joe C Adams,

The T-box transcription factor TBX1 has been identified as the major gene responsible for the etiology of velocardiofacial syndrome/DiGeorge syndrome (VCFS/DGS). Conductive hearing loss occurs in a majority of patients with this syndrome, while sensorineural deafness has also been reported in some cases. Mutations in POU3F4/BRN4, a POU domain transcription ... Read more >>

J Assoc Res Otolaryngol (Journal of the Association for Research in Otolaryngology : JARO)
[2008, 9(1):33-43]

Cited: 32 times

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BMP signaling mutant mice exhibit glial cell maturation defects.

Jill See, Polina Mamontov, Kyung Ahn, Lara Wine-Lee, E Bryan Crenshaw, Judith B Grinspan,

Bone morphogenetic proteins have been implicated in the development of oligodendrocytes and astrocytes, however, a role for endogenous BMP signaling in glial development has not been demonstrated in a genetic model. Using mice in which signaling via type I BMP receptors Bmpr1a and Bmpr1b have been inactivated in the neural ... Read more >>

Mol Cell Neurosci (Molecular and cellular neurosciences)
[2007, 35(1):171-182]

Cited: 55 times

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Bmp and Wnt/beta-catenin signals control expression of the transcription factor Olig3 and the specification of spinal cord neurons.

Dietmar Zechner, Thomas Müller, Hagen Wende, Ingrid Walther, Makoto M Taketo, E Bryan Crenshaw, Mathias Treier, Walter Birchmeier, Carmen Birchmeier,

In the developing spinal cord, signals of the roof plate pattern the dorsal progenitor domain and control the specification of three neuron types, dorsal interneurons dI1, dI2, and dI3. Bmp and Wnt/beta-catenin signals as well as transcription factors like Olig3 or Ngn1/2 are essential in this process. We have studied ... Read more >>

Dev Biol (Developmental biology)
[2007, 303(1):181-190]

Cited: 48 times

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Genetic analyses demonstrate that bone morphogenetic protein signaling is required for embryonic cerebellar development.

Lihua Qin, Lara Wine-Lee, Kyung J Ahn, E Bryan Crenshaw,

The cerebellum has been a useful model for studying many aspects of neural development because of its relatively simple cytoarchitecture and developmental program. Yet, the genetic mechanisms underlying early differentiation and patterning of the cerebellum are still poorly characterized. Cell expression studies and culture experiments have suggested the importance of ... Read more >>

J Neurosci (The Journal of neuroscience : the official journal of the Society for Neuroscience)
[2006, 26(7):1896-1905]

Cited: 36 times

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Noncanonical Wnt signaling and neural polarity.

Mireille Montcouquiol, E Bryan Crenshaw, Matthew W Kelley,

The Wnt signaling pathway regulates multiple events in development and disease in both vertebrates and invertebrates. Recently, the noncanonical Wnt signaling cascades, those that do not signal through beta-catenin, have gained prominence for their role in the regulation of cellular polarity. It is not surprising that cellular polarization influences a ... Read more >>

Annu Rev Neurosci (Annual review of neuroscience)
[2006, 29:363-386]

Cited: 102 times

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Abnormal mesenchymal differentiation in the superior semicircular canal of brn4/pou3f4 knockout mice.

Steven E Sobol, Xiuyin Teng, E Bryan Crenshaw,

<h4>Objective</h4>To examine the developmental time course of the mutant phenotype and cellular mechanisms that result in malformations of the superior semicircular canal (SSCC) in Brn4 knockout mice. Mutations in the Brn4/Pou3f4 gene result in characteristic inner ear abnormalities in mutant mouse pedigrees, and the findings in these mice are similar ... Read more >>

Arch Otolaryngol Head Neck Surg (Archives of otolaryngology--head & neck surgery)
[2005, 131(1):41-45]

Cited: 7 times

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Mutation of the POU-domain gene Brn4/Pou3f4 affects middle-ear sound conduction in the mouse.

Daniel S Samadi, James C Saunders, E Bryan Crenshaw,

Mutagenesis of the POU-domain gene Brn4/Pou3f4 causes defects in the cochlear duct, semicircular canal, temporal bone and stapes footplate. The footplate defect suggested a middle-ear conductive component to the hearing loss associated with this mutation. This was examined by measuring velocity transfer functions at the umbo of wild type and ... Read more >>

Hear Res (Hearing research)
[2005, 199(1-2):11-21]

Cited: 14 times

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The role of Brn4/Pou3f4 and Pax6 in forming the pancreatic glucagon cell identity.

R Scott Heller, Doris A Stoffers, Aihua Liu, Andreas Schedl, E Bryan Crenshaw, Ole D Madsen, Palle Serup,

Brain 4 (Brn4/Pou3f4) and Pax6 are POU-homeodomain and paired-homeodomain transcription factors, respectively, that are expressed in the brain and the glucagon-expressing cells in the pancreas. Brn4 expression begins at embryonic day 10 in the pancreas, just before pax6 and both appear in the glucagon immunoreactive cells. At a later time ... Read more >>

Dev Biol (Developmental biology)
[2004, 268(1):123-134]

Cited: 58 times

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Signaling through BMP type 1 receptors is required for development of interneuron cell types in the dorsal spinal cord.

Lara Wine-Lee, Kyung J Ahn, Rory D Richardson, Yuji Mishina, Karen M Lyons, E Bryan Crenshaw,

During spinal cord development, distinct classes of interneurons arise at stereotypical locations along the dorsoventral axis. In this paper, we demonstrate that signaling through bone morphogenetic protein (BMP) type 1 receptors is required for the formation of two populations of commissural neurons, DI1 and DI2, that arise within the dorsal ... Read more >>

Development (Development (Cambridge, England))
[2004, 131(21):5393-5403]

Cited: 97 times

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Bone morphogenetic protein 4 (BMP4): a regulator of capsule chondrogenesis in the developing mouse inner ear.

Wei Liu, Seung Ha Oh, Yong koo Kang Yk, Geming Li, Thao M Doan, Markus Little, Lijun Li, Kyung Ahn, E Bryan Crenshaw, Dorothy A Frenz,

Formation of the cartilaginous otic capsule is directed by otic epithelial-periotic mesenchymal interactions. In response to induction by otic epithelium, condensations of mesenchyme appear in the periotic region and form a chondrified otic capsule that serves as the template for the subsequent formation of the endochondral bony labyrinth. Previous studies ... Read more >>

Dev Dyn (Developmental dynamics : an official publication of the American Association of Anatomists)
[2003, 226(3):427-438]

Cited: 38 times

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Epithelial Bmpr1a regulates differentiation and proliferation in postnatal hair follicles and is essential for tooth development.

Thomas Andl, Kyung Ahn, Alladin Kairo, Emily Y Chu, Lara Wine-Lee, Seshamma T Reddy, Nirvana J Croft, Judith A Cebra-Thomas, Daniel Metzger, Pierre Chambon, Karen M Lyons, Yuji Mishina, John T Seykora, E Bryan Crenshaw, Sarah E Millar,

Bone morphogenetic protein (BMP) signaling is thought to perform multiple functions in the regulation of skin appendage morphogenesis and the postnatal growth of hair follicles. However, definitive genetic evidence for these roles has been lacking. Here, we show that Cre-mediated mutation of the gene encoding BMP receptor 1A in the ... Read more >>

Development (Development (Cambridge, England))
[2004, 131(10):2257-2268]

Cited: 225 times

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Regulation of outgrowth and apoptosis for the terminal appendage: external genitalia development by concerted actions of BMP signaling [corrected].

Kentaro Suzuki, Daniel Bachiller, YiPing P Chen, Mami Kamikawa, Hidenao Ogi, Ryama Haraguchi, Yukiko Ogino, Yasuhiro Minami, Yuji Mishina, Kyung Ahn, E Bryan Crenshaw, Gen Yamada,

Extra-corporal fertilization depends on the formation of copulatory organs: the external genitalia. Coordinated growth and differentiation of the genital tubercle (GT), an embryonic anlage of external genitalia, generates a proximodistally elongated structure suitable for copulation, erection, uresis and ejaculation. Despite recent progress in molecular embryology, few attempts have been made ... Read more >>

Development (Development (Cambridge, England))
[2003, 130(25):6209-6220]

Cited: 78 times

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Genetic interaction between Wnt/beta-catenin and BMP receptor signaling during formation of the AER and the dorsal-ventral axis in the limb.

Natalia Soshnikova, Dietmar Zechner, Joerg Huelsken, Yuji Mishina, Richard R Behringer, Makoto M Taketo, E Bryan Crenshaw, Walter Birchmeier,

By conditional gene ablation in mice, we found that beta-catenin, an essential downstream effector of canonical Wnt signaling, is a key regulator of formation of the apical ectodermal ridge (AER) and of the dorsal-ventral axis of the limbs. By generation of compound mutants, we also show that beta-catenin acts downstream ... Read more >>

Genes Dev (Genes & development)
[2003, 17(16):1963-1968]

Cited: 86 times

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