2020
DOI: 10.3389/fcell.2020.609643
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An Essential Requirement for Fgf10 in Pinna Extension Sheds Light on Auricle Defects in LADD Syndrome

Abstract: The pinna (or auricle) is part of the external ear, acting to capture and funnel sound toward the middle ear. The pinna is defective in a number of craniofacial syndromes, including Lacrimo-auriculo-dento-digital (LADD) syndrome, which is caused by mutations in FGF10 or its receptor FGFR2b. Here we study pinna defects in the Fgf10 knockout mouse. We show that Fgf10 is expressed in both the muscles and forming cartilage of the developing external ear, with loss of signaling leading to a failure in the normal ex… Show more

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Cited by 7 publications
(4 citation statements)
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“…Additionally, tamoxifen treatment at E15.5 revealed strong fluorescent signal in the pinna of the developing ear as well as in the trachea and in between the cartilage rings (Figure 4C). These two additional expression domains are consistent with sites of Fgf10 expression (Sala et al, 2011;Zhang et al, 2020). We therefore conclude that Cre expression reflects Fgf10 expression and that this line can be used to target FGF10 Pos cells.…”
Section: Validation Of Cre Activity To Label Fgf10 Pos Cells During Embryonic Developmentsupporting
confidence: 73%
“…Additionally, tamoxifen treatment at E15.5 revealed strong fluorescent signal in the pinna of the developing ear as well as in the trachea and in between the cartilage rings (Figure 4C). These two additional expression domains are consistent with sites of Fgf10 expression (Sala et al, 2011;Zhang et al, 2020). We therefore conclude that Cre expression reflects Fgf10 expression and that this line can be used to target FGF10 Pos cells.…”
Section: Validation Of Cre Activity To Label Fgf10 Pos Cells During Embryonic Developmentsupporting
confidence: 73%
“…12 Conditional knockout of Fgf10 in mice was not succeed in recurrencing ear anomalies phenotypes, suggesting that ear deformities is due to network compensatory regulation when single gene deletion. 13 FGFR1 played an important role for localized signaling in the pharyngeal ectoderm and major in the embryological second branchial arch development.…”
Section: Discussionmentioning
confidence: 99%
“…Both mandible and forming cartilage of the development of pinna require enough expression of FGF10, with loss of signaling failing to normal formation of auricle extension and mandible 12 . Conditional knockout of Fgf10 in mice was not succeed in recurrencing ear anomalies phenotypes, suggesting that ear deformities is due to network compensatory regulation when single gene deletion 13 …”
Section: Discussionmentioning
confidence: 99%
“…The outer ear, essential for capturing sound waves and implicated in conductive hearing loss when dysfunctional, is less explored than its middle and inner counterparts 3 . This part, comprising the tragus and helix, is not only important for auditory function but also demonstrates considerable morphological diversity throughout evolution as well as in many types of diseases [4][5][6][7] . Unraveling the molecular mechanisms underlying this diversity is an ongoing field of investigation.…”
Section: Introductionmentioning
confidence: 99%