Helmholtz Gemeinschaft

Search
Browse
Statistics
Feeds

Teashirt 1 (Tshz1) is essential for the development, survival and function of hypoglossal and phrenic motor neurons in mouse

[thumbnail of Original Article]
Preview
PDF (Original Article) - Requires a PDF viewer such as GSview, Xpdf or Adobe Acrobat Reader
9MB
[thumbnail of Supplementary Information]
Preview
PDF (Supplementary Information) - Requires a PDF viewer such as GSview, Xpdf or Adobe Acrobat Reader
5MB

Item Type:Article
Title:Teashirt 1 (Tshz1) is essential for the development, survival and function of hypoglossal and phrenic motor neurons in mouse
Creators Name:Chaimowicz, C., Ruffault, P.L., Chéret, C., Woehler, A., Zampieri, N., Fortin, G., Garratt, A.N. and Birchmeier, C.
Abstract:Feeding and breathing are essential motor functions and rely on the activity of hypoglossal and phrenic motor neurons that innervate the tongue and diaphragm, respectively. Little is known about the genetic programs that control the development of these neuronal subtypes. The transcription factor Tshz1 is strongly and persistently expressed in developing hypoglossal and phrenic motor neurons. We used conditional mutation of Tshz1 in the progenitor zone of motor neurons (Tshz1(MNΔ)) to show that Tshz1 is essential for survival and function of hypoglossal and phrenic motor neurons. Hypoglossal and phrenic motor neurons are born in correct numbers, but many die between embryonic day 13.5 and 14.5 in Tshz1(MNΔ) mutant mice. In addition, innervation and electrophysiological properties of phrenic and hypoglossal motor neurons are altered. Severe feeding and breathing problems accompany this developmental deficit. Although motor neuron survival can be rescued by elimination of the pro-apoptotic factor Bax, innervation, feeding and breathing defects persist in Bax(-/-); Tshz1(MNΔ) mutants. We conclude that Tshz1 is an essential transcription factor for the development and physiological function of phrenic and hypoglossal motor neurons.
Keywords:Tshz1, Motor Neurons, Phrenic Motor Column, Hypoglossal Nucleus, Aerophagia, Breathing, Animals, Mice
Source:Development
ISSN:0950-1991
Publisher:Company of Biologists
Volume:146
Number:17
Page Range:dev174045
Date:6 September 2019
Official Publication:https://doi.org/10.1242/dev.174045
PubMed:View item in PubMed

Repository Staff Only: item control page

Downloads

Downloads per month over past year

Open Access
MDC Library