TY - JOUR
T1 - RBMX gene is essential for brain development in zebrafish
AU - Tsend-Ayush, Enkhjargal
AU - O'Sullivan, Lynda A.
AU - Grützner, Frank S.
AU - Onnebo, Sara M.N.
AU - Lewis, Rowena S.
AU - Delbridge, Margaret L.
AU - Graves, Jennifer A.Marshall
AU - Ward, Alister C.
PY - 2005/11
Y1 - 2005/11
N2 - The human RBMX gene was discovered recently through its homology to the spermatogenesis candidate gene RBMY. Its position on the human X chromosome suggests that it may be involved in X-linked mental retardation syndromes. However, to date there is scant information on the in vivo role of RBMX. To address this issue, we have isolated a zebrafish rbmx orthologue and characterized its embryonic expression pattern. Zebrafish rbmx is maternally expressed and then widely expressed in the embryo up to 24 hr postfertilization. In later stages of embryonic development, rbmx transcripts are localized predominantly in the brain, branchial arches, and liver primordium. The function of rbmx during embryonic development was examined by the use of an antisense morpholino targeting rbmx. The rbmx-morphants displayed an underdeveloped head and eyes, reduced body size, defective somite patterning, and absence of jaws. Furthermore, in the absence of functional rbmx, expression of specific markers for the fore- and hindbrain (otx2, krox20) was severely reduced. These studies demonstrate for the first time that rbmx is required for normal embryonic development, in particular of the brain, consistent with a role in X-linked mental retardation.
AB - The human RBMX gene was discovered recently through its homology to the spermatogenesis candidate gene RBMY. Its position on the human X chromosome suggests that it may be involved in X-linked mental retardation syndromes. However, to date there is scant information on the in vivo role of RBMX. To address this issue, we have isolated a zebrafish rbmx orthologue and characterized its embryonic expression pattern. Zebrafish rbmx is maternally expressed and then widely expressed in the embryo up to 24 hr postfertilization. In later stages of embryonic development, rbmx transcripts are localized predominantly in the brain, branchial arches, and liver primordium. The function of rbmx during embryonic development was examined by the use of an antisense morpholino targeting rbmx. The rbmx-morphants displayed an underdeveloped head and eyes, reduced body size, defective somite patterning, and absence of jaws. Furthermore, in the absence of functional rbmx, expression of specific markers for the fore- and hindbrain (otx2, krox20) was severely reduced. These studies demonstrate for the first time that rbmx is required for normal embryonic development, in particular of the brain, consistent with a role in X-linked mental retardation.
KW - Brain
KW - Embryogenesis
KW - Morpholino
KW - Ventricle
KW - Zebrafish
KW - hnRNPG
KW - rbmx
UR - http://www.scopus.com/inward/record.url?scp=27444433335&partnerID=8YFLogxK
U2 - 10.1002/dvdy.20432
DO - 10.1002/dvdy.20432
M3 - Article
SN - 1058-8388
VL - 234
SP - 682
EP - 688
JO - Developmental Dynamics
JF - Developmental Dynamics
IS - 3
ER -