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Cleft Palate, Moderate Lung Developmental Retardation and Early Postnatal Lethality in Mice Deficient in the Kir7.1 Inwardly Rectifying K+ Channel.

Villanueva S, Burgos J, López-Cayuqueo KI, Lai KM, Valenzuela DM, Cid LP, Sepúlveda FV - PLoS ONE (2015)

Bottom Line: Kir7.1 is present in epithelial tissues where it colocalizes with the Na+/K+-pump probably serving to recycle K+ taken up by the pump.Kir7.1 is expressed in the epithelium covering the palatal processes at the time at which palate sealing takes place and our results suggest it might play an essential role in late palatogenesis.Our work also reveals a second unexpected role in the development and the physiology of the respiratory system, where Kir7.1 is expressed in epithelial cells all along the respiratory tree.

View Article: PubMed Central - PubMed

Affiliation: Centro de Estudios Científicos (CECs), Valdivia, Chile.

ABSTRACT
Kir7.1 is an inwardly rectifying K+ channel of the Kir superfamily encoded by the kcnj13 gene. Kir7.1 is present in epithelial tissues where it colocalizes with the Na+/K+-pump probably serving to recycle K+ taken up by the pump. Human mutations affecting Kir7.1 are associated with retinal degeneration diseases. We generated a mouse lacking Kir7.1 by ablation of the Kcnj13 gene. Homozygous mutant mice die hours after birth and show cleft palate and moderate retardation in lung development. Kir7.1 is expressed in the epithelium covering the palatal processes at the time at which palate sealing takes place and our results suggest it might play an essential role in late palatogenesis. Our work also reveals a second unexpected role in the development and the physiology of the respiratory system, where Kir7.1 is expressed in epithelial cells all along the respiratory tree.

No MeSH data available.


Related in: MedlinePlus

Expression of Kir7.1 in the developing palate of the mouse.Coronal sections of embryo heads at E14.5, E15.5 and E18.5 (a) or postnatal day P0 (b). Kcnj13+/- and Kcnj13-/- samples show β-galactosidase staining (dark blue) in cells that express Kir7.1. a. β-galactosidase activity can be observed at E15.5 mainly in respiratory epithelium and at the palatine process (pp) at E18.5 (enlarged view in inset). The choroid plexus (chp) is shown as positive control at E14.5. b. β-galactosidase activity in mice at P0 is observed in palatal (pe) (nasal side), respiratory (re) and olfactory epithelium (oe). Each section was photographed at 40X magnification. ns: nasal septum, p: palate, T: tongue.
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pone.0139284.g006: Expression of Kir7.1 in the developing palate of the mouse.Coronal sections of embryo heads at E14.5, E15.5 and E18.5 (a) or postnatal day P0 (b). Kcnj13+/- and Kcnj13-/- samples show β-galactosidase staining (dark blue) in cells that express Kir7.1. a. β-galactosidase activity can be observed at E15.5 mainly in respiratory epithelium and at the palatine process (pp) at E18.5 (enlarged view in inset). The choroid plexus (chp) is shown as positive control at E14.5. b. β-galactosidase activity in mice at P0 is observed in palatal (pe) (nasal side), respiratory (re) and olfactory epithelium (oe). Each section was photographed at 40X magnification. ns: nasal septum, p: palate, T: tongue.

Mentions: We undertook a histological examination of the developing palate in Kcjn13+/+ and Kcjn13-/- embryos. Fig 6a shows medial coronal sections that reveal the palatal processes apposed but not fused at E14.5 in both Kcjn13+/+ and Kcjn13-/-, and already fused at E15.5 in the WT but not in the mutant mouse. The sites of Ki7.1 expression were identified by β-galactosidase staining of Kcjn13-/- mice. Kir7.1 expression is absent at 14.5 dpc, a stage at which robust expression is already present in the choroid plexus (see inset). Kir 7.1 expression is evident at E15.5 in the respiratory epithelium as well as that covering the palatal processes but here only in the nasal side. The same is seen at E18.5, where the higher magnification picture corroborates that Kir7.1 expression stops towards the tip of the palatal process. Fig 6b shows that Kir7.1 expression persists after birth and covers the epithelium of the nasopharinx in the non-cleft palate Kcjn13+/- tissue. Expression is also present in the respiratory and olphactory epithelia after birth as seen in the Kcjn13-/- section.


Cleft Palate, Moderate Lung Developmental Retardation and Early Postnatal Lethality in Mice Deficient in the Kir7.1 Inwardly Rectifying K+ Channel.

Villanueva S, Burgos J, López-Cayuqueo KI, Lai KM, Valenzuela DM, Cid LP, Sepúlveda FV - PLoS ONE (2015)

Expression of Kir7.1 in the developing palate of the mouse.Coronal sections of embryo heads at E14.5, E15.5 and E18.5 (a) or postnatal day P0 (b). Kcnj13+/- and Kcnj13-/- samples show β-galactosidase staining (dark blue) in cells that express Kir7.1. a. β-galactosidase activity can be observed at E15.5 mainly in respiratory epithelium and at the palatine process (pp) at E18.5 (enlarged view in inset). The choroid plexus (chp) is shown as positive control at E14.5. b. β-galactosidase activity in mice at P0 is observed in palatal (pe) (nasal side), respiratory (re) and olfactory epithelium (oe). Each section was photographed at 40X magnification. ns: nasal septum, p: palate, T: tongue.
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Related In: Results  -  Collection

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pone.0139284.g006: Expression of Kir7.1 in the developing palate of the mouse.Coronal sections of embryo heads at E14.5, E15.5 and E18.5 (a) or postnatal day P0 (b). Kcnj13+/- and Kcnj13-/- samples show β-galactosidase staining (dark blue) in cells that express Kir7.1. a. β-galactosidase activity can be observed at E15.5 mainly in respiratory epithelium and at the palatine process (pp) at E18.5 (enlarged view in inset). The choroid plexus (chp) is shown as positive control at E14.5. b. β-galactosidase activity in mice at P0 is observed in palatal (pe) (nasal side), respiratory (re) and olfactory epithelium (oe). Each section was photographed at 40X magnification. ns: nasal septum, p: palate, T: tongue.
Mentions: We undertook a histological examination of the developing palate in Kcjn13+/+ and Kcjn13-/- embryos. Fig 6a shows medial coronal sections that reveal the palatal processes apposed but not fused at E14.5 in both Kcjn13+/+ and Kcjn13-/-, and already fused at E15.5 in the WT but not in the mutant mouse. The sites of Ki7.1 expression were identified by β-galactosidase staining of Kcjn13-/- mice. Kir7.1 expression is absent at 14.5 dpc, a stage at which robust expression is already present in the choroid plexus (see inset). Kir 7.1 expression is evident at E15.5 in the respiratory epithelium as well as that covering the palatal processes but here only in the nasal side. The same is seen at E18.5, where the higher magnification picture corroborates that Kir7.1 expression stops towards the tip of the palatal process. Fig 6b shows that Kir7.1 expression persists after birth and covers the epithelium of the nasopharinx in the non-cleft palate Kcjn13+/- tissue. Expression is also present in the respiratory and olphactory epithelia after birth as seen in the Kcjn13-/- section.

Bottom Line: Kir7.1 is present in epithelial tissues where it colocalizes with the Na+/K+-pump probably serving to recycle K+ taken up by the pump.Kir7.1 is expressed in the epithelium covering the palatal processes at the time at which palate sealing takes place and our results suggest it might play an essential role in late palatogenesis.Our work also reveals a second unexpected role in the development and the physiology of the respiratory system, where Kir7.1 is expressed in epithelial cells all along the respiratory tree.

View Article: PubMed Central - PubMed

Affiliation: Centro de Estudios Científicos (CECs), Valdivia, Chile.

ABSTRACT
Kir7.1 is an inwardly rectifying K+ channel of the Kir superfamily encoded by the kcnj13 gene. Kir7.1 is present in epithelial tissues where it colocalizes with the Na+/K+-pump probably serving to recycle K+ taken up by the pump. Human mutations affecting Kir7.1 are associated with retinal degeneration diseases. We generated a mouse lacking Kir7.1 by ablation of the Kcnj13 gene. Homozygous mutant mice die hours after birth and show cleft palate and moderate retardation in lung development. Kir7.1 is expressed in the epithelium covering the palatal processes at the time at which palate sealing takes place and our results suggest it might play an essential role in late palatogenesis. Our work also reveals a second unexpected role in the development and the physiology of the respiratory system, where Kir7.1 is expressed in epithelial cells all along the respiratory tree.

No MeSH data available.


Related in: MedlinePlus