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Previous study has identified  SLC7A14  as a new causative gene of retinitis pigmentosa (RP). However, the role of  SLC7A14  has not been fully characterized. The goal of this study was to investigate the biological features of  slc7a14  in zebrafish. To determine the expression of  slc7a14  in developing zebrafish, we performed  in situ  hybridization (ISH) and quantitative real-time PCR. Morpholino knockdown and overexpression experiments were performed to study the role of  slc7a14  in zebrafish retinas. Immunostaining was carried out to observe structural changes. Visual motor responses (VMR) and optokinetic responses (OKR) were analyzed to assess visual behaviors. Terminal deoxynucleotidyl transferase (dUTP) nick-end labeling (TUNEL) staining was performed to survey apoptotic retinal cells. We found that  slc7a14  was highly expressed in neuronal tissues, including the brain, spinal cord and retina, and that the expression levels increased during early embryogenesis. Consistently, ISH showed a similar expression pattern. Knockdown of  slc7a14  led to dose-dependent microphthalmia that was reversed by overexpression. The immunostaining results revealed that the rod-specific protein zpr-3 and the retinal pigment epithelium-specific protein zpr-2 (decreased to 44.48%) were significantly suppressed in the  slc7a14 -silenced morphants. Notably, visual behaviors (the VMR and the OKR) were severely impaired in the  slc7a14 -deficient morphant, especially the VMR OFF response. In addition, apoptotic cells were observed in the retina at 3 days post fertilization (dpf) and 5 dpf by TUNEL assay. Our results demonstrated that  slc7a14  is essential for visually mediated behaviors in zebrafish. Temporary silencing of  slc7a14  in larvae led to severe visual impairments, consistent with the manifestations observed in RP patients. Our findings provide further insights into the genetic mechanisms of RP predisposition caused by  SLC7A14  mutations.

Slc7a14 Is Indispensable in Zebrafish Retinas