A surgical approach appropriate for targeted cochlear gene therapy in the mouse

Jero, Jussi; Tseng, Charles J.; Mhatre, Anand N.; Lalwani, Anil K.

doi:10.1016/s0378-5955(00)00216-1

Cited by 87 publications

(75 citation statements)

References 14 publications

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“…The Callithrix sp is promising in the application of these study models since surgically we have been able to locate the same anatomical structures mentioned in these papers [1][2][3][4][5][6][7][8][9][10] . However, some details must be stressed.…”

Section: Discussionmentioning

confidence: 99%

“…We did not see a stapedial artery that crosses over the round window niche as described in the rat5, absent in humans, and this would also represent an easiness to work in this region. Through posterior antrostomy we have access to the vestibule windows, where we could, for example, carry out gene transfer to the inner ear, as proposed in rats 5 . We see a cochlea made up of 2.5 turns, similar to that in humans (2.5-2.75 turns), and different from that of mice (1.5 turn) and guinea pigs (4.5 turns)6, and this means another advantage.…”

Section: Discussionmentioning

confidence: 99%

“…Otology also requires such progress, and many experimental models are reported in the literature. Small size mammals such as rats, mice and guinea pigs [1][2][3][4][5] are commonly used in studies of temporal bones, and their anatomy and physiology are broadly described. Other mammals such as racoons 6 and pigs 7 are also well defined experimental models.…”

Section: Introductionmentioning

confidence: 99%

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Estudo anatômico do osso temporal de um primata não-humano (Callithrix sp)

Borin

Covolan

Mello

et al. 2008

Rev. Bras. Otorrinolaringol.

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The search for experimental (animal) models is essential to the development of clinical studies. Aim: To demonstrate, by means of micro dissection techniques, the anatomical structures of temporal bones from the primate Callithrix sp. Study design: Experimental. Methods: Dissection of temporal bone structures of Callithrix sp and photographic documentation. Results: We identified the main constituents of the temporal bone (external, medium and inner ear and facial nerve). Conclusion: The non-human primate Callithrix sp. is an adequate experimental model for the studies of temporal bone structures given its close anatomical similarities to that found in humans.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Estudo anatômico do osso temporal de um primata não-humano (Callithrix sp)

Borin

Covolan

Mello

et al. 2008

Rev. Bras. Otorrinolaringol.

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“…Body temperature was maintained with a heat pad controlled by an animal temperature controller and monitored continuously. The cochlea was accessed via a longitudinal, ventral, paramedian approach, as described previously (58). After exposure of the bulla, a small opening was made with the help of forceps and a microdrill, taking care that the tympanic membrane and the stapes…”

Section: Methodsmentioning

confidence: 99%

Tricellulin deficiency affects tight junction architecture and cochlear hair cells

Nayak¹,

Lee²,

Yousaf³

et al. 2013

J. Clin. Invest.

116

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The two compositionally distinct extracellular cochlear fluids, endolymph and perilymph, are separated by tight junctions that outline the scala media and reticular lamina. Mutations in TRIC (also known as MARVELD2), which encodes a tricellular tight junction protein known as tricellulin, lead to nonsyndromic hearing loss (DFNB49). We generated a knockin mouse that carries a mutation orthologous to the TRIC coding mutation linked to DFNB49 hearing loss in humans. Tricellulin was absent from the tricellular junctions in the inner ear epithelia of the mutant animals, which developed rapidly progressing hearing loss accompanied by loss of mechanosensory cochlear hair cells, while the endocochlear potential and paracellular permeability of a biotin-based tracer in the stria vascularis were unaltered. Freeze-fracture electron microscopy revealed disruption of the strands of intramembrane particles connecting bicellular and tricellular junctions in the inner ear epithelia of tricellulin-deficient mice. These ultrastructural changes may selectively affect the paracellular permeability of ions or small molecules, resulting in a toxic microenvironment for cochlear hair cells. Consistent with this hypothesis, hair cell loss was rescued in tricellulin-deficient mice when generation of normal endolymph was inhibited by a concomitant deletion of the transcription factor, Pou3f4. Finally, comprehensive phenotypic screening showed a broader pathological phenotype in the mutant mice, which highlights the non-redundant roles played by tricellulin.

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“…Bullectomy and cochleostomy was performed on all rats via a retroauricular approach (10). Preoperative antibiotherapy (cefazolin at 500 mg/kg daily, IM) for prophylaxis and IM cyclosporine (15 mg/kg daily) from preoperative day 2 until postoperative day 7 to prevent immune reactions were administered to all animals (11).…”

Section: Transplantation Into the Cochleamentioning

confidence: 99%

Study of mouse induced pluripotent stem cell transplantation intoWistar albino rat cochleae after hair cell damage*

et al. 2016

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Background/aim: As the regeneration capacity of hair cells is limited, inner ear stem cell therapies hold promise. Effects of mouse induced pluripotent stem cells (IPSCs) on Wistar albino rats (WARs) with hearing impairment were investigated.Materials and methods: Thirty-five adult WARs with normal hearing were divided into 4 groups. Excluding the study group (n = 15), the other three groups served as control groups for ototoxicity and IPSC injection models. IPSC injections were performed via cochleostomy after a retroauricular approach. Auditory functions were evaluated with auditory brainstem responses (ABRs) before and after the injections. After a final hearing assessment the WARs were sacrificed and cochleae were extracted to see the biologic behavior of IPSCs in the inner ear by light microscopy and immunohistochemistry.Results: There were no significant differences in the click-ABR thresholds in the study group after IPSC transplantation. The mean hearing threshold in the study group after ototoxic agent injection was 53.2 dB (10-90 dB). There was no significant difference between groups (P > 0.05) and no differentiated stem cells were observed immunohistochemically. Conclusion:Transplanted IPSCs did not show a therapeutic effect in this trial. We discuss potential pitfalls and factors affecting the therapeutic effect.

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A surgical approach appropriate for targeted cochlear gene therapy in the mouse

Cited by 87 publications

References 14 publications

Estudo anatômico do osso temporal de um primata não-humano (Callithrix sp)

Estudo anatômico do osso temporal de um primata não-humano (Callithrix sp)

Tricellulin deficiency affects tight junction architecture and cochlear hair cells

Study of mouse induced pluripotent stem cell transplantation intoWistar albino rat cochleae after hair cell damage*

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