ArticlePloS one2010
Carboxypeptidase A6 in zebrafish development and implications for VIth cranial nerve pathfinding.
Article in PloS one, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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Who cites it
15 citing papers in PubMed, 29 citations in OpenAlex.
- Cell-type brain-region specific changes in prefrontal cortex of a mouse model of alcohol dependence.Neurobiology of disease · 2024Article
- Enhanced Production of ECM Proteins for Pharmaceutical Applications Using Mammalian Cells and Sodium Heparin Supplementation.Pharmaceutics · 2022Article
- Fxr signaling and microbial metabolism of bile salts in the zebrafish intestine.Science advances · 2021Article
- The BMP signaling gradient is interpreted through concentration thresholds in dorsal-ventral axial patterning.PLoS biology · 2021Article
- Carboxypeptidase A6 was identified and validated as a novel potential biomarker for predicting the occurrence of active ulcerative colitis.Journal of cellular and molecular medicine · 2020Article
- Identification of DEP domain-containing proteins by a machine learning method and experimental analysis of their expression in human HCC tissues.Scientific reports · 2016Article
- Article
- Positional plasticity in regenerating Amybstoma mexicanum limbs is associated with cell proliferation and pathways of cellular differentiation.BMC developmental biology · 2015Article
- Novel carboxypeptidase A6 (CPA6) mutations identified in patients with juvenile myoclonic and generalized epilepsy.PloS one · 2015Article
- Strabismus genetics across a spectrum of eye misalignment disorders.Clinical genetics · 2014Review
- The extreme anterior domain is an essential craniofacial organizer acting through Kinin-Kallikrein signaling.Cell reports · 2014Article
- Carboxypeptidases in disease: insights from peptidomic studies.Proteomics. Clinical applications · 2014Review
- 8q12 microduplication including CHD7: clinical report on a new patient with Duane retraction syndrome type 3.Molecular cytogenetics · 2013Article
- Naturally occurring carboxypeptidase A6 mutations: effect on enzyme function and association with epilepsy.The Journal of biological chemistry · 2012Article
- Duplication 8q12: confirmation of a novel recognizable phenotype with duane retraction syndrome and developmental delay.European journal of human genetics : EJHG · 2012Article
Corrections and comments
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Authors and funding
4 authors at 2 institutions in 1 country.
Funding
Abstract
Carboxypeptidase A6 (CPA6) is an extracellular protease that cleaves carboxy-terminal hydrophobic amino acids and has been implicated in the defective innervation of the lateral rectus muscle by the VIth cranial nerve in Duane syndrome. In order to investigate the role of CPA6 in development, in particular its potential role in axon guidance, the zebrafish ortholog was identified and cloned. Zebrafish CPA6 was secreted and interacted with the extracellular matrix where it had a neutral pH optimum and specificity for C-terminal hydrophobic amino acids. Transient mRNA expression was found in newly formed somites, pectoral fin buds, the stomodeum and a conspicuous condensation posterior to the eye. Markers showed this tissue was not myogenic in nature. Rather, the CPA6 localization overlapped with a chondrogenic site which subsequently forms the walls of a myodome surrounding the lateral rectus muscle. No other zebrafish CPA gene exhibited a similar expression profile. Morpholino-mediated knockdown of CPA6 combined with retrograde labeling and horizontal eye movement analyses demonstrated that deficiency of CPA6 alone did not affect either VIth nerve development or function in the zebrafish. We suggest that mutations in other genes and/or enhancer elements, together with defective CPA6 expression, may be required for altered VIth nerve pathfinding. If mutations in CPA6 contribute to Duane syndrome, our results also suggest that Duane syndrome can be a chondrogenic rather than a myogenic or neurogenic developmental disorder.
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