Zebrafish dscaml1 deficiency impairs retinal patterning and oculomotor function

Manxiu Ma, Alexandro D. Ramirez, Tong Wang, Rachel L. Roberts, Katherine E. Harmon, David Schoppik, Avirale Sharma, Christopher Kuang, Stephanie L. Goei, James A. Gagnon, Steve Zimmerman, Shengdar Q. Tsai, Deepak Reyon, J. Keith Joung, Emre R.F. Aksay, Alexander F. Schier, Y. Albert Pan

Research output: Contribution to journalArticlepeer-review

6 Scopus citations


Down syndrome cell adhesion molecules (dscam and dscaml1) are essential regulators of neural circuit assembly, but their roles in vertebrate neural circuit function are still mostly unexplored. We investigated the functional consequences of dscaml1 deficiency in the larval zebrafish (sexually undifferentiated) oculomotor system, where behavior, circuit function, and neuronal activity can be precisely quantified. Genetic perturbation of dscaml1 resulted in deficits in retinal patterning and light adaptation, consistent with its known roles in mammals. Oculomotor analyses revealed specific deficits related to the dscaml1 mutation, including severe fatigue during gaze stabilization, reduced saccade amplitude and velocity in the light, greater disconjugacy, and impaired fixation. Two-photon calcium imaging of abducens neurons in control and dscaml1 mutant animals confirmed deficits in saccade-command signals (indicative of an impairment in the saccadic premotor pathway), whereas abducens activation by the pretectum-vestibular pathway was not affected. Together, we show that loss of dscaml1 resulted in impairments in specific oculomotor circuits, providing a new animal model to investigate the development of oculomotor premotor pathways and their associated human ocular disorders.

Original languageEnglish (US)
Pages (from-to)143-158
Number of pages16
JournalJournal of Neuroscience
Issue number1
StatePublished - Jan 2 2020


  • Development
  • Dscaml1
  • Eye movement
  • Retina
  • Saccade
  • Zebrafish

ASJC Scopus subject areas

  • Neuroscience(all)


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