Despite their widespread use, organophosphate pesticides cause developmental neurotoxicity through a mechanism based on their function as an acetylcholinesterase inhibitor. Animals in early developmental stages are especially vulnerable to developmental neurotoxicity induced by organophosphate pesticides. The embryos and larvae of sea urchin provide a promising invertebrate model system for evaluating developmental neurotoxicity induced by organophosphate pesticides, as they develop quickly with well-characterized morphological and biochemical features, possess similar processes of neurogenesis and signaling cascades to chordates, and are vulnerable to pollutants. Both the neurodevelpment of sea urchin during early development stage and the regulation on the early development were included. Particularly, the neurotoxic effects of organophosphate pesticides on the early development and its potential neurotoxicity mechanism for sea urchin are discussed, and future prospects of this field are provided.