Development of multisensory integration following prolonged early-onset visual deprivationHighlights d Congenitally blind individuals regaining sight late acquire multisensory integration abilities d The ability to integrate vision and touch develops quickly after surgery d Some individuals reach optimal integration levels within years to benefit perception d The development is based on experience and depends on post-surgical visual acuity

We can estimate the veridical size of nearby objects reasonably well irrespective of their viewing distance. This perceptual capability, termed size constancy, is accomplished by combining information about retinal image size together with the viewing distance, or using the relational information available in the scene, via direct perception [1]. A previous study [2] showed that children typically underestimate the size of a distant object. This underestimation is reduced with time, suggesting that years of visual experience may be essential for attaining true size constancy. But what if you have had very limited vision during the early years of life? We studied 23 Ethiopian children suffering from bilateral, early-onset cataract, who were surgically treated only years after birth. Surprisingly, most children were able to estimate object size reasonably well irrespective of distance; in fact, they usually tended to overestimate the far-object size. Closer examination indicated that, although before surgery the patients were diagnosed as having a full, mature bilateral cataract, they nevertheless had some residual form of vision, typically limited to very close range. Gandhi et al.[3] earlier reported immediate susceptibility to geometric visual illusions in a similar group of newly-sighted children, concluding that size constancy was probably innate. We suggest that their immediate ability to judge physical size irrespective of distance is more likely to result from their previous visual experience.

Genetic defects in the three SH3 and multiple ankyrin repeat domains (SHANK) genes (SHANK1, SHANK2, and SHANK3) are associated with multiple major neuropsychiatric disorders, including autism spectrum disorder (ASD), schizophrenia (SCZ), and bipolar disorder (BPD). Psychostimulant-induced hyperactivity is a commonly applied paradigm to assess behavioral phenotypes related to BPD and considered to be the gold standard for modeling mania-like elevated drive in mouse models. Therefore, the goal of our present study was to test whether Shank1 plays a role in the behavioral effects of psychostimulants and whether this is associated with genotype-dependent neurochemical alterations. To this aim, male and female null mutant Shank1-/- mice were treated with d-amphetamine (AMPH; 2.5 mg/kg) and 3,4-methylenedioxymethamphetamine (MDMA, commonly known as ecstasy; 20 mg/kg), and psychostimulant-induced hyperactivity was compared to heterozygous Shank1+/- and wildtype Shank1+/+ littermate controls. Results show that Shank1-/- mice display reduced psychostimulant-induced hyperactivity, although psychostimulants robustly stimulated locomotor activity in littermate controls. Shank1 deletion effects emerged throughout development, were particularly prominent in adulthood, and seen in response to both psychostimulants, i.e., AMPH and MDMA. Specifically, while AMPH-induced hyperactivity was reduced but still detectable in Shank1-/- mice, MDMA-induced hyperactivity was robustly blocked and completely absent in Shank1-/- mice. Reduced efficacy of psychostimulants to stimulate hyperactivity in Shank1-/- mice might be associated with alterations in the neurochemical architecture in prefrontal cortex, nucleus accumbens, and hypothalamus. Our observation that psychostimulant-induced hyperactivity is reduced rather than enhanced in Shank1-/- mice clearly speaks against a behavioral phenotype with relevance to BPD. Lack of BPD-like phenotype is consistent with currently available human data linking mutations in SHANK2 and SHANK3 but not SHANK1 to BPD.