Some science: searching for brain protective proteins and aiming to unravel genes shaping our brains, at the turn of the century, we have published our first paper describing the cloning and characterization of a novel complementary DNA encoding a protein, which we called activity-dependent neuroprotective protein (ADNP)(Bassan et al. 1999). We went on to clone the human ADNP (hADNP) from a fetal brain cDNA library. Comparative sequence analysis of mouse and man indicated 90% identity with the mouse (Zamostiano et al. 2001). Our recent analysis revealed high conservation and appearance only in vertebrates, indicating an important function in higher organisms, with a complex brain structure (Gozes et al. 2015). We further showed that the hADNP gene structure spans~ 40 kilobases and includes five exons and four introns with alternative splicing of an untranslated second exon (chromosome 20q12-13.2, a region associated with aggressive tumor growth). As we eluded to in 2001 (Zamostiano et al. 2001), and later described in detail, hADNP is also mutated in cancer (Gozes et al. 2015). In an attempt to identify novel genes responsible for autism, a first de novo p. Lys408Valfs* 31 mutation in the ADNP gene was identified in a large cohort of autistic patients (O’Roak et al. 2012b). Sequencing of the 209 families in this cohort did not reveal a second mutation in ADNP nor hardly in any other candidate gene; thus, a large-scale resequencing study was initiated (O’Roak et al. 2012a). In this study of 2446 probands, an additional p. Tyr719* de novo ADNP mutation was identified. By combining the data from WES and targeted resequencing studies initiated in multiple centers, Helsmoortel et al.(2014) identified a total of 10 patients with mutations in ADNP, including the two patients identified in both earlier studies. As all patients suffered from autism and shared characteristic facial features, it could be concluded that mutations in ADNP cause a syndromic form of autism. Remarkably, ADNP is one of a relatively small group of genes that appear to lead to autism in a substantial proportion of cases. Immediately following our initial report, two additional patients were identified that share many of the reported characteristics (Pescosolido et al. 2014; Vandeweyer et al. 2014). On top of that, Coe et al.(2014) reported five patients with a truncating ADNP mutation in a screening of 4716 patients with autism/ID. Most recently, De Rubeis et al.(2014) identified three more patients of a total of 3871 screened, and the DDD project reported four novel cases out of 1133 screened (Deciphering Developmental Disorders 2015). Taking advantage of mouse genetics, we have shown that ADNP is crucial for brain formation (Pinhasov et al. 2003) and learning and memory (Vulih-Shultzman et al. 2007), in a sex-dependent manner (Malishkevich et al. 2015; Kleiman et al. 2015). Using biochemical and cell biology techniques,