We report a 61‐year‐old female with 22q11.2 deletion syndrome (22q11.2DS) and a novel heterozygous nonsense variant in MAP1A, identified through whole‐genome sequencing (WGS), who presented with intellectual developmental disorder, treatment‐resistant schizophrenia, and multiple congenital anomalies. This evidence concerns the gene MAP1A and schizophrenia.