A Caucasian woman with a diagnosis of hypopituitarism (GH deficiency, central hypothyroidism, secondary AI, and hypogonadotropic hypogonadism) resulting from a homozygous pathogenic variant c.150del (p.Arg53AspfsTer112) in the PROP1 gene was first referred to our Endocrinology clinic at the age of 19 years. This evidence concerns the gene PROP1 and hypopituitarism.