Anthony Monaco is a well-respected researcher in the field of neurogenetics. He is currently the principle scientist and director of the Wellcome Trust Centre for Human Genetics (Oxford, UK). Under his direction, the center has become a leading institute for the study of complex genetic diseases. Monaco is responsible for elegant work on the cloning of Duchenne muscular dystrophy, as well as classic and insightful work during the early days of gene cloning.

From the beginning of his career, Monaco was intrigued by the brain and behavior. He therefore majored in neuroscience as an undergraduate at Princeton University. His early research as an M.D./Ph.D student at Harvard Medical School in the mid-1980s led to the isolation of candidate genes that cause Duchenne muscular dystrophy, results that were published in the highly influential scientific journal, nature. In 1988, after receiving his graduate degrees, Monaco decided to forego his medical internship training and instead moved to London to work at the Imperial Cancer Research Fund, where he became involved in the human genome project in its early stages. In 1990, he moved to Oxford. Eight years later, he was appointed director of the human genetics center, whose focus lies in uncovering the genetic basis behind complex but common diseases.

His current research interests lie in the genetic basis of neurodevelopmental disorders in children, particularly, autism, dyslexia, and specific language impairment. His research in the area of language impairment involves finding the genes that are behind the difficulties that 2-5% of children have in acquiring language skills, despite normal intelligence and adequate opportunity. In particular, his research group is interested in the pathways that lead to the disorders. His group has mapped the gene involved in a rare speech and language disorder to a small region on chromosome 7, work that should lead to the isolation of the causal gene itself. Other diseases that his group studies include chorea acanthocytosis, X-linked Dystonia-Parkinsons, Menkes disease, and Charcot-Marie-Tooth peripheral neuropathy. In December 1998, Monaco received the Medical Research Council Award for his work on the genetics of autism.