Usher syndrome, the most common case of deaf - blindness, may be associated with various psychiatric disorders. Inability of communication through spoken language in association with progressive visual impairment affects diagnostics and management in case of co-morbidity with mental disorder. A patient with Usher syndrome and psychiatric symptoms is described and the difficulties in psychiatric assessment in her case are discussed. A 28 years old woman with hearing impairment diagnosed at the age of 3 months and progressive pigmentary retinopathy diagnosed at the age of 19 years, has been treated for ADHD in childhood, eating disorder in adolescence and psychosis-like disorder in adult life. Direct observation of patient behavior and the effects of pharmacotherapy were the main diagnostic procedures, since the use of sign language and handwriting was very limited. The limitations of management are discussed.

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