title: Cases of Psychiatric Morbidity in Pediatric Patients After Remission of Cushing Syndrome.
abstract: Endogenous Cushing syndrome (CS) may have different effects in children than what has been described in adults. Previous studies of children and adolescents with CS have identified cognitive decline despite reversal of brain atrophy after remission of CS. Although the observations of parents of children and adolescents with CS support personality changes, significant psychopathology has not been described in the literature. We report 9 children who underwent successful surgery (transsphenoidal surgery [TSS] or resection of bronchial carcinoid) for treatment of CS and subsequently developed significant affective pathology. Affective symptoms included anger-rage outbursts, suicidal ideation, irritability, anxiety, and depression. One child, who committed suicide 60 months after TSS, had recently discontinued antidepressant medication. She had a history of anxiety during active CS and was treated with an anxiolytic. The 7 patients with onset of symptoms within 7 months of TSS were on glucocorticoid replacement, and 1-year follow-up evaluation showed recovery of hypothalamic-pituitary-adrenal axis and biochemical evidence of remission. The 2 patients who presented with onset of symptoms at 48 months or later underwent endocrine evaluation that showed biochemical evidence of remission and normal anterior pituitary hormone levels. This is the first report of affective symptoms and behavioral dysregulation, including suicidal ideation, in a subgroup of children and adolescents after remission of CS. Health care providers caring for children with CS who have been cured should continue to screen for mental illness, monitor for changes in behavior, and refer as appropriate to mental health professionals.
authors.affiliation: Program on Developmental Endocrinology & Genetics and Pediatric Endocrinology Inter-institute Training Program, National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, Maryland; keilm@mail.nih.gov.
authors.forename: Margaret F
authors.initials: MF
authors.lastname: Keil
authors.affiliation: Private practice, Kensington, Maryland; and.
authors.forename: Alan
authors.initials: A
authors.lastname: Zametkin
authors.affiliation: Clinical Center Nursing Department, National Institutes of Health, Bethesda, Maryland.
authors.forename: Celia
authors.initials: C
authors.lastname: Ryder
authors.affiliation: Program on Developmental Endocrinology & Genetics and Pediatric Endocrinology Inter-institute Training Program, National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, Maryland;
authors.forename: Maya
authors.initials: M
authors.lastname: Lodish
authors.affiliation: Program on Developmental Endocrinology & Genetics and Pediatric Endocrinology Inter-institute Training Program, National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, Maryland;
authors.forename: Constantine A
authors.initials: CA
authors.lastname: Stratakis
mesh_terms: D000293:Adolescent; D002648:Child; D015897:Comorbidity; D003480:Cushing Syndrome / Q000175:diagnosis / Q000523:psychology* / Q000601:surgery*; D005260:Female; D006801:Humans; D008297:Male; D001523:Mental Disorders / Q000175:diagnosis / Q000523:psychology*; D012074:Remission Induction / Q000379:methods