ABSTRACT
BACKGROUND: Anorexia Nervosa is highly comorbid with depressive, anxiety, and obsessive-compulsive spectrum disorders. However, it has not previously been reported as comorbid with antisocial personality traits, except when substance use disorder is also identified. We present an unusual case of a patient with resistant anorexia nervosa and comorbid conduct disorder. This case was also unique in that the juvenile justice system was involved during treatment. CASE PRESENTATION: A 13-year-old female was admitted to our pediatric hospital for the treatment of anorexia nervosa. She had a history of violent behaviors toward family members, often jeopardizing her care. During hospitalization, she physically attacked a physician on her care team shortly before she transitioned to an eating disorders treatment program. She was diagnosed with conduct disorder, and following discharge, she attacked her father in a premeditated act. This led to her entry into the juvenile justice system. While under the custody of the juvenile justice system, she was readmitted to our hospital for further treatment of anorexia nervosa. Our treatment strategy included psychotropics, positive reinforcement, close interdisciplinary coordination among the various hospital teams, and the juvenile justice system. Following discharge from her second hospitalization back to the juvenile detention system, our patient maintained a healthy weight and appeared to show improvements in the cognitive distortions related to her eating disorder. CONCLUSIONS: To our knowledge, this is the first reported successful treatment of an individual with resistant anorexia nervosa and conduct disorder. It was likely a combination of weight gain, psychotropic medications, and the structured milieu provided by the juvenile justice system that led to the effective treatment of our patient. This case illustrates that a non-traditional healthcare setting can be an asset to treatment through persistence and close collaboration across institutions.
ABSTRACT
BACKGROUND: Recent advances in understanding molecular and synaptic mechanisms of intellectual disabilities (ID) in fragile X syndrome (FXS) and Down syndrome (DS) through animal models have led to targeted controlled trials with pharmacological agents designed to normalize these underlying mechanisms and improve clinical outcomes. However, several human clinical trials have failed to demonstrate efficacy of these targeted treatments to improve surrogate behavioral endpoints. Because the ultimate index of disease modification in these disorders is amelioration of ID, the validation of cognitive measures for tracking treatment response is essential. Here, we present preliminary research to validate the National Institutes of Health Toolbox Cognitive Battery (NIH-TCB) for ID. METHODS: We completed three pilot studies of patients with FXS (total n = 63; mean age 19.3 ± 8.3 years, mean mental age 5.3 ± 1.6 years), DS (n = 47; mean age 16.1 ± 6.2, mean mental age 5.4 ± 2.0), and idiopathic ID (IID; n = 16; mean age 16.1 ± 5.0, mean mental age 6.6 ± 2.3) measuring processing speed, executive function, episodic memory, word/letter reading, receptive vocabulary, and working memory using the web-based NIH-TB-CB, addressing feasibility, test-retest reliability, construct validity, ecological validity, and syndrome differences and profiles. RESULTS: Feasibility was good to excellent (≥80 % of participants with valid scores) for above mental age 4 years for all tests except list sorting (working memory). Test-retest stability was good to excellent, and convergent validity was similar to or better than results obtained from typically developing children in the normal sample for executive function and language measures. Examination of ecological validity revealed moderate to very strong correlations between the NIH-TCB composite and adaptive behavior and full-scale IQ measures. Syndrome/group comparisons demonstrated significant deficits for the FXS and DS groups relative to IID on attention and inhibitory control, a significant reading weakness for FXS, and a receptive vocabulary weakness for DS. CONCLUSIONS: The NIH-TCB has potential for assessing important dimensions of cognition in persons with ID, and several tests may be useful for tracking response to intervention. However, more extensive psychometric studies, evaluation of the NIH-TCB's sensitivity to change, both developmentally and in the context of treatment, and perhaps establishing links to brain function in these populations, are required to determine the true utility of the battery as a set of outcome measures.
