Age-dependent change in behavioral feature in Rubinstein-Taybi syndrome

Tatsuhiko Yagihashi, Kenjiro Kosaki, Nobuhiko Okamoto, Seiji Mizuno, Kenji Kurosawa, Takao Takahashi, Yuji Sato, Rika Kosaki

Research output: Contribution to journalArticlepeer-review

8 Citations (Scopus)

Abstract

Rubinstein-Taybi syndrome (RTS) is characterized by developmental delay, postnatal growth retardation, typical facial appearance, and broad thumbs and big toes. The behavioral phenotype of children with RTS has been described as friendly and having good social contacts; however, a short attention span and hyperactivity are sometimes present. Little attention has been paid to the behavioral aspects of adults with RTS. We conducted an observational study focusing on behavioral problems in adolescents and adults with RTS compared with children with RTS. A total of 63 patients with RTS and their caretakers answered self-administered questionnaires regarding behavioral features including the Child Behavior Checklist (CBCL). High total CBCL scores were observed, and the mean score was beyond the clinical cut-off point. After stratification into two groups according to age, the older group (≥14years) displayed statistically significant higher scores for Anxious/Depression (P=0.002) and Aggressive Behavior (P=0.036) than the younger group (≤13years). In analyses of single items, statistically significant differences between the younger group and the older group were found for 'Nervous, high-strung, or tense' (31.3% vs 67.7%, P=0.004) and 'Too fearful or anxious' (37.5% vs 64.5%, P=0.032). Here, we showed that the specific behavioral phenotypes of RTS change during adolescence, with anxiety, mood instability, and aggressive behavior emerging as patients age. A clear need exists to follow-up patients with RTS to catch the eventual emergence of psychiatric problems with age. If necessary, pharmacological treatment should be considered.

Original languageEnglish
Pages (from-to)82-86
Number of pages5
JournalCongenital anomalies
Volume52
Issue number2
DOIs
Publication statusPublished - 2012 Jun

Keywords

  • Age-dependent change
  • Behavioral problem
  • Child Behavior Checklist
  • Depression
  • Rubinstein-Taybi syndrome

ASJC Scopus subject areas

  • Medicine(all)

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