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. Author manuscript; available in PMC: 2010 Feb 1.

Published in final edited form as: Am J Med Genet A. 2009 Mar;149A(3):302–314. doi: 10.1002/ajmg.a.32652

FIG. 5 — Quantification of social behavior in Subject 5889 versus WS, Down syndrome (DS) and controls (typically developed, TD). Data replotted from Doyle et al., 2004 and Järvinen-Pasley et al., 2008. A: The atypical social behavior of Subject 5889 was reflected in parental ratings of social behavior using the SISQ at age 2.55 [Doyle et al., 2004, versus WS only] and were repeated at age 4.6 years [Järvinen-Pasley et al., 2008 versus a single typical WS case only]. In contrast with age-matched typical WS group, as well as children with DS and TD, the parental ratings (converted to z-scores for comparison) revealed that 5889's Global Sociability and Approach Strangers scores both at 2.55 years (z = -2.59 and -2.39, respectively) and at 4.86 years (z = -2.6 and -3.2), were significantly different from her peers in the typical WS group. B: A number of key differences in direct social behavior were noted between the Subject 5889 and children with typical WS deletions [shown in Järvinen-Pasley et al., 2008], relative to control children with DS for the mean total durations of three ethogram behaviors. Overall, WS children with full deletions were significantly less likely than children with DS to spend time far away in proximity from (p=.007), and to engage in non-social activities with (p=.01), with adults, whether familiars (mothers) or novel (the experimenter), highlighting their tendency toward social over non-social activities. Furthermore, WS children with full deletions were significantly more likely to maintain eye contact with the novel adult than did children with DS (p=.038). Compared to the typical WS group, Subject 5889 (tested at age 2.5y) spent greater amount of time far away in proximity from the novel adult (z = 7.96), and engaged in non-social activities for greater amount of time with both the parent (z = 2.39) and the novel adult (z = 6.00). The difference in the amount of eye contact toward the unfamiliar adult failed to reach significance (z = 1.24), due to a large SD within the WS group. These results provide converging evidence for the SISQ data presented above, showing that Subject 5889 did not display the hypersociability characteristic of individuals with WS full deletions.

FIG. 5 — Quantification of social behavior in Subject 5889 versus WS, Down syndrome (DS) and controls (typically developed, TD). Data replotted from Doyle et al., 2004 and Järvinen-Pasley et al., 2008. A: The atypical social behavior of Subject 5889 was reflected in parental ratings of social behavior using the SISQ at age 2.55 [Doyle et al., 2004, versus WS only] and were repeated at age 4.6 years [Järvinen-Pasley et al., 2008 versus a single typical WS case only]. In contrast with age-matched typical WS group, as well as children with DS and TD, the parental ratings (converted to z-scores for comparison) revealed that 5889's Global Sociability and Approach Strangers scores both at 2.55 years (z = -2.59 and -2.39, respectively) and at 4.86 years (z = -2.6 and -3.2), were significantly different from her peers in the typical WS group. B: A number of key differences in direct social behavior were noted between the Subject 5889 and children with typical WS deletions [shown in Järvinen-Pasley et al., 2008], relative to control children with DS for the mean total durations of three ethogram behaviors. Overall, WS children with full deletions were significantly less likely than children with DS to spend time far away in proximity from (p=.007), and to engage in non-social activities with (p=.01), with adults, whether familiars (mothers) or novel (the experimenter), highlighting their tendency toward social over non-social activities. Furthermore, WS children with full deletions were significantly more likely to maintain eye contact with the novel adult than did children with DS (p=.038). Compared to the typical WS group, Subject 5889 (tested at age 2.5y) spent greater amount of time far away in proximity from the novel adult (z = 7.96), and engaged in non-social activities for greater amount of time with both the parent (z = 2.39) and the novel adult (z = 6.00). The difference in the amount of eye contact toward the unfamiliar adult failed to reach significance (z = 1.24), due to a large SD within the WS group. These results provide converging evidence for the SISQ data presented above, showing that Subject 5889 did not display the hypersociability characteristic of individuals with WS full deletions.

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