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. 2019 Jun 6;4(11):e124510. doi: 10.1172/jci.insight.124510

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© 2019 American Society for Clinical Investigation

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(A) Map of Ts65Dn and Tc1 trisomic regions. (B–E) Submucosal plexus of adult Euploid, Dscam^+/+; Ts65Dn, Dscam^+/+/⁺; Euploid, Dscam^+/–; and Ts65Dn, Dscam^+/+/–; and (F–I) Euploid, Dyrk1a^+/+; Ts65Dn, Dyrk1a ^+/+/+; Euploid, Dyrk1a ^+/–; and Ts65Dn, Dyrk1a ^+/+/– mice stained for HuC/D (red). (J and K) Quantification of neuron density from images, such as B–I, indicates normalizing copy number for Dscam and Dyrk1a did not prevent distal colon hypoganglionosis (P = 0.201 Euploid, Dscam^+/+ versus Ts65Dn, Dscam^+/+/– and P = 0.884 Ts65Dn, Dscam^+/+/⁺ versus Ts65Dn, Dscam^+/+/–, ANOVA with post-hoc Tukey test; n = 9 [Euploid, Dscam^+/+], n = 7 [Ts65Dn, Dscam^+/+/+], n = 6 [Euploid, Dscam^+/–], and n = 6 [Ts65Dn, Dscam^+/+/–]) or DYRK1A (P = 0.048 Euploid, Dyrk1a^+/+ versus Ts65Dn, Dyrk1a^+/+/–, and P = 0.999 Ts65Dn, Dyrk1a^+/+/– versus Ts65Dn, Dyrk1a^+/+/+, ANOVA with post-hoc Tukey test; n = 4 [Euploid, Dyrk1a^+/+], n = 3 [Ts65Dn, Dyrk1a^+/+/+], n = 3 [Euploid, Dyrk1a^+/–], and n = 3 [Ts65Dn, Dyrk1a^+/+/–]). Scale bar: 100 μm. *P < 0.05, **P < 0.01.