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- W2994731240 abstract "Abstract Background Down syndrome (DS), caused by the triplication of chromosome 21, results in a constellation of clinical features including changes in intellectual and motor function. Although altered neural development and function have been well described in people with DS, few studies have investigated the etiology underlying the observed motor phenotypes. Here, we examine the development, patterning, and organization of the spinal cord throughout life in the Ts65Dn mouse, a model that recapitulates many of the motor changes observed in people with DS. Methods Spinal cords from embryonic to adult animals were processed for gene and protein expression (immunofluorescence) to track the spatiotemporal development of excitatory and inhibitory neurons and oligodendroglia. Postnatal analyses were focused on the lumbar region due to the reflex and gait abnormalities found in Ts65Dn mice and locomotive alterations seen in people with DS. Results Between embryonic days E10.5 and E14.5, we found a larger motor neuron progenitor domain in Ts65Dn animals containing more OLIG2-expressing progenitor cells. These disturbed progenitors are delayed in motor neuron production but eventually generate a large number of ISL1+ migrating motor neurons. We found that higher numbers of PAX6+ and NKX2.2+ interneurons (INs) are also produced during this time frame. In the adult lumbar spinal cord, we found an increased level of Hb9 and a decreased level of Irx3 gene expression in trisomic animals. This was accompanied by an increase in Calretinin+ INs, but no changes in other neuronal populations. In aged Ts65Dn animals, both Calbindin+ and ChAT+ neurons were decreased compared to euploid controls. Additionally, in the dorsal corticospinal white matter tract, there were significantly fewer CC1+ mature OLs in 30- and 60-day old trisomic animals and this normalized to euploid levels at 10–11 months. In contrast, the mature OL population was increased in the lateral funiculus, an ascending white matter tract carrying sensory information. In 30-day old animals, we also found a decrease in the number of nodes of Ranvier in both tracts. This decrease normalized both in 60-day old and aged animals. Conclusions We show marked changes in both spinal white matter and neuronal composition that change regionally over the life span. In the embryonic Ts65Dn spinal cord, we observe alterations in motor neuron production and migration. In the adult spinal cord, we observe changes in oligodendrocyte maturation and motor neuron loss, the latter of which has also been observed in human spinal cord tissue samples. This work uncovers multiple cellular perturbations during Ts65Dn development and aging, many of which may underlie the motor deficits found in DS." @default.
- W2994731240 created "2019-12-26" @default.
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- W2994731240 date "2019-12-01" @default.
- W2994731240 modified "2023-10-14" @default.
- W2994731240 title "Spatiotemporal development of spinal neuronal and glial populations in the Ts65Dn mouse model of Down syndrome" @default.
- W2994731240 cites W1261086304 @default.
- W2994731240 cites W1588980714 @default.
- W2994731240 cites W1807690494 @default.
- W2994731240 cites W1820550045 @default.
- W2994731240 cites W1834662352 @default.
- W2994731240 cites W1940356484 @default.
- W2994731240 cites W1975104207 @default.
- W2994731240 cites W1976527218 @default.
- W2994731240 cites W1982058282 @default.
- W2994731240 cites W1984191219 @default.
- W2994731240 cites W1987113148 @default.
- W2994731240 cites W1987510426 @default.
- W2994731240 cites W1989536487 @default.
- W2994731240 cites W1990701803 @default.
- W2994731240 cites W1993982470 @default.
- W2994731240 cites W1995488673 @default.
- W2994731240 cites W1997623074 @default.
- W2994731240 cites W2001784063 @default.
- W2994731240 cites W2005491819 @default.
- W2994731240 cites W2006121336 @default.
- W2994731240 cites W2009828482 @default.
- W2994731240 cites W2012078396 @default.
- W2994731240 cites W2016830113 @default.
- W2994731240 cites W2019373887 @default.
- W2994731240 cites W2021103771 @default.
- W2994731240 cites W2030669013 @default.
- W2994731240 cites W2032903160 @default.
- W2994731240 cites W2033558355 @default.
- W2994731240 cites W2034143722 @default.
- W2994731240 cites W2036979318 @default.
- W2994731240 cites W2038829496 @default.
- W2994731240 cites W2038934257 @default.
- W2994731240 cites W2040114944 @default.
- W2994731240 cites W2045461693 @default.
- W2994731240 cites W2065268546 @default.
- W2994731240 cites W2066729924 @default.
- W2994731240 cites W2076962859 @default.
- W2994731240 cites W2081314633 @default.
- W2994731240 cites W2082807347 @default.
- W2994731240 cites W2083551906 @default.
- W2994731240 cites W2084233122 @default.
- W2994731240 cites W2089405384 @default.
- W2994731240 cites W2091728674 @default.
- W2994731240 cites W2096225459 @default.
- W2994731240 cites W2104407051 @default.
- W2994731240 cites W2108000512 @default.
- W2994731240 cites W2113276791 @default.
- W2994731240 cites W2116219489 @default.
- W2994731240 cites W2118000416 @default.
- W2994731240 cites W2122209646 @default.
- W2994731240 cites W2126559811 @default.
- W2994731240 cites W2129371937 @default.
- W2994731240 cites W2135241886 @default.
- W2994731240 cites W2139031198 @default.
- W2994731240 cites W2139190679 @default.
- W2994731240 cites W2144118821 @default.
- W2994731240 cites W2144835441 @default.
- W2994731240 cites W2146517909 @default.
- W2994731240 cites W2152508103 @default.
- W2994731240 cites W2157340167 @default.
- W2994731240 cites W2159266478 @default.
- W2994731240 cites W2160708539 @default.
- W2994731240 cites W2162484161 @default.
- W2994731240 cites W2167526106 @default.
- W2994731240 cites W2168075066 @default.
- W2994731240 cites W2178297453 @default.
- W2994731240 cites W2262837973 @default.
- W2994731240 cites W2272609131 @default.
- W2994731240 cites W2287180374 @default.
- W2994731240 cites W2306947905 @default.
- W2994731240 cites W2396170582 @default.
- W2994731240 cites W2412523821 @default.
- W2994731240 cites W2518820902 @default.
- W2994731240 cites W2791662659 @default.
- W2994731240 cites W2801858511 @default.
- W2994731240 cites W2802259607 @default.
- W2994731240 doi "https://doi.org/10.1186/s11689-019-9294-9" @default.
- W2994731240 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/6913030" @default.
- W2994731240 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/31839007" @default.
- W2994731240 hasPublicationYear "2019" @default.
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