Lifespan analysis of brain development, gene expression and behavioral phenotypes in the Ts1Cje, Ts65Dn and Dp(16)1/Yey mouse models of Down syndrome
Open accessAbstract
Down syndrome (DS) results from triplication of human chromosome 21. Neuropathological hallmarks of DS include atypical central nervous system development that manifests prenatally and extends throughout life. As a result, individuals with DS exhibit cognitive and motor deficits and have delays in achieving developmental milestones. To determine whether different mouse models of DS recapitulate the human prenatal and postnatal phenotypes, here we directly compared brain histogenesis, gene expression, and behavior over the lifespan of three cytogenetically distinct mouse models of DS: Ts1Cje, Ts65Dn and Dp(16)1/Yey. Histological data indicated that Ts65Dn mice were the most consistently affected with respect to somatic growth, neurogenesis, and brain morphogenesis. Embryonic and adult gene expression results showed that Ts1Cje and Ts65Dn brains had considerably more differentially expressed (DEX) genes compared to Dp(16)1/Yey mice, despite the larger number of triplicated genes in the latter model. In addition, DEX genes showed little overlap in identity and chromosomal distribution in the three models, leading to dissimilarities in affected functional pathways. Perinatal and adult behavioral testing also highlighted differences among the models in their abilities to achieve various developmental milestones and perform hippocampal- and motor-based tasks. Interestingly, Dp(16)1/Yey mice showed no abnormalities in prenatal brain phenotypes, yet they manifested behavioral deficits starting at postnatal day 15 that continued through adulthood. In contrast, Ts1Cje mice showed mildly abnormal embryonic brain phenotypes, but only select behavioral deficits as neonates and adults. Altogether, our data showed widespread and unexpected fundamental differences in behavioral, gene expression, and brain development phenotypes between these three mouse models. Our findings illustrate unique limitations of each model when studying aspects of brain development and function in DS. This work helps to inform model selection in future studies investigating how observed neurodevelopmental abnormalities arise, how they contribute to cognitive impairment, and when testing therapeutic molecules to ameliorate the intellectual disability associated with DS.
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
Dp16 mice showed less freezing behavior compared to WT mice after the second shock on day 1 in contextual fear conditioning.
“As previously reported,38,40 Dp16 mice showed less freezing behavior compared to WT mice after the second shock on day 1 (Figure 2B).”
From Interferon signaling modulates Down syndrome-associated Alzheimer’s disease pathology in a mouse model (2025) · cites this paper as [40]
The Results (Adult behavior, Fig. 6C) report that Dp(16)1/Yey mice showed higher, not lower, freezing than euploid littermates 60 seconds after the second shock on day 1. The lower freezing reported for these mice occurred on day 2 (Fig. 6F). (LLM, conf=0.95)
Checked by a reasoning model — may be wrong.
Experimental — accuracy not yet measuredRecent studies indicate an increased ratio of inhibitory to excitatory neurons in the cortex and hippocampus in Dp16 mice that is associated with deficits in learning, memory, and motor function.
“recent studies that indicate an increased ratio of inhibitory to excitatory neurons in the cortex and hippocampus in Dp16 mice that is associated with deficits in learning, memory, and motor function.”
From Interferon signaling modulates Down syndrome-associated Alzheimer’s disease pathology in a mouse model (2025) · cites this paper as [40]
The Results section (Excitatory and inhibitory neuronal density; Fig. 4) reports decreased cortical PV+ and SS+ inhibitory neurons and no change in hippocampal inhibitory populations in Dp(16)1/Yey mice, not an increased inhibitory-to-excitatory ratio in both regions. The Discussion reports behavioral deficits in these mice, but does not establish the claimed association with an increased ratio. Page numbers were unavailable in our copy. (LLM, conf=0.89)
Checked by a reasoning model — may be wrong.
Experimental — accuracy not yet measuredDp16 mice exhibit hyperactive IFN signaling and recapitulate many phenotypes of DS including developmental delays, cranial malformations, and cognitive deficits.
“Notably, Dp16 mice exhibit hyperactive IFN signaling and recapitulate many phenotypes of DS including developmental delays, cranial malformations, and cognitive deficits.38,40,42,43,44,45”
From Interferon signaling modulates Down syndrome-associated Alzheimer’s disease pathology in a mouse model (2025) · cites this paper as [40]
The Results report upregulated interferon signaling in Dp(16)1/Yey brains, and the Discussion reports delays in late-acquired developmental milestones and adult learning and memory deficits. The source does not establish cranial malformations in these mice; its Summary of brain phenotypes by model instead reports no measurable embryonic brain-growth or forebrain-morphogenesis defects (page unavailable). (LLM, conf=0.88)
Experimental — accuracy not yet measuredThe Ts65Dn DS mouse model demonstrates embryonic and early postnatal gene expression, neuroanatomical, and developmental behavioral deficits like those observed in DS.
“The Ts65Dn DS mouse model demonstrates embryonic and early postnatal gene expression, neuroanatomical, and developmental behavioral deficits like those observed in DS, whereas other DS models, including Dp1Yey and TcMAC21, do not have or have not yet been tested for these phenotypes (Aziz et al., 2018; Goodliffe et al., 2016).”
From Sex-specific developmental alterations in DYRK1A expression in the brain of a Down syndrome mouse model (2024) · cites this paper as Aziz et al., 2018
The Results report altered embryonic gene expression in Ts65Dn mice, while the Discussion describes embryonic brain abnormalities and perinatal developmental-milestone delays resembling aspects of Down syndrome. The paper reports gene-expression measurements in embryonic and adult brains, not early postnatal brains, so the early postnatal gene-expression part is unsupported. (LLM, conf=0.84)
Experimental — accuracy not yet measuredDp16 mice exhibit impaired motor coordination at 3 and 5 months of age relative to WT littermates, with a trend toward poorer performance at 7 and 9 months of age.
“We next tested motor coordination using the accelerating rotarod test DS-AD mice (Data S4), and consistent with previous reports,38,40 we found that Dp16 mice exhibit impaired motor coordination at 3 and 5 months of age relative to WT littermates, with a trend toward poorer performance at 7 and 9 months of age (Figure 2A).”
From Interferon signaling modulates Down syndrome-associated Alzheimer’s disease pathology in a mouse model (2025) · cites this paper as [40]
The partial rescue of 5xFAD behavioral phenotypes in 5xFAD-Dp16 mice could be explained in part by Dp16 mice having impairment in motor coordination and associative learning and memory, ameliorating the cognitive profile observed in AD mice.
“this could be explained in part by the fact that Dp16 mice present an impairment in motor coordination, and associative learning and memory ameliorating the cognitive profile observed in AD mice.”
From Interferon signaling modulates Down syndrome-associated Alzheimer’s disease pathology in a mouse model (2025) · cites this paper as [40]
In Ts65Dn, the number of actively proliferating cells in the germinal zone of the dorsal telencephalon is decreased and this deficit persists postnatally, with fewer proliferating cells being found in the hippocampus.
“In concert, the number of actively proliferating cells in the germinal zone of the dorsal telencephalon is decreased (Aziz et al., 2018; Shaw et al., 2020) and this deficit persists postnatally, with fewer proliferating cells being found in the hippocampus (Contestabile et al., 2007).”
From Neurodevelopment in Down syndrome: Concordance in humans and models (2022) · cites this paper as Aziz et al., 2018
Contrary to the existing human data, these proliferation changes in Ts65Dn appear to be region specific as an increase in number of actively proliferating cells was observed in the embryonic medial ganglionic eminence, the site of interneuron production.
“Contrary to the existing human data, these proliferation changes appear to be region specific as an increase in number of actively proliferating cells was observed in the embryonic medial ganglionic eminence, the site of interneuron production (Chakrabarti et al., 2010; Aziz et al., 2018).”
From Neurodevelopment in Down syndrome: Concordance in humans and models (2022) · cites this paper as Aziz et al., 2018
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
The self-check needs the paper’s full text.
Cites
Controlling the False Discovery Rate: A Practical and Powerful Approach to Multiple Testing
Benjamini, Y., & Hochberg, Y. (1995) · Journal of the Royal Statistical Society Series B: Statistical Methodology · doi:10.1111/j.2517-6161.1995.tb02031.x · Cited by 109,482 · see citing works
Systematic and integrative analysis of large gene lists using DAVID bioinformatics resources
Huang, D. W., Sherman, B. T., & Lempicki, R. A. (2009) · Nature Protocols · doi:10.1038/nprot.2008.211 · Cited by 35,890 · see citing works
MicroRNAs: Target Recognition and Regulatory Functions
Bartel, D. P. (2009) · Cell · doi:10.1016/j.cell.2009.01.002 · Cited by 20,068 · see citing works
Morris water maze: procedures for assessing spatial and related forms of learning and memory
Vorhees, C. V., & Williams, M. T. (2006) · Nature Protocols · doi:10.1038/nprot.2006.116 · Cited by 4,629 · see citing works
Bushati, N., & Cohen, S. M. (2007) · Annual Review of Cell and Developmental Biology · doi:10.1146/annurev.cellbio.23.090506.123406 · Cited by 2,563 · see citing works
A mouse model for Down syndrome exhibits learning and behaviour deficits
Reeves, R. H., Irving, N. G., Moran, T. H., Wohn, A., Kitt, C., Sisodia, S. S., Schmidt, C., Bronson, R. T., & Davisson, M. T. (1995) · Nature Genetics · doi:10.1038/ng1095-177 · Cited by 913 · see citing works
Lu, Q. R., Yuk, D.-I., Alberta, J., Zhu, Z.-M., Pawlitzky, I., Chan, J., McMahon, A., Stiles, C., & Rowitch, D. H. (2000) · Neuron · doi:10.1016/s0896-6273(00)80897-1 · Cited by 883 · see citing works
Rogers, D. C., Fisher, E. M. C., Brown, S. D. M., Peters, J., Hunter, A. J., & Martin, J. E. (1997) · Mammalian Genome · doi:10.1007/s003359900551 · Cited by 795 · see citing works
Reflex-ontogeny and behavioural development of the mouse
Fox, W. M. (1965) · Animal Behaviour · doi:10.1016/0003-3472(65)90041-2 · Cited by 699 · see citing works
Aneuploidy Affects Proliferation and Spontaneous Immortalization in Mammalian Cells
Williams, B. R., Prabhu, V. R., Hunter, K. E., Glazier, C. M., Whittaker, C. A., Housman, D. E., & Amon, A. (2008) · Science · doi:10.1126/science.1160058 · Cited by 665 · see citing works
Assessing Spatial Learning and Memory in Rodents
Vorhees, C. V., & Williams, M. T. (2014) · ILAR Journal · doi:10.1093/ilar/ilu013 · Cited by 543 · see citing works
The neuropsychology of down syndrome: evidence for hippocampal dysfunction.
Pennington, B. F., Moon, J., Edgin, J., Stedron, J., & Nadel, L. (2003) · Child Development · doi:10.1111/1467-8624.00522 · Cited by 514 · see citing works
A comparative phenotypic and genomic analysis of C57BL/6J and C57BL/6N mouse strains
Simon, M. M., Greenaway, S., White, J. K., Fuchs, H., Gailus-Durner, V., Wells, S., Sorg, T., Wong, K., Bedu, E., Cartwright, E. J., et al. (2013) · Genome Biology · doi:10.1186/gb-2013-14-7-r82 · Cited by 507 · see citing works
Neuroanatomy of Down’s Syndrome: A High-Resolution MRI Study
Pinter, J. D., Eliez, S., Schmitt, J. E., Capone, G. T., & Reiss, A. L. (2001) · American Journal of Psychiatry · doi:10.1176/appi.ajp.158.10.1659 · Cited by 452 · see citing works
Developmental abnormalities and age-related neurodegeneration in a mouse model of Down syndrome
Holtzman, D. M., Santucci, D., Kilbridge, J., Chua-Couzens, J., Fontana, D. J., Daniels, S. E., Johnson, R. M., Chen, K., Sun, Y., Carlson, E., et al. (1996) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.93.23.13333 · Cited by 448 · see citing works
Takebayashi, H., Yoshida, S., Sugimori, M., Kosako, H., Kominami, R., Nakafuku, M., & Nabeshima, Y. (2000) · Mechanisms of Development · doi:10.1016/s0925-4773(00)00466-4 · Cited by 409 · see citing works
Miyoshi, G., Butt, S. J. B., Takebayashi, H., & Fishell, G. (2007) · The Journal of Neuroscience · doi:10.1523/jneurosci.1807-07.2007 · Cited by 400 · see citing works
Sago, H., Carlson, E. J., Smith, D. J., Kilbridge, J., Rubin, E. M., Mobley, W. C., Epstein, C. J., & Huang, T.-T. (1998) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.95.11.6256 · Cited by 382 · see citing works
(1990) · PubMed · Cited by 351 · see citing works
Takashima, S., Becker, L. E., Armstrong, D. L., & Chan, F. (1981) · Brain Research · doi:10.1016/0006-8993(81)90314-0 · Cited by 307 · see citing works
Wisniewski, K. E. (2005) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320370755 · Cited by 297 · see citing works
Cerebral Morphologic Distinctions Between Williams and Down Syndromes
Jernigan, T. L., Bellugi, U., Sowell, E. R., Doherty, S., & Hesselink, J. R. (1993) · Archives of Neurology · doi:10.1001/archneur.1993.00540020062019 · Cited by 293 · see citing works
Down's syndrome: a genetic disorder in biobehavioral perspective
Nadel, L. (2003) · Genes Brain & Behavior · doi:10.1034/j.1601-183x.2003.00026.x · Cited by 291 · see citing works
de Graaf, G., Buckley, F., & Skotko, B. G. (2015) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.37001 · Cited by 291 · see citing works
Selective neuroanatornic abnormalities in Down's syndrome and their cognitive correlates
Raz, N., Torres, I. J., Briggs, S. D., Spencer, W. D., Thornton, A. E., Loken, W. J., Gunning, F. M., McQuain, J., Driesen, N. R., & Acker, J. D. (1995) · Neurology · doi:10.1212/wnl.45.2.356 · Cited by 287 · see citing works
Contestabile, A., Fila, T., Ceccarelli, C., Bonasoni, P., Bonapace, L., Santini, D., Bartesaghi, R., & Ciani, E. (2007) · Hippocampus · doi:10.1002/hipo.20308 · Cited by 281 · see citing works
Dendritic atrophy in children with down’s syndrome.
Becker, L. E., Armstrong, D. L., & Chan, F. (1986) · Annals of Neurology · doi:10.1002/ana.410200413 · Cited by 279 · see citing works
Guidi, S., Bonasoni, P., Ceccarelli, C., Santini, D., Gualtieri, F., Ciani, E., & Bartesaghi, R. (2008) · Brain Pathology · doi:10.1111/j.1750-3639.2007.00113.x · Cited by 276 · see citing works
Olig1 and Olig2 triplication causes developmental brain defects in down syndrome.
Chakrabarti, L., Best, T. K., Cramer, N. P., Carney, R. S. E., Isaac, J. T. R., Galdzicki, Z., & Haydar, T. F. (2010) · Nature Neuroscience · doi:10.1038/nn.2600 · Cited by 258 · see citing works
Self-citationKahlem, P., Sultan, M., Herwig, R., Steinfath, M., Balzereit, D., Eppens, B., Saran, N. G., Pletcher, M. T., South, S. T., Stetten, G., et al. (2004) · Genome Research · doi:10.1101/gr.1951304 · Cited by 249 · see citing works
Olfactory regulation of maternal behavior in mammals
Lévy, F., Keller, M., & Poindron, P. (2004) · Hormones and Behavior · doi:10.1016/j.yhbeh.2004.02.005 · Cited by 248 · see citing works
The neurobiologie consequences of down syndrome
Coyle, J. T., Oster-Granite, M. L., & Gearhart, J. D. (1986) · Brain Research Bulletin · doi:10.1016/0361-9230(86)90074-2 · Cited by 248 · see citing works
Housing, husbandry and handling of rodents for behavioral experiments
Deacon, R. M. J. (2006) · Nature Protocols · doi:10.1038/nprot.2006.120 · Cited by 246 · see citing works
Olmos-Serrano, J. L., Kang, H. J., Tyler, W. A., Silbereis, J. C., Cheng, F., Zhu, Y., Pletikos, M., Jankovic-Rapan, L., Cramer, N. P., Galdzicki, Z., et al. (2016) · Neuron · doi:10.1016/j.neuron.2016.01.042 · Cited by 239 · see citing works
Self-citationUnderstanding the Basis for Down Syndrome Phenotypes
Roper, R. J., & Reeves, R. H. (2006) · PLoS Genetics · doi:10.1371/journal.pgen.0020050 · Cited by 233 · see citing works
Trisomy 21 causes changes in the circulating proteome indicative of chronic autoinflammation
Sullivan, K. D., Evans, D., Pandey, A., Hraha, T. H., Smith, K. P., Markham, N., Rachubinski, A. L., Wolter-Warmerdam, K., Hickey, F., Espinosa, J. M., et al. (2017) · Scientific Reports · doi:10.1038/s41598-017-13858-3 · Cited by 228 · cited 2× · see citing works
“This finding, which is confirmed by both GSEA and DAVID analyses, correlates well with recent work assessing proteomic changes in blood samples from people with DS (Sullivan et al., 2017) and another study analyzing interferon-related gene networks in postnatal Ts1Cje brains (Ling et al., 2014).”
ResultNeurological phenotypes for Down syndrome across the life span
Lott, I. T. (2012) · Progress in Brain Research · doi:10.1016/b978-0-444-54299-1.00006-6 · Cited by 225 · see citing works
Duchon, A., Raveau, M., Chevalier, C., Nalesso, V., Sharp, A. J., & Herault, Y. (2011) · Mammalian Genome · doi:10.1007/s00335-011-9356-0 · Cited by 224 · see citing works
Li, Z., Yu, T., Morishima, M., Pao, A., LaDuca, J., Conroy, J., Nowak, N., Matsui, S.-I., Shiraishi, I., & Yu, Y. E. (2007) · Human Molecular Genetics · doi:10.1093/hmg/ddm086 · Cited by 218 · see citing works
Behavioral Differences among C57BL/6 Substrains: Implications for Transgenic and Knockout Studies
Bryant, C. D., Zhang, N. N., Sokoloff, G., Fanselow, M. S., Ennes, H. S., Palmer, A. A., & McRoberts, J. A. (2008) · Journal of Neurogenetics · doi:10.1080/01677060802357388 · Cited by 210 · see citing works
Development of the superior temporal neocortex is anomalous in trisomy 21
Golden, J. A., & Hyman, B. T. (1994) · Journal of Neuropathology & Experimental Neurology · doi:10.1097/00005072-199409000-00011 · Cited by 210 · see citing works
(1991) · PubMed · Cited by 209 · see citing works
Gene Expression From the Aneuploid Chromosome in a Trisomy Mouse Model of Down SyndromePreprint
Lyle, R., Gehrig, C., Neergaard-Henrichsen, C., Deutsch, S., & Antonarakis, S. E. (2004) · Genome Research · doi:10.1101/gr.2090904 · Cited by 202 · see citing works
Belichenko, P. V., Kleschevnikov, A. M., Salehi, A., Epstein, C. J., & Mobley, W. C. (2007) · The Journal of Comparative Neurology · doi:10.1002/cne.21433 · Cited by 202 · see citing works
(1990) · PubMed · Cited by 201 · see citing works
Trisomy 21 and early brain development
Haydar, T. F., & Reeves, R. H. (2012) · Trends in Neurosciences · doi:10.1016/j.tins.2011.11.001 · Cited by 200 · see citing works
Self-citationChakrabarti, L., Galdzicki, Z., & Haydar, T. F. (2007) · The Journal of Neuroscience · doi:10.1523/jneurosci.3406-07.2007 · Cited by 199 · see citing works
Self-citationEducation and children with Down syndrome: Neuroscience, development, and intervention
Fidler, D. J., & Nadel, L. (2007) · Mental Retardation and Developmental Disabilities Research Reviews · doi:10.1002/mrdd.20166 · Cited by 198 · see citing works
Amygdala and hippocampal volumes in children with Down syndrome: A high-resolution MRI study
Pinter, J. D., Brown, W. E., D, S. E. M., Schmitt, J. E., Capone, G. T., & Reiss, A. L. (2001) · Neurology · doi:10.1212/wnl.56.7.972 · Cited by 197 · see citing works
Rodent models in Down syndrome research: impact and future opportunities
Hérault, Y., Delabar, J. M., Fisher, E., Tybulewicz, V. L. J., Yu, E. Y., & Brault, V. (2017) · Disease Models & Mechanisms · doi:10.1242/dmm.029728 · Cited by 191 · see citing works
Down's syndrome: Is there a decreased population of neurons?
Ross, M. H., Galaburda, A. M., & Kemper, T. L. (1984) · Neurology · doi:10.1212/wnl.34.7.909 · Cited by 191 · see citing works
Molecular characterization of the translocation breakpoints in the down syndrome mouse model Ts65Dn
Reinholdt, L. G., Ding, Y., Gilbert, G. T., Czechanski, A., Solzak, J. P., Roper, R. J., Johnson, M. T., Donahue, L. R., Lutz, C., & Davisson, M. T. (2011) · Mammalian Genome · doi:10.1007/s00335-011-9357-z · Cited by 186 · see citing works
Mouse models of Down syndrome: gene content and consequences
Gupta, M., Dhanasekaran, A. R., & Gardiner, K. (2016) · Mammalian Genome · doi:10.1007/s00335-016-9661-8 · Cited by 167 · see citing works
Primary and secondary transcriptional effects in the developing human down syndrome brain and heart
Mao, R., Wang, X., Spitznagel, E. L., Frelin, L. P., Ting, J. C., Ding, H., Kim, J.-W., Ruczinski, I., Downey, T. J., & Pevsner, J. (2005) · Genome Biology · doi:10.1186/gb-2005-6-13-r107 · Cited by 166 · see citing works
Olson, L. E., Roper, R. J., Baxter, L. L., Carlson, E. J., Epstein, C. J., & Reeves, R. H. (2004) · Developmental Dynamics · doi:10.1002/dvdy.20079 · Cited by 165 · see citing works
Mental health, behaviour and intellectual abilities of people with Down syndrome
Määttä, T., Tervo-Määttä, T., Taanila, A. M., Kaski, M., & Iivanainen, M. V. (2006) · Down Syndrome Research and Practice · doi:10.3104/reports.313 · Cited by 154 · see citing works
Teipel, S. J., Alexander, G. E., Schapiro, M. B., Möller, H., Rapoport, S. I., & Hampel, H. J. (2004) · Brain · doi:10.1093/brain/awh101 · Cited by 154 · see citing works
A voxel-based morphometric study of nondemented adults with Down Syndrome
White, N. S., Alkire, M. T., & Haier, R. J. (2003) · NeuroImage · doi:10.1016/s1053-8119(03)00273-8 · Cited by 146 · see citing works
Global disruption of the cerebellar transcriptome in a Down syndrome mouse model
Saran, N. G. (2003) · Human Molecular Genetics · doi:10.1093/hmg/ddg217 · Cited by 143 · see citing works
Vilardell, M., Rasche, A., Thormann, A., Maschke-Dutz, E., Jurado, L. A. P., Lehrach, H. R., & Herwig, R. (2011) · BMC Genomics · doi:10.1186/1471-2164-12-229 · Cited by 127 · see citing works
Whittle, N., Sartori, S. B., Dierssen, M., Lubec, G., & Singewald, N. (2007) · Pediatrics · doi:10.1542/peds.2006-3448 · Cited by 127 · see citing works
Inbred mouse strains and genetic stability: a review
Casellas, J. (2010) · animal · doi:10.1017/s1751731110001667 · Cited by 124 · see citing works
Weitzdoerfer, R., Dierssen, M., Fountoulakis, M., & Lübec, G. (2001) · Springer eBooks · doi:10.1007/978-3-7091-6262-0_5 · Cited by 122 · see citing works
Pritchard, M. A., & Kola, I. (1999) · Springer eBooks · doi:10.1007/978-3-7091-6380-1_20 · Cited by 116 · see citing works
Relation of Corpus Callosum and Hippocampal Size to Age in Nondemented Adults With Down’s Syndrome
Teipel, S. J., Schapiro, M. B., Alexander, G. E., Krasuski, J., Horwitz, B., Hoehne, C., Möller, H., Rapoport, S. I., & Hampel, H. J. (2003) · American Journal of Psychiatry · doi:10.1176/appi.ajp.160.10.1870 · Cited by 113 · see citing works
Ishihara, K., Amano, K., Takaki, E., Shimohata, A., Sago, H., Epstein, C. J., & Yamakawa, K. (2009) · Cerebral Cortex · doi:10.1093/cercor/bhp176 · Cited by 106 · see citing works
Hyperkalemia during Anesthesia
Wisniewski, K. E., Laure‐Kamionowska, M., & Wisniewski, H. M. (1984) · New England Journal of Medicine · doi:10.1056/nejm198411013111818 · Cited by 105 · see citing works
Rachidi, M., & Lopes, C. (2008) · European Journal of Paediatric Neurology · doi:10.1016/j.ejpn.2007.08.010 · Cited by 101 · see citing works
The use of mouse models to understand and improve cognitive deficits in Down syndrome
Das, I., & Reeves, R. H. (2011) · Disease Models & Mechanisms · doi:10.1242/dmm.007716 · Cited by 99 · see citing works
The development of learning difliculties in children with Down's syndrome
Wishart, J. G. (1993) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.1993.tb00882.x · Cited by 96 · see citing works
Villar, A. J., Belichenko, P. V., Gillespie, A. M., Kozy, H. M., Mobley, W. C., & Epstein, C. J. (2005) · Mammalian Genome · doi:10.1007/s00335-004-2428-7 · Cited by 96 · see citing works
(1989) · PubMed · Cited by 96 · see citing works
5-Ethynyl-2′-deoxycytidine as a new agent for DNA labeling: Detection of proliferating cells
Qu, D., Wang, G., Wang, Z., Zhou, L., Chi, W., Cong, S., Ren, X., Liang, P., & Zhang, B. (2011) · Analytical Biochemistry · doi:10.1016/j.ab.2011.05.037 · Cited by 93 · see citing works
Autosomal aneuploidy in mice: Generation and developmental consequences
Gearhart, J. D., Davisson, M. T., & Oster‐Granite, M. L. (1986) · Brain Research Bulletin · doi:10.1016/0361-9230(86)90075-4 · Cited by 91 · see citing works
Reduced cell number in the neocortical part of the human fetal brain in Down syndrome
Larsen, K. B., Laursen, H., Græm, N., Samuelsen, G. B., Bogdanovic, N., & Pakkenberg, B. (2008) · Annals of Anatomy - Anatomischer Anzeiger · doi:10.1016/j.aanat.2008.05.007 · Cited by 87 · cited 4× · see citing works
“…to identify suitable models for basic and translational studies (Bahado-Singh et al., 1992; Cardoso et al., 2015; Guidi et al., 2008; Haim et al., 2009; Larsen et al., 2008; Lott, 2012; Nilholm, 1999; Vicari et al., 2013; Winter et al., 1998; Wisniewski and Kida, 1994; Wisniewski et al., 1984).”
BackgroundInfluentialDauphinot, L., Lyle, R., Rivals, I., Dang, M., Moldrich, R. X., Golfier, G., Ettwiller, L. M., Toyama, K., Rossier, J., Personnaz, L., et al. (2004) · Human Molecular Genetics · doi:10.1093/hmg/ddi033 · Cited by 86 · see citing works
Biometry of Face and Brain in Fetuses with Trisomy 21
Guihard-Costa, A.-M., Khung‐Savatovsky, S., Delbecque, K., Ménez, F., & Delezoide, A. (2005) · Pediatric Research · doi:10.1203/01.pdr.0000190580.88391.9a · Cited by 85 · see citing works
Petit, T. L., LeBoutillier, J. C., Alfano, D. P., & Becker, L. E. (1984) · Experimental Neurology · doi:10.1016/0014-4886(84)90041-4 · Cited by 84 · see citing works
Role of Founder Cell Deficit and Delayed Neuronogenesis in Microencephaly of the Trisomy 16 Mouse
Haydar, T. F., Nowakowski, R. S., Yarowsky, P., & Krueger, B. K. (2000) · Journal of Neuroscience · doi:10.1523/jneurosci.20-11-04156.2000 · Cited by 83 · see citing works
Self-citationElton, T. S., Sansom, S. E., & Martin, M. M. (2010) · RNA Biology · doi:10.4161/rna.7.5.12685 · Cited by 83 · see citing works
Evidence of Arrest of Neurogenesis and Synaptogenesis in Brains of Patients with Down's Syndrome
(1984) · New England Journal of Medicine · doi:10.1056/nejm198411013111819 · Cited by 82 · see citing works
Costa, A. C. S., Stasko, M. R., Schmidt, C., & Davisson, M. T. (2009) · Behavioural Brain Research · doi:10.1016/j.bbr.2009.08.034 · Cited by 80 · see citing works
Stasko, M. R., & Costa, A. C. S. (2004) · Behavioural Brain Research · doi:10.1016/j.bbr.2004.01.012 · Cited by 79 · see citing works
Gene expression variation in Down's syndrome mice allows prioritization of candidate genes
Sultan, M., Piccini, I., Balzereit, D., Herwig, R., Saran, N. G., Lehrach, H., Reeves, R. H., & Yaspo, M.-L. (2007) · Genome Biology · doi:10.1186/gb-2007-8-5-r91 · Cited by 78 · see citing works
ARID1B-mediated disorders: Mutations and possible mechanisms
Sim, J. C. H., White, S. M., & Lockhart, P. J. (2014) · Intractable & Rare Diseases Research · doi:10.5582/irdr.2014.01021 · Cited by 77 · see citing works
Verbal short‐term memory in Down's syndrome: An articulatory loop deficit?
Vicari, S., Marotta, L., & Carlesimo, G. A. (2004) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.2004.00478.x · Cited by 76 · see citing works
Neurodevelopmental and psychiatric issues in Down’s syndrome
Vicari, S., Pontillo, M., & Armando, M. (2013) · Psychiatric Genetics · doi:10.1097/ypg.0b013e32835fe426 · Cited by 71 · see citing works
Ling, K., Hewitt, C., Tan, K.-L., Cheah, P.-S., Vidyadaran, S., Lai, M. I., Lee, H.-C., Simpson, K., Hyde, L., Pritchard, M. A., et al. (2014) · BMC Genomics · doi:10.1186/1471-2164-15-624 · Cited by 66 · see citing works
Mouse Models of Cognitive Disorders in Trisomy 21: A Review
Sérégaza, Z., Roubertoux, P. L., Jamon, M., & Soumireu-Mourat, B. (2006) · Behavior Genetics · doi:10.1007/s10519-006-9056-9 · Cited by 66 · see citing works
Consequences of Trisomy 16 for Mouse Brain Development: Corticogenesis in a Model of Down Syndrome
Haydar, T. F., Blue, M. E., Molliver, M. E., Krueger, B. K., & Yarowsky, P. (1996) · Journal of Neuroscience · doi:10.1523/jneurosci.16-19-06175.1996 · Cited by 66 · see citing works
Self-citationAbsence of Prenatal Forebrain Defects in the Dp(16)1Yey/+ Mouse Model of down Syndrome.
Goodliffe, J. W., Olmos-Serrano, J. L., Aziz, N. M., Pennings, J. L. A., Guedj, F., Bianchi, D. W., & Haydar, T. F. (2016) · The Journal of Neuroscience · doi:10.1523/jneurosci.2513-15.2016 · Cited by 65 · see citing works
Self-citationGrowing up with Down syndrome: Development from 6 months to 10.7 years
Marchal, J. P., Maurice‐Stam, H., Houtzager, B. A., van Rozenburg-Marres, S. L. R., Oostrom, K. J., Grootenhuis, M. A., & van Trotsenburg, A. S. P. (2016) · Research in Developmental Disabilities · doi:10.1016/j.ridd.2016.09.019 · Cited by 65 · see citing works
Mammary Odor Cues and Pheromones
Schaal, B. (2010) · Vitamins and hormones · doi:10.1016/s0083-6729(10)83004-3 · Cited by 64 · see citing works
Contestabile, A., Fila, T., Bartesaghi, R., & Ciani, E. (2008) · Brain Pathology · doi:10.1111/j.1750-3639.2008.00168.x · Cited by 63 · see citing works
Survival of children with mosaic Down syndrome
Shin, M., Siffel, C., & Correa, A. (2010) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.33295 · Cited by 60 · see citing works
Guedj, F., Pennings, J. L. A., Massingham, L. J., Wick, H. C., Siegel, A. E., Tantravahi, U., & Bianchi, D. W. (2016) · Scientific Reports · doi:10.1038/srep32353 · Cited by 59 · see citing works
Self-citationLaffaire, J., Rivals, I., Dauphinot, L., Pasteau, F., Wehrlé, R., Larrat, B., Vitalis, T., Moldrich, R. X., Rossier, J., Sinkus, R., et al. (2009) · BMC Genomics · doi:10.1186/1471-2164-10-138 · Cited by 56 · see citing works
Memory development and intellectual disabilities
Vicari, S. (2004) · Acta Paediatrica · doi:10.1111/j.1651-2227.2004.tb03059.x · Cited by 56 · see citing works
Potier, M., Rivals, I., MERCIER, G., Ettwiller, L. M., Moldrich, R. X., Laffaire, J., Personnaz, L., Rossier, J., & Dauphinot, L. (2006) · Journal of Neurochemistry · doi:10.1111/j.1471-4159.2005.03624.x · Cited by 55 · see citing works
Kittler, P. M., Krinsky‐McHale, S. J., & Devenny, D. A. (2004) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.2004.00500.x · Cited by 54 · see citing works
Motor Performance of Children With Down Syndrome and Typical Development at 2 to 4 and 26 Months
Cardoso, A. C. D. N., de Campos, A. C., dos Santos, M. M., Santos, D. C. C., & Rocha, N. A. C. F. (2015) · Pediatric Physical Therapy · doi:10.1097/pep.0000000000000120 · Cited by 50 · see citing works
Starbuck, J. M., Dutka, T., Ratliff, T. S., Reeves, R. H., & Richtsmeier, J. T. (2014) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.36594 · Cited by 50 · see citing works
The power of comparative and developmental studies for mouse models of Down syndrome
Moore, C. S., & Roper, R. J. (2007) · Mammalian Genome · doi:10.1007/s00335-007-9030-8 · Cited by 48 · see citing works
Bahado-Singh, R. O., Wyse, L., Dorr, M. A., Copel, J. A., O'Connor, T., & Hobbins, J. C. (1992) · American Journal of Obstetrics and Gynecology · doi:10.1016/s0002-9378(12)80029-9 · Cited by 46 · see citing works
Fetal transcerebellar diameter in down syndrome
Rotmensch, S., Goldstein, I. H., Liberati, M., Shalev, J., Ben‐Rafael, Z., & Copel, J. A. (1997) · Obstetrics and Gynecology · doi:10.1016/s0029-7844(97)00076-8 · Cited by 46 · see citing works
Olmos-Serrano, J. L., Tyler, W. A., Cabral, H., & Haydar, T. F. (2016) · Experimental Neurology · doi:10.1016/j.expneurol.2016.02.005 · Cited by 42 · see citing works
Self-citationZhang, L., Meng, K., Jiang, X.-L., Liu, C., Pao, A., Belichenko, P., Kleschevnikov, A., Josselyn, S., Liang, P., Ye, P., et al. (2014) · Human Molecular Genetics · doi:10.1093/hmg/ddt446 · Cited by 40 · see citing works
Widespread impairment of cell proliferation in the neonate Ts65Dn mouse, a model for Down syndrome
Contestabile, A., Fila, T., Cappellini, A., Bartesaghi, R., & Ciani, E. (2009) · Cell Proliferation · doi:10.1111/j.1365-2184.2009.00587.x · Cited by 38 · see citing works
Analysis of microRNA expression profile by small RNA sequencing in Down syndrome fetuses
Xu, Y. J., Li, W., Liu, X., Ma, H., Tu, Z., & Dai, Y. (2013) · International Journal of Molecular Medicine · doi:10.3892/ijmm.2013.1499 · Cited by 33 · see citing works
Guedj, F., Pennings, J. L. A., Ferres, M., Graham, L. C., Wick, H. C., Miczek, K. A., Slonim, D. K., & Bianchi, D. W. (2015) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.37156 · Cited by 32 · see citing works
Self-citationMouse-based genetic modeling and analysis of Down syndrome
Xing, Z., Li, Y., Pao, A., Bennett, A. S., Tycko, B., Mobley, W. C., & Yu, Y. E. (2016) · British Medical Bulletin · doi:10.1093/bmb/ldw040 · Cited by 30 · see citing works
Age and pattern of intellectual decline among Down Syndrome and other mentally retarded adults
Gibson, D., Groeneweg, G., Jerry, P., & Harris, A. (1988) · International Journal of Rehabilitation Research · doi:10.1097/00004356-198803000-00006 · Cited by 30 · see citing works
Olfaction in People with Down Syndrome: A Comprehensive Assessment across Four Decades of Age
Cecchini, M. P., Viviani, D., Sandri, M. F., Haehner, A., Hummel, T. R., & Zancanaro, C. (2016) · PLoS ONE · doi:10.1371/journal.pone.0146486 · Cited by 30 · see citing works
Fetal therapy for Down syndrome: an ethical exploration
de Wert, G. M. W. R., Dondorp, W., & Bianchi, D. W. (2016) · Prenatal Diagnosis · doi:10.1002/pd.4995 · Cited by 29 · see citing works
Self-citationCytogenetic Evaluation of Down Syndrome: A Review of 1020 Referral Cases
Chandra, N., Cyrus, C., Lakshminarayana, P., Nallasivam, P., Ramesh, A., Gopinath, P. M., & Marimuthu, K. M. (2010) · International Journal of Human Genetics · doi:10.1080/09723757.2010.11886090 · Cited by 29 · see citing works
Contingency learning in 9-month-old infants with Down syndrome.
Ohr, P. S., & Fagen, J. W. (1994) · PubMed · Cited by 28 · see citing works
Johnson, K. L., Tao, K. X., Stroh, H., Kallenbach, L. R., Peter, I., Richey, L. J., Rust, D. W., & Bianchi, D. W. (2009) · Fertility and Sterility · doi:10.1016/j.fertnstert.2009.08.042 · Cited by 24 · see citing works
Self-citationGuedj, F., Pennings, J. L. A., Wick, H. C., & Bianchi, D. W. (2014) · Brain Pathology · doi:10.1111/bpa.12151 · Cited by 23 · see citing works
Self-citationDevelopmental milestones in toddlers with atypical development
Horovitz, M., & Matson, J. L. (2011) · Research in Developmental Disabilities · doi:10.1016/j.ridd.2011.07.039 · Cited by 19 · see citing works
Liu, W., Han, F., JIANG, X. H., Chen, H., Zhao, H., Liu, Y., Li, Y., Huang, C., Cao, J., & Liu, J.-Y. (2015) · Oncotarget · doi:10.18632/oncotarget.4237 · Cited by 19 · see citing works
Frontal lobe shortening in second-trimester fetuses with trisomy 21: usefulness as a US marker.
Winter, T. C., Reichman, J. A., Luna, J. A., Cheng, E. Y., Doll, A. M., Komarniski, C. A., Nghiem, H. V., Schmiedl, U. P., Shields, L. E., & Uhrich, S. B. (1998) · Radiology · doi:10.1148/radiology.207.1.9530318 · Cited by 19 · see citing works
Ishihara, K., Kanai, S., Sago, H., Yamakawa, K., & Akiba, S. (2014) · Neuroscience · doi:10.1016/j.neuroscience.2014.09.039 · Cited by 18 · see citing works
Implicit and explicit olfactory memory in people with and without Down syndrome
Johns, A., Homewood, J., Stevenson, R. J., & Taylor, A. (2011) · Research in Developmental Disabilities · doi:10.1016/j.ridd.2011.11.001 · Cited by 13 · see citing works
The zone of proximal development: A comparison of children with Down syndrome and typical children
Nilholm, C. (1999) · Journal of Intellectual & Developmental Disability · doi:10.1080/13668259900034031 · Cited by 11 · see citing works
Ferres, M., Bianchi, D. W., Siegel, A. E., Bronson, R. T., Huggins, G. S., & Guedj, F. (2016) · PLoS ONE · doi:10.1371/journal.pone.0168009 · Cited by 11 · see citing works
Self-citationWedatilake, Y., Plagnol, V., Anderson, G. W., Paine, S., Clayton, P. T., Jacques, T. S., & Rahman, S. (2014) · Neuropathology and Applied Neurobiology · doi:10.1111/nan.12190 · Cited by 10 · see citing works
He, X., Xiao, Y., Zhang, Q., Ma, L., Li, N., & Yang, J. (2013) · Chinese Medical Journal · doi:10.3760/cma.j.issn.0366-6999.20121155 · Cited by 4 · see citing works
Prasher, V. P. (1995) · Down Syndrome Research and Practice · doi:10.3104/reports.45 · Cited by 4 · see citing works
[Locomotion and posture disabilities in Down syndrome].
Haim, A., Lahav, A., Pritsch, T., Drexler, M., & Yizhar, Z. (2009) · PubMed · Cited by 2 · see citing works
Flagged first, then by citations
6 of the 135 references couldn’t be matched to a known paper.
Research works printed but not linked: 1
- Hill J. M. Lim M. A. and Stone M. M. (2008). Developmental Milestones in the Newborn Mouse. Neuropeptide Techniques. In Neuropeptide Techniques (ed. I. Gozes), 131-149. Clifton: Humana Press.
Cited by
Sorted by citations
Antonarakis, S. E., Skotko, B. G., Rafii, M. S., Strydom, A., Pape, S. E., Bianchi, D. W., Sherman, S. L., & Reeves, R. H. (2020) · Nature Reviews Disease Primers · doi:10.1038/s41572-019-0143-7 · Cited by 964 · see citing works
Self-citationThe diverse consequences of aneuploidy
Chunduri, N. K., & Štorchová, Z. (2018) · Nature Cell Biology · doi:10.1038/s41556-018-0243-8 · Cited by 207 · see citing works
Xu, R., Brawner, A. T., Li, S., Liu, J.-J., Kim, H., Xue, H., Pang, Z. P., Kim, W.-Y., Hart, R. P., Liu, Y., et al. (2019) · Cell Stem Cell · doi:10.1016/j.stem.2019.04.014 · Cited by 193 · see citing works
Trisomy 21 activates the kynurenine pathway via increased dosage of interferon receptors
Powers, R. K., Culp‐Hill, R., Ludwig, M. P., Smith, K. P., Waugh, K. A., Minter, R., Tuttle, K. D., Lewis, H. C., Rachubinski, A. L., Granrath, R. E., et al. (2019) · Nature Communications · doi:10.1038/s41467-019-12739-9 · Cited by 139 · see citing works
Down Syndrome and COVID-19: A Perfect Storm?
Espinosa, J. M. (2020) · Cell Reports Medicine · doi:10.1016/j.xcrm.2020.100019 · Cited by 130 · see citing works
Waugh, K. A., Minter, R., Baxter, J., Chi, C., Galbraith, M. D., Tuttle, K. D., Eduthan, N. P., Kinning, K. T., Andrysik, Z., Araya, P., et al. (2023) · Nature Genetics · doi:10.1038/s41588-023-01399-7 · Cited by 117 · see citing works
Galbraith, M. D., Rachubinski, A. L., Smith, K. P., Araya, P., Waugh, K. A., Enriquez-Estrada, B., Worek, K., Granrath, R. E., Kinning, K. T., Paul Eduthan, N., et al. (2023) · Science Advances · doi:10.1126/sciadv.adg6218 · Cited by 88 · see citing works
β2-microglobulin functions as an endogenous NMDAR antagonist to impair synaptic function
Gao, Y., Hong, Y., Huang, L., Zheng, S., Zhang, H., Wang, S., Yao, Y., Zhao, Y., Zhu, L., Xu, Q., et al. (2023) · Cell · doi:10.1016/j.cell.2023.01.021 · Cited by 75 · see citing works
Brain insulin resistance triggers early onset Alzheimer disease in Down syndrome
Tramutola, A., Lanzillotta, C., Di Domenico, F., Head, E., Butterfield, D. A., Perluigi, M., & Barone, E. (2020) · Neurobiology of Disease · doi:10.1016/j.nbd.2020.104772 · Cited by 73 · see citing works
JAK1 Inhibition Blocks Lethal Immune Hypersensitivity in a Mouse Model of Down Syndrome
Tuttle, K. D., Waugh, K. A., Araya, P., Minter, R., Orlicky, D. J., Ludwig, M., Andrysik, Z., Burchill, M. A., Tamburini, B. A. J., Sempeck, C., et al. (2020) · Cell Reports · doi:10.1016/j.celrep.2020.108407 · Cited by 72 · see citing works
New approaches to studying early brain development in Down syndrome
Baburamani, A. A., Patkee, P. A., Arichi, T., & Rutherford, M. A. (2019) · Developmental Medicine & Child Neurology · doi:10.1111/dmcn.14260 · Cited by 74 · see citing works
Ts66Yah, a mouse model of down syndrome with improved construct and face validity
Duchon, A., del Mar Muñiz Moreno, M., Chevalier, C., Nalesso, V., Andre, P., Fructuoso-Castellar, M., Mondino, M., Po, C., Noblet, V., Birling, M.-C., et al. (2022) · Disease Models & Mechanisms · doi:10.1242/dmm.049721 · Cited by 58 · see citing works
Network analysis of Down syndrome and SARS-CoV-2 identifies risk and protective factors for COVID-19
Toma, I. D., & Dierssen, M. (2021) · Scientific Reports · doi:10.1038/s41598-021-81451-w · Cited by 51 · see citing works
Martín, J. Z. D. S., Donato, C., Peixoto, J., Aguirre, A., Choudhary, V., Stasi, A. M. D., Lourenço, J., Potier, M., & Bacci, A. (2020) · eLife · doi:10.7554/elife.58731 · Cited by 49 · see citing works
Lanzillotta, C., Tramutola, A., Di Giacomo, G., Marini, F., Butterfield, D. A., Di Domenico, F., Perluigi, M., & Barone, E. (2021) · Free Radical Biology and Medicine · doi:10.1016/j.freeradbiomed.2021.01.042 · Cited by 47 · see citing works
Altered Hippocampal-Prefrontal Neural Dynamics in Mouse Models of Down Syndrome
Chang, P., Bush, D., Schorge, S., Good, M., Canonica, T., Shing, N., Noy, S., Wiseman, F. K., Burgess, N., Tybulewicz, V. L. J., et al. (2020) · Cell Reports · doi:10.1016/j.celrep.2019.12.065 · Cited by 47 · see citing works
Neurodevelopment in Down syndrome: Concordance in humans and models
Klein, J. A., & Haydar, T. F. (2022) · Frontiers in Cellular Neuroscience · doi:10.3389/fncel.2022.941855 · Cited by 47 · see citing works
“Similar to human studies, neuronal hypocellularity has been detected in multiple structures including the cortex, cerebellum, and hippocampus in postnatal trisomic animals (Insausti et al., 1998; Baxter et al., 2000; Olson et al., 2004; Roper et al., 2006; Chakrabarti et al., 2010; Aziz et al., 2018).”
BackgroundResultSelf-citationLaufer, B. I., Hwang, H., Ciernia, A. V., Mordaunt, C. E., & LaSalle, J. M. (2019) · Epigenetics · doi:10.1080/15592294.2019.1609867 · Cited by 47 · see citing works
Shaw, P. R., Klein, J. A., Aziz, N. M., & Haydar, T. F. (2020) · Disease Models & Mechanisms · doi:10.1242/dmm.046243 · Cited by 45 · see citing works
Self-citationDuchon, A., Moreno, M. D. M. M., Lorenzo, S. M., de Souza, M. P. S., Chevalier, C., Nalesso, V., Méziane, H., de Sousa, P. L., Noblet, V., Armspach, J., et al. (2021) · Human Molecular Genetics · doi:10.1093/hmg/ddab012 · Cited by 38 · see citing works
Navarro-Romero, A., Vázquez‐Oliver, A., Gomis‐González, M., Garzón-Montesinos, C., Falcón‐Moya, R., Pastor, A., Martín‐García, E., Pizarro, N., Busquets-García, A., Revest, J., et al. (2019) · Neurobiology of Disease · doi:10.1016/j.nbd.2019.01.014 · Cited by 35 · see citing works
Guedj, F., Kane, E., Bishop, L. A., Pennings, J. L. A., Hérault, Y., & Bianchi, D. W. (2023) · Biological Psychiatry · doi:10.1016/j.biopsych.2023.02.012 · Cited by 36 · see citing works
Self-citationAdvances and Applications of Brain Organoids
Li, Y., Zeng, P.-M., Wu, J., & Luo, Z. (2023) · Neuroscience Bulletin · doi:10.1007/s12264-023-01065-2 · Cited by 35 · see citing works
Lana-Elola, E., Cater, H., Watson-Scales, S., Greenaway, S., Müller-Winkler, J., Gibbins, D., Nemes, M., Slender, A., Hough, T., Keskivali-Bond, P., et al. (2021) · Disease Models & Mechanisms · doi:10.1242/dmm.049157 · Cited by 35 · see citing works
Perluigi, M., Picca, A., Montanari, E., Calvani, R., Marini, F., Matassa, R., Tramutola, A., Villani, A., Familiari, G., Domenico, F. D., et al. (2022) · Alzheimer's & Dementia · doi:10.1002/alz.12499 · Cited by 35 · see citing works
Thomas, M. S. C., Ojinaga-Alfageme, O., D’Souza, H., Patkee, P. A., Rutherford, M., Mok, K. Y., Hardy, J., & Karmiloff‐Smith, A. (2020) · Research in Developmental Disabilities · doi:10.1016/j.ridd.2020.103638 · Cited by 32 · see citing works
Guedj, F., Siegel, A. E., Pennings, J. L. A., Alsebaa, F., Massingham, L. J., Tantravahi, U., & Bianchi, D. W. (2020) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2020.10.001 · Cited by 31 · see citing works
Self-citationJAK inhibition decreases the autoimmune burden in Down syndrome
Rachubinski, A. L., Wallace, E., Gurnee, E., Enriquez-Estrada, B. A., Worek, K. R., Smith, K. P., Araya, P., Waugh, K. A., Granrath, R. E., Britton, E., et al. (2024) · eLife · doi:10.7554/elife.99323 · Cited by 32 · see citing works
Dysregulated systemic metabolism in a Down syndrome mouse model
Sarver, D. C., Xu, C., Vélez, L. M., Aja, S. M., Jaffe, A. E., Seldin, M. M., Reeves, R. H., & Wong, G. W. (2022) · Molecular Metabolism · doi:10.1016/j.molmet.2022.101666 · Cited by 31 · see citing works
Edlow, A. G., Guedj, F., Sverdlov, D., Pennings, J. L. A., & Bianchi, D. W. (2019) · Frontiers in Neuroscience · doi:10.3389/fnins.2019.01335 · Cited by 30 · see citing works
Self-citationAhmed, M. M., Wang, A. C., Elos, M. T., Chial, H. J., Sillau, S., Solano, D. A., Coughlan, C. M., Aghili, L., Anton, P. E., Markham, N. E., et al. (2022) · Neurobiology of Disease · doi:10.1016/j.nbd.2022.105694 · Cited by 30 · see citing works
Brain circuit pathology in Down syndrome: from neurons to neural networks
Bartesaghi, R. (2023) · Reviews in the Neurosciences · doi:10.1515/revneuro-2022-0067 · Cited by 25 · see citing works
Tramutola, A., Lanzillotta, S., Aceto, G., Pagnotta, S. E., Ruffolo, G., Cifelli, P., Marini, F., Ripoli, C., Palma, E., Grassi, C., et al. (2023) · Antioxidants · doi:10.3390/antiox12010111 · Cited by 25 · see citing works
Challenges and Opportunities for Translation of Therapies to Improve Cognition in Down Syndrome
Lee, S. E., Duran-Martinez, M., Khantsis, S., Bianchi, D., & Guedj, F. (2019) · Trends in Molecular Medicine · doi:10.1016/j.molmed.2019.10.001 · Cited by 23 · see citing works
Self-citationXing, Z., Li, Y., Cortes-Gomez, E., Jiang, X., Gao, S., Pao, A., Shan, J., Song, Y., Perez, A., Yu, T., et al. (2023) · Human Molecular Genetics · doi:10.1093/hmg/ddad056 · Cited by 23 · see citing works
Beckman, E. S., Deuitch, N. T., Michie, M., Allyse, M. A., Riggan, K. A., & Ormond, K. E. (2019) · The CRISPR Journal · doi:10.1089/crispr.2019.0021 · Cited by 22 · see citing works
Rescue of deficits by Brwd1 copy number restoration in the Ts65Dn mouse model of Down syndrome
Fulton, S. L., Wenderski, W., Lepack, A. E., Eagle, A. L., Fanutza, T., Bastle, R. M., Ramakrishnan, A., Hays, E. C., Neal, A., Bendl, J. M., et al. (2022) · Nature Communications · doi:10.1038/s41467-022-34200-0 · Cited by 21 · see citing works
Type I interferon signalling and interferon‐responsive microglia in health and disease
López‐Atalaya, J. P., & Bhojwani-Cabrera, A. M. (2025) · FEBS Journal · doi:10.1111/febs.70126 · Cited by 19 · see citing works
Mitsogiannis, M. D., Pancho, A., Aerts, T., Sachse, S., Vanlaer, R., Noterdaeme, L., Schmucker, D., & Seuntjens, E. (2021) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2020.624181 · Cited by 20 · see citing works
Adams, A. D., Guedj, F., & Bianchi, D. W. (2019) · Placenta · doi:10.1016/j.placenta.2019.10.002 · Cited by 18 · see citing works
Self-citationMoyer, A. J., Fernandez, F., Li, Y., Klinedinst, D. K., Florea, L., Kazuki, Y., Oshimura, M., & Reeves, R. H. (2023) · Disease Models & Mechanisms · doi:10.1242/dmm.049712 · Cited by 17 · see citing works
Down syndrome: A curative prospect?
Rondal, J. A. (2020) · AIMS neuroscience · doi:10.3934/neuroscience.2020012 · Cited by 14 · see citing works
Behavioral Phenotyping for Down Syndrome in Mice
Roper, R. J., Goodlett, C., de Lagrán, M. M., & Dierssen, M. (2020) · Current Protocols in Mouse Biology · doi:10.1002/cpmo.79 · Cited by 10 · see citing works
Cristancho, A. G., Gadra, E. C., Samba, I., Zhao, C., Ouyang, M., Magnitsky, S. G., Huang, H., Viaene, A. N., Anderson, S. A., & Marsh, E. D. (2022) · Developmental Neuroscience · doi:10.1159/000524045 · Cited by 14 · see citing works
Biphasic cell cycle defect causes impaired neurogenesis in down syndrome
Sharma, V., Nehra, S., Do, L. H., Ghosh, A., Deshpande, A. J., & Singhal, N. (2022) · Frontiers in Genetics · doi:10.3389/fgene.2022.1007519 · Cited by 14 · see citing works
Cates, H. M., Benca‐Bachman, C. E., de Guglielmo, G., Schoenrock, S. A., Shu, C., & Kallupi, M. (2019) · Genes Brain & Behavior · doi:10.1111/gbb.12577 · Cited by 13 · see citing works
Watson, L. A., & Meharena, H. S. (2023) · Frontiers in Genetics · doi:10.3389/fgene.2023.1198129 · Cited by 13 · see citing works
Interferon receptor gene dosage determines diverse hallmarks of Down syndromePreprint
Waugh, K. A., Minter, R., Baxter, J. A., Chi, C., Tuttle, K. D., Eduthan, N. P., Galbraith, M. D., Kinning, K. T., Andrysík, Z., Araya, P., et al. (2022) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2022.02.03.478982 · Cited by 13 · see citing works
Neocortical neuronal production and maturation defects in the TcMAC21 mouse model of Down syndrome
Kurabayashi, N., Fujii, K., Otobe, Y., Hiroki, S., Hiratsuka, M., Yoshitane, H., Kazuki, Y., & Takao, K. (2023) · iScience · doi:10.1016/j.isci.2023.108379 · Cited by 13 · see citing works
Lanzillotta, S., Esteve, D., Lanzillotta, C., Tramutola, A., Lloret, A., Forte, E., Pesce, V., Picca, A., Domenico, F. D., Marzia Perluigi, A., et al. (2024) · Free Radical Biology and Medicine · doi:10.1016/j.freeradbiomed.2024.11.043 · Cited by 12 · see citing works
Early impacts of modified food consistency on oromotor outcomes in mouse models of Down syndrome
Glass, T. J., Twadell, S., Valmadrid, L. C., & Connor, N. P. (2018) · Physiology & Behavior · doi:10.1016/j.physbeh.2018.11.031 · Cited by 12 · see citing works
Viard, J., Loe-Mie, Y., Daudin, R., Khelfaoui, M., Plançon, C., Boland, A., Tejedor, F. J., Huganir, R. L., Kim, E., Kinoshita, M., et al. (2022) · Life Science Alliance · doi:10.26508/lsa.202101205 · Cited by 11 · see citing works
Dierssen, M., Hérault, Y., Helguera, P. R., de Lagrán, M. M., Vázquez, A. V., Christian, B. T., Carmona‐Iragui, M., Wiseman, F. K., Mobley, W. C., Fisher, E., et al. (2021) · Molecular Syndromology · doi:10.1159/000514437 · Cited by 11 · see citing works
Self-citationHawley, L. E., Prochaska, F., Stringer, M., Goodlett, C. R., & Roper, R. J. (2022) · Pharmacology Biochemistry and Behavior · doi:10.1016/j.pbb.2022.173404 · Cited by 11 · see citing works
Hawley, L. E., Stringer, M., Deal, A. J., Folz, A., Goodlett, C. R., & Roper, R. J. (2024) · Neurobiology of Disease · doi:10.1016/j.nbd.2023.106359 · Cited by 10 · see citing works
Fosu, K., Quarshie, J. T., Sarpong, K. A. N., & Aikins, A. R. (2023) · Genes · doi:10.3390/genes14040800 · Cited by 10 · see citing works
Llambrich, S., González, R., Albaigès, J., Wouters, J., Marain, F., Himmelreich, U., Sharpe, J. A., Dierssen, M., Gsell, W., Martínez‐Abadías, N., et al. (2022) · Frontiers in Medicine · doi:10.3389/fmed.2022.815739 · Cited by 10 · see citing works
Ultrasonic vocalization phenotypes in the Ts65Dn and Dp(16)1Yey mouse models of Down syndrome
Glass, T. J., Lenell, C., Fisher, E. H., Yang, Q., & Connor, N. P. (2023) · Physiology & Behavior · doi:10.1016/j.physbeh.2023.114323 · Cited by 9 · see citing works
Influence of allelic differences in Down syndrome
Roper, R. J., Hawley, L. E., & Goodlett, C. R. (2020) · Progress in brain research · doi:10.1016/bs.pbr.2019.09.001 · Cited by 9 · see citing works
Aziz, N. M., Klein, J. A., Brady, M. R., Olmos-Serrano, J. L., Gallo, V., & Haydar, T. F. (2019) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-019-9294-9 · Cited by 9 · see citing works
Self-citationIshihara, K. (2021) · Genes · doi:10.3390/genes12101598 · Cited by 8 · see citing works
Li, Y., Xing, Z., Yu, T., Pao, A., Daadi, M. M., & Yu, Y. E. (2021) · Genes · doi:10.3390/genes12081215 · Cited by 9 · see citing works
Islam, R., Jackson, B., Naomi, M. T., Schatz, B., Tan, N., Murdock, M. H., Park, D. S., Rodrígues-Amorím, D., Jiang, X., Pineda, S. S., et al. (2025) · PLoS ONE · doi:10.1371/journal.pone.0317428 · Cited by 8 · see citing works
Llambrich, S., Wouters, J., Himmelreich, U., Dierssen, M., Sharpe, J. A., Gsell, W., Martínez‐Abadías, N., & Velde, G. V. (2020) · Scientific Reports · doi:10.1038/s41598-020-75720-3 · Cited by 8 · see citing works
From the lab to the people: major challenges in the biological treatment of Down syndrome
Rondal, J. A. (2021) · AIMS neuroscience · doi:10.3934/neuroscience.2021015 · Cited by 8 · see citing works
Araya, P., Niemeyer, B., Schade, K., Dunn, L. N., Waugh, K., Busquet, N., Brindley, C., Brown, C., Winkler, C., Lyford, H. R., et al. (2025) · iScience · doi:10.1016/j.isci.2025.113130 · Cited by 7 · see citing works
“We next tested motor coordination using the accelerating rotarod test DS-AD mice (Data S4), and consistent with previous reports, 38,40 we found that Dp16 mice exhibit impaired motor co-ordination at 3 and 5 months of age relative to WT littermates, with a trend toward poorer performance at 7 and 9…”
InfluentialTolu, S., Hamzé, R., Moreau, M., Bertrand, R., Janel, N., & Movassat, J. (2025) · Diabetes Obesity and Metabolism · doi:10.1111/dom.16155 · Cited by 7 · see citing works
Embryonic statistical analyses reveal 2 growth phenotypes in mouse models of Down syndrome
Adams, A. D., Lin, J., Bianchi, D. W., Bishop, L. A., Sato, T., Baxter, L. L., Hoffmann, V. J., Koehly, L. M., & Guedj, F. (2023) · American Journal of Obstetrics and Gynecology · doi:10.1016/j.ajog.2023.07.056 · Cited by 7 · see citing works
Self-citationFranco, N. D., Drutel, G., Roullot, V., Julio‐Kalajzić, F., Lalanne, V., Grel, A., Lesté-Lasserre, T., Matias, I., Cannich, A., Gonzales, D., et al. (2022) · Molecular and Cellular Neuroscience · doi:10.1016/j.mcn.2022.103705 · Cited by 7 · see citing works
Ahmed, M. M., Block, A., Busquet, N., & Gardiner, K. (2021) · Genes · doi:10.3390/genes12101528 · Cited by 7 · see citing works
Patthy, Á., Hanics, J., Zachár, G., Kovács, G., Harkany, T., & Alpár, A. (2023) · Neuropathology and Applied Neurobiology · doi:10.1111/nan.12887 · Cited by 5 · see citing works
Guez‐Barber, D., Eisch, A. J., & Cristancho, A. G. (2023) · Developmental Neuroscience · doi:10.1159/000530745 · Cited by 7 · see citing works
Shimizu, R., Ishihara, K., Kawashita, E., Sago, H., Yamakawa, K., Mizutani, K.-I., & Akiba, S. (2020) · Biochemical and Biophysical Research Communications - BBRC · doi:10.1016/j.bbrc.2020.12.026 · Cited by 3 · see citing works
Novel insights from fetal and placental phenotyping in 3 mouse models of Down syndrome
Adams, A. D., Hoffmann, V. J., Koehly, L. M., Guedj, F., & Bianchi, D. W. (2021) · American Journal of Obstetrics and Gynecology · doi:10.1016/j.ajog.2021.03.019 · Cited by 5 · see citing works
Self-citationLlambrich, S., Tielemans, B., Saliën, E., Atzori, M., Wouters, K., Bulck, V. V., Platt, M., Vanherp, L., Fernandez, N. G., de la Fuente, L. G., et al. (2024) · eLife · doi:10.7554/elife.89763.3 · Cited by 5 · see citing works
Ahmed, M. M., Wang, A. C.-J., Elos, M. T., Chial, H. J., Sillau, S. H., Solano, D., Coughlan, C. M., Aghili, L., Anton, P., Markham, N., et al. (2021) · bioRxiv · doi:10.1101/2021.07.04.451073 · Cited by 2 · see citing works
Shao, L., & Stafstrom, C. E. (2025) · Epilepsy Research · doi:10.1016/j.eplepsyres.2025.107686 · Cited by 4 · see citing works
Fatemi, S. H., Otte, E. D., Folsom, T. D., Eschenlauer, A. C., Roper, R. J., Aman, J. W., & THURAS, P. D. (2024) · Genes · doi:10.3390/genes15040452 · Cited by 4 · see citing works
Silva, A. L. T. E., de Oliveira, P. H. P., Yokota-Moreno, B. Y., Fausto, J. D. S., Avila, J. P., Nakaya, H. I., Sertie, A. L., & Zampieri, B. L. (2025) · Acta Neuropathologica Communications · doi:10.1186/s40478-025-02090-0 · Cited by 5 · see citing works
Canonica, T., Kidd, E. J., Gibbins, D., Lana‐Elola, E., Fisher, E., Tybulewicz, V. L. J., & Good, M. A. (2024) · Frontiers in Behavioral Neuroscience · doi:10.3389/fnbeh.2024.1428146 · Cited by 3 · see citing works
Characterization of Apathy-Like Behaviors in Mouse Models of Down Syndrome
Zhang, T., Wang, X., Jester, H. M., Zhou, X.-Y., & Ma, T. (2024) · Journal of Alzheimer's Disease · doi:10.3233/jad-240675 · Cited by 3 · see citing works
Jin, J., Doan, J., Fernandez, C., Nguyen, S., Spencer, C., & Kleschevnikov, A. M. (2025) · Experimental Neurology · doi:10.1016/j.expneurol.2025.115171 · Cited by 3 · see citing works
Siegel, A. E., Bianchi, D. W., & Guedj, F. (2023) · Journal of Neuroscience Research · doi:10.1002/jnr.25160 · Cited by 3 · see citing works
Self-citationThomas, S. N., Niemeyer, B. F., Jiménez-Valdés, R. J., Kaiser, A. J., Espinosa, J. M., Sullivan, K. D., Goodspeed, A. E., Costello, J. C., Alder, J. K., Cañas‐Arranz, R., et al. (2023) · iScience · doi:10.1016/j.isci.2023.107361 · Cited by 3 · see citing works
JAK inhibition decreases the autoimmune burden in Down syndromePreprint
Rachubinski, A. L., Wallace, E. B., Gurnee, E., Estrada, B. A. E., Worek, K. R., Smith, K. P., Araya, P., Waugh, K. A., Granrath, R. E., Britton, E. C., et al. (2024) · eLife · doi:10.7554/elife.99323.1 · Cited by 3 · see citing works
Atak, Z., Srivastava, A., Gandhi, S., Park, E., Williams, J., Torii, M., & Haydar, T. F. (2025) · Journal of Neuroscience · doi:10.1523/jneurosci.0040-25.2025 · Cited by 2 · see citing works
Self-citationHaydar, T. F. (2020) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-125.2.100 · Cited by 2 · see citing works
Self-citationMitigating Cognitive Deficits in Down Syndrome by Managing Microglia Activation
Shaw, P. R., & Haydar, T. F. (2020) · Neuron · doi:10.1016/j.neuron.2020.11.017 · Cited by 2 · see citing works
Self-citationSiegel, A. E., Bianchi, D. W., & Guedj, F. (2020) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2020.07.21.214106 · Cited by 2 · see citing works
Self-citationGuedj, F., Kane, E., Bishop, L. A., Pennings, J. L. A., Hérault, Y., & Bianchi, D. W. (2022) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2022.09.23.509067 · Cited by 2 · see citing works
Self-citationComplement system alteration in iPSC-derived astrocytes from individuals with Down syndromePreprint
Silva, A. L. T. E., de Oliveira, P. H. P., Yokota-Moreno, B. Y., Avila, J. P., Nakaya, H. I., Fausto, J. D. S., Sertie, A. L., & Zampieri, B. L. (2024) · Research Square · doi:10.21203/rs.3.rs-5319960/v1 · Cited by 2 · see citing works
Gadra, E. C., & Cristancho, A. G. (2022) · BIO-PROTOCOL · doi:10.21769/bioprotoc.4519 · Cited by 1 · see citing works
Adeeb, N., Farooqui, S. I., Hasan, Z. M., Khan, A., & Rizvi, J. (2024) · Developmental Neurorehabilitation · doi:10.1080/17518423.2024.2365798 · Cited by 1 · see citing works
Mapping behavioral landscapes in Down syndrome animal models
de Lagrán, M. M. (2020) · Progress in brain research · doi:10.1016/bs.pbr.2020.01.005 · Cited by 1 · see citing works
Modeling Down Syndrome Neurodevelopment with Dosage Compensation
Czerminski, J. T. (2019) · eScholarship@UMassChan · doi:10.13028/j7xf-1q21 · Cited by 1 · see citing works
Llambrich, S., Tielemans, B., Saliën, E., Atzori, M., Wouters, K., Bulck, V. V., Platt, M., Vanherp, L., Fernandez, N. G., de la Fuente, L. G., et al. (2023) · eLife · doi:10.1101/2023.07.31.551282 · Cited by 6 · see citing works
JAK inhibition decreases the autoimmune burden in Down syndromePreprint
Rachubinski, A. L., Wallace, E. B., Gurnee, E., Estrada, B. A. E., Worek, K. R., Smith, K. P., Araya, P., Waugh, K. A., Granrath, R. E., Britton, E. C., et al. (2024) · eLife · doi:10.7554/elife.99323.2 · Cited by 1 · see citing works
Cognitive function in mice with segmental trisomy of human chromosome 21 Down Syndrome genes
Canonica, T. (2019) · ORCA Online Research @Cardiff (Cardiff University)
Detection of Novel Genes on Recessive Heterozygous Alleles in Pig Breeding: A Review
Tran, H. T., Linh, N. T., Le, T. M., Nam, T. V., & Bui, A. P. N. (2022) · Indian Journal of Animal Research · doi:10.18805/ijar.bf-1486
Llambrich, S., Tielemans, B., Saliën, E., Atzori, M., Wouters, K., Bulck, V. V., Platt, M., Vanherp, L., Fernandez, N. G., de la Fuente, L. G., et al. (2023) · eLife · doi:10.7554/elife.89763.1
- Authors
- Aziz, N. M., Guedj, F., Pennings, J. L. A., Olmos-Serrano, J. L., Siegel, A., Haydar, T. F., & Bianchi, D. W.
- Affiliations
- Boston University (US)
- National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland 20892, USA
- National Institute for Public Health and the Environment (NL)
- National Institutes of Health (US)
- Corresponding author
- Aziz, Nadine M.
- ORCID
- Venue
- Disease Models & Mechanisms
- Publisher
- The Company of Biologists
- Published
- 2018-01-01
- Volume
- 11
- Issue
- 6
- ISSN
- 1754-8403 (ISSN-L)
- 1754-8411
- PMC
- PMC6031353
- OpenAlex
- W2801858511
- Cited by
- 118
- References
- 135
- Citations by year
- 2026: 4 · 2025: 12 · 2024: 19 · 2023: 21 · 2022: 16 · 2021: 13 · 2020: 18 · 2019: 13 · 2018: 2
- FWCI
- 10.97
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- Disease Models & Mechanisms (publishedVersion, cc-by)
- Europe PMC (PubMed Central) (publishedVersion, other-oa)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion)
- HighWire Press Open Archive (submittedVersion)
- Rivm Repository (Netherlands National Institute for Public Health and the Environment) (submittedVersion)
- Rivm (National Institute for Public Health and the Environment) (submittedVersion)
- Funders
- National Institutes of Health (R01NS076503, R01HD058880)
- Illumina (250-10396-12547000001)
- Anna and John J. Sie Foundation (250-10488-40026002016)
- Topics
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Down syndrome and intellectual disability research
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
- Keywords
- Down syndrome, Ts65Dn mouse model, mouse models of Down syndrome, brain development, neurogenesis, brain morphogenesis, physical growth, differentially expressed genes, gene expression, intellectual disability, motor behavior, model comparison
- MeSH terms
- Animals, Animals, Newborn, Behavior, Animal, Brain, Disease Models, Animal, Down Syndrome, Female, Gene Expression Regulation, Hippocampus, Longevity, Male, Mice, Inbred C57BL, Motor Activity, Neurons, Phenotype, Genome, Neurogenesis
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review
