Evidence map›Paper›PMID 40220293›Full record

ArticleCell reports2025

Multi-site investigation of gut microbiota in CDKL5 deficiency disorder mouse models: Targeting dysbiosis to improve neurological outcomes.

Francesca Damiani, Maria Grazia Giuliano, Sara Cornuti, Elena Putignano, Andrea Tognozzi, Vanessa Suckow, Vera M Kalscheuer, Tommaso Pizzorusso, Paola Tognini

Abstract read
In one paragraph

Article in Cell reports, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

4 citing papers in PubMed.

  1. Article
  2. The Critical Period Microbiota Shape Brain Plasticity.bioRxiv : the preprint server for biology · 2026
    Article
  3. Review
  4. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors.

Francesca DamianiLaboratory of Biology BIO@SNS, Scuola Normale Superiore, Piazza dei Cavalieri 7, 56126 Pisa, Italy.
Maria Grazia GiulianoLaboratory of Biology BIO@SNS, Scuola Normale Superiore, Piazza dei Cavalieri 7, 56126 Pisa, Italy; Health Science Interdisciplinary Center, Sant'Anna School of Advanced Studies, Piazza Martiri della Libertà 33, 56127 Pisa, Italy.
Sara CornutiLaboratory of Biology BIO@SNS, Scuola Normale Superiore, Piazza dei Cavalieri 7, 56126 Pisa, Italy.
Elena PutignanoInstitute of Neuroscience, National Research Council, Via G. Moruzzi 1, 56124 Pisa, Italy.
Andrea TognozziLaboratory of Biology BIO@SNS, Scuola Normale Superiore, Piazza dei Cavalieri 7, 56126 Pisa, Italy; PhD Program in Clinical and Translational Science, University of Pisa, Via Savi 10, 56126 Pisa, Italy.
Vanessa SuckowMax Planck Institute for Molecular Genetics, Ihnestraße 63, 14195 Berlin, Germany.
Vera M KalscheuerMax Planck Institute for Molecular Genetics, Ihnestraße 63, 14195 Berlin, Germany.
Tommaso PizzorussoLaboratory of Biology BIO@SNS, Scuola Normale Superiore, Piazza dei Cavalieri 7, 56126 Pisa, Italy; Institute of Neuroscience, National Research Council, Via G. Moruzzi 1, 56124 Pisa, Italy.
Paola TogniniHealth Science Interdisciplinary Center, Sant'Anna School of Advanced Studies, Piazza Martiri della Libertà 33, 56127 Pisa, Italy. Electronic address: paola.tognini@santannapisa.it.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder (CDD) is a rare neurodevelopmental disorder often associated with gastrointestinal (GI) issues and subclinical immune dysregulation, suggesting a link to the gut microbiota. We analyze the fecal microbiota composition in two CDKL5 knockout (KO) mouse models at postnatal days (P) 25, 32 (youth), and 70 (adulthood), revealing significant microbial imbalances, particularly during juvenile stages. To investigate the role of the intestinal microbiota in CDD and assess causality, we administer antibiotics, which lead to improved visual cortical responses and reduce hyperactivity. Additionally, microglia morphology changes, indicative of altered surveillance and activation states, are reversed. Strikingly, fecal transplantation from CDKL5 KO to wild-type (WT) recipient mice successfully transfers both visual response deficits and hyperactive behavior. These findings show that gut microbiota alterations contribute to the severity of neurological symptoms in CDD, shedding light on the interplay between microbiota, microglia, and neurodevelopmental outcomes.

Indexed as

DysbiosisEpileptic SyndromesGastrointestinal MicrobiomeProtein Serine-Threonine KinasesRett SyndromeSpasms, InfantileAnimalsDisease Models, AnimalFecal Microbiota TransplantationFemaleMaleMiceMice, Inbred C57BLMice, KnockoutMicrogliaCDKL5 protein, mouseProtein Serine-Threonine KinasesantibioticsCDKL5CP: MicrobiologyCP: Neurosciencedysbiosisfecal transplantationgut-brain axisgut microbiotamicroglianeurodevelopmental disorder

Identifiers

PMID40220293
PMCPMC12014524

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.