The Complex Causes of Autism Presenter Slide Deck

What causes autism? In Autism New Jersey’s original webinar series, Rutgers researchers will break down the science in simple terms. To watch the recorded presentations, visit www.autismnj.org/article/the-complex-causes-of-autism-webinar-series

Questions? Call 800.4.AUTISM or visit www.autismnj.org

M. Chiara Manzini, PhD Associate Professor Neuroscience and Cell Biology Rutgers Robert Wood Johnson Medical School

Child Health Institute of New Jersey

LANGUAGE USED

- autistic person/child will be used for individuals with an autism spectrum disorder diagnosis

- male and female will be used for individuals with XY or XX chromosomal sex, respectively

- profound autism will be used for autistic people who are nonverbal/ minimally verbal, with moderate to severe intellectual disability, and need 24/7 supervision

- (scientific jargon) in parentheses

THE AUTISM SPECTRUM

Sex bias 4:1 Male:Female

(Rosen et al., J of Autism and Developmental Disorders, 2021)

WHY SHOULD WE LOOK AT GENES?

❑ 60-90% of identical (monozygotic) twins both have ASD ❑ Fraternal (dizygotic) twins have a much lower chance of both having ASD (3-30%)

Autism is highly genetic, but how is it inherited?

HALF OF OUR GENOME COMES FROM EACH PARENT

chromosome

Identical twins come from the same cell = same genome

HOW ARE GENETIC CONDITIONS INHERITED?

5%

2%

One genetic change

Multiple genetic changes

100%

10%

8%

Multiple genetic changes (polygenic) ➤ each adds some risk for the condition Type 2 diabetes, Polycystic ovary syndrome

One genetic change (monogenic) ➤ altering one gene is sufficient

Sickle cell disease, Duchenne muscular dystrophy

IS THERE ONLY ONE GENETIC CAUSE OR MANY?

The same gene or combination of genes lead to ASD

Many different genetic disruptions can lead to ASD

THE CHALLENGES

❑ Single-gene or multi-gene conditions are studied in different ways ❑ Single-gene: Collect DNA from autistic individuals and family members ❑ Multi-gene: Genome-wide association studies (GWAS) with DNA from large numbers of cases and controls

❑ ASD presentation is variable

❑ Severe ASD can be part of a more complex genetic syndrome (e.g. Fragile X Syndrome)

❑ Initial studies revealed that we need thousands of participants for either one

Large research groups

Studies

(de Sousa Nobrega et al., Int J Mol Sci, 2024)

THERE ARE MANY DIFFERENT GENETIC CAUSES OF ASD

(Autism Science Foundation)

THERE ARE MANY DIFFERENT GENETIC CAUSES OF ASD – SINGLE-GENE (FRAGILE X SYNDROME)

❑ Intellectual disability ❑ 20-30% are diagnosed with ASD ❑ Facial dysmorphism, epilepsy ❑ on the X chromosome – more severe in boys

❑ FMR1 regulates a large network of other genes

❑ Accounts for up to 1% of autistic individuals ❑ Inherited single gene variants account for 3-5% of ASD

THERE ARE MANY DIFFERENT GENETIC CAUSES OF ASD – SINGLE NUCLEOTIDE VARIANTS (SNV)

de novo

(Fu et al, Nature Genetics, 2022)

WHAT IS A DE NOVO VARIANT?

❑ Only found in the autistic child on one chromosome (heterozygous) ❑ Originated in the sperm or egg of the parent

❑ De novo variants are preferentially inherited from older fathers ❑ Sperm is produced throughout life and is more susceptible to de novo changes with age

(Antaki et al, Nature Genetics, 2022)

THERE ARE MANY DIFFERENT GENETIC CAUSES OF ASD – COPY NUMBER VARIANTS (CNV)

(Searles Quick et al., Neuropsychopharmacology, 2021)

WHAT ABOUT THE MULTI-GENE CAUSES?

(Autism Science Foundation)

THERE ARE MANY DIFFERENT GENETIC CAUSES OF ASD – COMMON VARIANTS (MULTI-GENE)

❑ Each variant adds a little bit of risk

ASD RISK VARIANTS ADD UP

2%

5%

2%

90%

90%

8%

8%

3%

++

+++

+

ASD severity

CAN GENETICS EXPLAIN THE MALE BIAS?

❑ YES and NO ❑ Girls need more genetic risk variants to develop ASD

❑ Current rare single gene changes have same risk in males and females

(Koko et al, Am J Human Genetics, 2025; 21,501 ASD, 9,223 TD sibs)

(Antaki et al., Nature Genetics, 2022; 12,270 ASD, 5,190 TD sibs, 19,917 parents)

WHY ARE THERE SO MANY GENES?!

One third (15,000+) of 20,000+ human genes are active in the brain

Neuron

(Bekkers, 2011)

EACH NEURON

❑ Can receive more than 10,000 contacts (synapses)

Information collection

❑ There are 80 billion neurons in the human brain

❑ 800 trillion contacts/synapses

Response

THE NEURON’S GENES

The neuron’s genes control: ▪ Identity ▪ Location ▪ Response ▪ Plasticity

GENES MUTATED IN ASD

ASD risk genes control: ▪ neuronal communication, synapse function and plasticity ▪ synaptic proteins, ion channels ▪ expression of hundreds of other genes ▪ epigenetic regulators, transcription factors ▪ neuronal responses to nutrients and growth factors ▪ signaling proteins

ASD risk genes are involved in fetal development They are often only partially changed in their function

HOW DOES THIS HELP ME?

❑ Inherited and de novo monogenic cases can benefit from research on their specific gene ❑ rate of inheritance ❑ knowledge on how the condition progresses ❑ specialized therapies (gene therapy?) ❑ We can learn how to group genetic causes for better therapeutics

❑ Results on brain changes found in genetic cases can be applied to other autistic individuals

(Chahrour et al., J Neuroscience, 2016)

WHAT ARE WE STILL MISSING?

❑ Most genetic research has been done of people of European ancestry ❑ Asia, Africa, and Latin America may have non-overlapping genetic causes ❑ More recruitment is needed in both European and non-European populations for polygenic and ultra-rare variants ❑ Maternal genetics – maternal risk and changes in the function of the placenta during fetal development ❑ Gene X environment interactions ❑ More comparison of single-gene causes to find patterns

RESOURCES

❑ Simons Foundation – SFARI Gene https://gene.sfari.org/ ❑ Recurrence rates from ASF - https://autismsciencefoundation.org/quick-facts- about-autism/

❑ Current genetic study recruitment in NJ ❑ Simons Foundation for Autism Research – SPARK project recruitment at Children’s Specialized Hospital ❑ https://sparkforautism.org/csh ❑ sparkcsh@childrens-specialized.org ❑ “ SPARK is a privately funded research study on autism, meaning we receive no money from the government. Your privacy is very important to us.”

Goals for today

• Define “environment” and how it is measured • Describe mechanisms by which environment and genetics interact • Provide a few (not exhaustive) examples of gene x environment interactions in autism

Core and Associated Features

Rosen, Lord and Volkmar, 2022

Autism is Complex

Diagnosis or Dimension?

Increase in prevalence = Something in the environment?

Increase in prevalence = Correlation does not mean causation

First genes, now environment

Typically, they have been studied separately

Typically, they have been studied separately

They are not always independent things

Environmental effects may reflect genetic influences

How are they studied?

Direct assessment

Electronic Medical Records Monitoring station

Self Report

Environmental Factors on Autism Diagnosis

Exposure

Relative Risk/Odds Ration

Air Pollution during pregnancy

1.25-2x

Illness during pregnancy

2-3x 1.3x 4-5x

Maternal diabetes (blood sugar?)

Prematurity /very low birth weight/traumatic birth

Parental age

1.3 – 1.4x

Valproic acid (anti – seizure)

5x

Studied, no association

Exposure Plasticizers

Medical procedure/medicine

Terbutaline Fluoxetine

Maternal cigarette smoking

Maternal marijuana

IVF

Breast feeding

C-section Vaccines acetaminophen Tomiprimate (anti-seizure) antipsychotics

A model for both Causes

Genetics of mother and father

Gene/ Environment

Environment

HOW??

Genetics of fetus

Autism Phenotype

Which genome? Which environment?

Gardner et al., 2024

A model for both

Gene-Environment

Gene susceptibility or resilience

Gene transcription

DNA damage or repair

Overlapping targets

epigenetics

Immune

microbiome

Example of one influencing the other: Advanced Parental Age

Result of genetics?

Older Parents

Dehesh et al., 2024

Each color a different family

Diagnosis or Dimension? Vitamin D

Small effect in f only

Lower Vitamin D levels, higher ASD features Result of genetics?

Horsdal et al., 2025

NT overall male female

Aargard et al, 2024

Wyndham et al, 2020

Maternal Conditions

What is the link? Underlying gene or high blood sugar?

Gestational diabetes

Sibling analyses

Sibling – no diabetes but ASD dx

Beier et al., 2025

Guo et al., 2022

Folic Acid Protection

ENVIRONMENT THAT TARGETS THOSE GENES

GENETIC INFLUENCES-MTHFR677

Schmidt et al., 2012

Focusing on the antioxidant pathway

Folic Acid + pesticide exposure

Schmidt et al., 2017

Takeaways • Autism is not caused by one thing, even though scientists have been studying one thing at a time

• Many environmental associations may be attributed to genetic variations

• Beware of correlative studies

• You can’t study gene x environment interactions without understanding genetics – support newborn screening and genetic testing in ASD

Ultrasound

40

ASD

35

no ASD

30

Ultrasound by itself – diagnosis? – no difference

25

20

15

10

5

0

0

1

2

3

4

5

6+

Grether et al., 2010

Copy Number Variation

Copy Number Variants

5 x more likely to have an autism diagnosis if there is a CNV present • 100+ genes • Also seen in ADHD, Schizophrenia and IDD

Mollon et al., 2023

CNV + ultrasound on behavior

Domain

Effect

Social affect

11.4 vs. 11 – small diff 6.4 to 6.6 – small diff

Restricted and repetitive behaviors using observation

In those with CNVs and autism and ultrasound Dimensional not diagnostic

Noverbal IQ

87 vs 76 – larger diff

Social reciprocity RRB using parent report

No diff No diff

Webb et al., 2016

CNV + infection on autism features

Domain

Effect alone

Effect combined

Social communication

No changes with each alone No changes with each alone

Changes with both (16 to 18)

Social Interaction Increase scores with both (20 to 22) Nonverbal IQ Effect on CNV Same as CNV alone Social responsiveness Slight decrease with CNV Changes with both RRB using parent report and observation Slight decrease with CNV Changes with both

Mazina et al. 2015

Interaction – mechanism?

40

35

30

25

20

15

10

5

0

communication

social interaction

RRB

No CNV no MI

No CNV + MI

CNV no MI

CNV + MI

Mazina et al. 2015

References 1. Rosen, N. E., Lord, C., & Volkmar, F. R. (2021). The Diagnosis of Autism: From Kanner to DSM-III to DSM-5 and Beyond . Journal of Autism and Developmental Disorders , 51 (12), 4253 – 4270. https://doi.org/10.1007/s10803-021-04904-1

2. Gardner, R. M., Brynge, M., Sjöqvist, H., Dalman, C., & Karlsson, H. (2024). Maternal Immune Activation and Autism in Offspring: What Is the Evidence for Causation? Biological Psychiatry , S0006-3223(24)01760-8. https://doi.org/10.1016/j.biopsych.2024.11.009

3. Dehesh, T., Mosleh-Shirazi, M. A., Jafari, S., Abolhadi, E., & Dehesh, P. (2024). A assessment of the effects of parental age on the development of autism in children: A systematic review and a meta-analysis. BMC Psychology , 12(1), 685. https://doi.org/10.1186/s40359-024-02184-9

4. Windham, G. C., Pearl, M., Poon, V., Berger, K., Soriano, J. W., Eyles, D., Lyall, K., Kharrazi, M., & Croen, L. A. (2020). Maternal Vitamin D Levels During Pregnancy in Association With Autism Spectrum Disorders (ASD) or Intellectual Disability (ID) in Offspring; Exploring Non-linear Patterns and Demographic Sub-groups. Autism Research: Official Journal of the International Society for Autism Research , 13(12), 2216 – 2229. https://doi.org/10.1002/aur.2424

References 5. Aagaard, K., Møllegaard Jepsen, J. R., Sevelsted, A., Horner, D., Vinding, R., Rosenberg, J. B., Brustad, N., Eliasen, A., Mohammadzadeh, P., Følsgaard, N., Hernández-Lorca, M., Fagerlund, B., Glenthøj, B. Y., Rasmussen, M. A., Bilenberg, N., Stokholm, J., Bønnelykke, K., Ebdrup, B. H., & Chawes, B. (2024). High-dose vitamin D3 supplementation in pregnancy and risk of neurodevelopmental disorders in the children at age 10: A randomized clinical trial. The American Journal of Clinical Nutrition , 119(2), 362 – 370. https://doi.org/10.1016/j.ajcnut.2023.12.002 6. Horsdal, H. T., Albiñana, C., Zhu, Z., Boelt, S. G., Borbye-Lorenzen, N., Cohen, A. S., Skogstrand, K., Melgaard, L., MacSween, N. J., Thorbek, M. J., Plana-Ripoll, O., Petersen, L. V., Bulik, C. M., BRglum, A. D., Mors, O., Nordentoft, M., Werge, T., Moen, G.-H., D’Urso, S., … McGrath, J. J. (2025). Convergent evidence linking neonatal vitamin D status and risk of neurodevelopmental disorders: A Danish case-cohort study. The Lancet Psychiatry , 12(6), 410 – 420. https://doi.org/10.1016/S2215-0366(25)00099-9 7. Guo, D., Ju, R., Dai, J., & Zhang, H. (2025). Autism Spectrum Disorders in Offspring Exposed to Maternal Gestational Diabetes: A Meta-Analysis and Systematic Review. Review Journal of Autism and Developmental Disorders , 12(1), 69 – 79. https://doi.org/10.1007/s40489-023-00380-8 8. Khachadourian , V., Arildskov, E. S., Grove, J., O’Reilly, P. F., Buxbaum, J. D., Reichenberg, A., Sandin, S., Croen, L. A., Schendel, D., Hansen, S. N., & Janecka, M. (2025). Familial confounding in the associations between maternal health and autism. Nature Medicine , 31(3), 996 – 1007. https://doi.org/10.1038/s41591-024-03479-5

References 9. Hope, H., Pierce, M., Gabr, H., Radojčić, M. R., Swift, E., Taxiarchi, V. P., & Abel, K. M. (2024). The causal association between maternal depression, anxiety, and infection in pregnancy and neurodevelopmental disorders among 410 461 children: A population study using quasi-negative control cohorts and sibling analysis . Psychological Medicine , 54(8), 1693 – 1701. https://doi.org/10.1017/S0033291723003604 10. Beier, M. A., Setoguchi, S., Gerhard, T., Roy, J., Koffman, D., Mendhe, D., Madej, J., Strom, B. L., Blaser, M. J., & Horton, D. B. (2025). Early childhood antibiotics and chronic pediatric conditions: A retrospective cohort study. The Journal of Infectious Diseases , jiaf191. https://doi.org/10.1093/infdis/jiaf191 11. Schmidt, R. J., Tancredi, D. J., Ozonoff, S., Hansen, R. L., Hartiala, J., Allayee, H., Schmidt, L. C., Tassone, F., & Hertz-Picciotto, I. (2012). Maternal periconceptional folic acid intake and risk of autism spectrum disorders and developmental delay in the CHARGE (CHildhood Autism Risks from Genetics and Environment) case-control study. The American Journal of Clinical Nutrition , 96(1), 80 – 89. https://doi.org/10.3945/ajcn.110.004416 12. Schmidt, R. J., Kogan, V., Shelton, J. F., Delwiche, L., Hansen, R. L., Ozonoff, S., Ma, C. C., McCanlies, E. C., Bennett, D. H., Hertz-Picciotto, I., Tancredi, D. J., & Volk, H. E. (2017). Combined Prenatal Pesticide Exposure and Folic Acid Intake in Relation to Autism Spectrum Disorder. Environmental Health Perspectives , 125 (9), 097007. https://doi.org/10.1289/EHP604

References

13. Grether, J. K., Li, S. X., Yoshida, C. K., & Croen, L. A. (2010). Antenatal ultrasound and risk of autism spectrum disorders. Journal of Autism and Developmental Disorders , 40(2), 238 – 245. https://doi.org/10.1007/s10803-009-0859-4

14. Mollon, J., Almasy, L., Jacquemont, S., & Glahn, D. C. (2023). The contribution of copy number variants to psychiatric symptoms and cognitive ability. Molecular Psychiatry , 28(4), 1480 – 1493. https://doi.org/10.1038/s41380- 023-01978-4

15. Webb, S. J., Garrison, M. M., Bernier, R., McClintic, A. M., King, B. H., & Mourad, P. D. (2017). Severity of ASD symptoms and their correlation with the presence of copy number variations and exposure to first trimester ultrasound. Autism Research: Official Journal of the International Society for Autism Research , 10(3), 472 – 484. https://doi.org/10.1002/aur.1690 16. Mazina, V., Gerdts, J., Trinh, S., Ankenman, K., Ward, T., Dennis, M. Y., Girirajan, S., Eichler, E. E., & Bernier, R. (2015). Epigenetics of autism-related impairment: Copy number variation and maternal infection. Journal of Developmental and Behavioral Pediatrics: JDBP , 36(2), 61 – 67. https://doi.org/10.1097/DBP.0000000000000126

Autism New Jersey | The Power of Connection

800.4.AUTISM | www.autismnj.org

The Complex Causes of Autism Webinar Brain Developmental Causes

Emanuel DiCicco-Bloom, MD Neuroscience and Cell Biology/Pediatrics Rutgers Robert Wood Johnson Medical School

May 22, 2025

New Jersey Autism Center of Excellence RUCARES

Rutgers, The State University of New Jersey

Neurodevelopmental Disorders What does that mean?

- Autism Spectrum Disorder (ASD)

- Developmental Delay

- Intellectual Disability

- Learning Disability

- Attention Deficit Hyperactivity Disorder

- Specific Language Impairment

- Tourette’s Syndrome

- Schizophrenia

- Bipolar Depression

What are the symptoms of ASD?

Problems with eye contact

No response to one’s name

Problems following another person’s gaze or pointing, “joint attention”

Reduced facial expression that does not match what is being said, or the expression’s of others

Excessive lining up of toys

Abnormally intense or focused interest

Preoccupation with moving objects, or parts of objects (truck wheels!)

Repetitive hand or arm movements (flapping)

Inflexible adherence to specific routines or rituals (nighttime)

Unusual language – few spontaneous words, echolalia

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

Hallmark Feature - Abnormal Social Interaction

RT Schultz et al, 2000

How Common is ASD?

Prevalence of ASD is 1:31 nationwide (3.22%)

40.4% with intellectual disability among boys

More common in males than females (3.4:1 ratio)

Ratio varies by cause

www.cdc.gov/

Autism Spectrum Disorders (ASD)- DSM-5 Diagnostic Criteria A. Persistent deficits in social communication and social interaction across multiple contexts B. Restricted, repetitive patterns of behavior, interests, or activities C. Present in early developmental period (Recognized 12 – 24 months) D. Significant impairment in social, occupational, or other functioning E. Not better explained by intellectual disability or global delay

Specifiers:

+/- intellectual impairment +/- language impairment +/- genetic, medical, environmental factor-FXS, Tuberous Sclerosis, Rett, Epilepsy, Valproic acid (Depakene), Very Low Birth Weight

Co-Morbidity: 70% with one another disorder, and 40% with two! Anxiety & Mood(>60%), Epilepsy (30%), ID (65%), ADHD, OCD

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

Autism Spectrum Disorder has a Genetic Basis

GENES

ENVIRONMENT

Twin Studies 61-94% concordance rate among identical/monozygotic twins

10-17% concordance rate among dizygotic/fraternal twins

Family Studies Risk of developing autism is 10 times greater for siblings of autistic individuals than for general population, occurring 10-20% We consider the non-genetic factors to be in part environmental, that is the intrauterine environment: Medications (Valproic acid), maternal infection, challenging delivery, very low birth weight; may be differences in placental function, maternal stress, chemicals like pesticides

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

CEREBRAL CORTEX

Parietal Cortex sensation association

Frontal Cortex planning language motor Temporal Cortex Hippocampus Amygdala social emotion fear

Occipital Cortex vision

CEREBELLUM movement cognition

learning/memory neurogenesis

Autism : Smaller cerebellar vermis Lost Purkinje neurons

Cerebral Cortex Neurons in Culture

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

The Synapse

Genetic changes of the

synapse cause multiple

neuropsychiatric disorders

of development

Stages of Brain Growth

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

Stages in Neuron Production

Noctor & Kriegstein

Neurite

Cell body

Cell proliferation

Process/Neurite growth

Migration

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

There is No Consistent Abnormality in ASD Postmortem Brains: All of the following differences have been seen: Brain Size ASD & Abnormal Brain Development

• 20% Macrocephaly (Bigger brains) • 10% Microcephaly (Smaller brains)

Proliferation

Location of Neurons -Incorrect positioning -Disrupted layers Neuron Abnormalities -Too many or too few neurites -Synapse number

Migration

Differentiation

-Abnormalities in neurotransmitters -Electrophysiological abnormalities DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

The Synapse

Genetic changes of the

synapse cause multiple

neuropsychiatric disorders

of development

Genetic changes of the synapse cause multiple neuropsychiatric disorders of development

Fragile X Syndrome mutation leads to enhanced Glutamate signaling

FMR-KO+ mGluR5 Het

FMR-KO

mGluR5 Het

Wild Type

Mushroom

Stalk

Glutamate mGluR5 receptor antagonism reverses the FMR1 disease

FXS accounts for ~1% of ASD

40% of those with FXS have ASD

G Dolen, M Bear, 2007

Signs of Tuberous Sclerosis – Neurocutaneous Disorder- Epiderm

Angiofibroma “bumps”

Hypopigmented patches

Tubers

NT

ASD

Nodules

Tang et al, http://dx.doi.org/10.1016/j.neuron.2014.07.040

Peters et al, Future Neurol. 2013 8(5):583-597

During development from the fetus to adulthood, neuron and synapse numbers are in part regulated by pruning!

Microglia are the ‘Gardeners’ of the Brain!

Penna et al, Cerebral Cortex Communications, 2021, 2, 1 – 14

Functional MRI studies in typical and autistic individuals

Attention task

Typical

Autism

Motor task

Autism

Typical

Allen and Courchesne, 2003

Axon Bundles = White Matter tracts Neurites → Axons and Dendrites What Does it All Mean?

Shu et al 2001 Alexander et al 2007

What about the connections between neurons and brain regions?

We can use Functional MRI (fMRI) to measure connectivity

Local hyperconnectivity?

Long distance hypoconnectivity?

Savant abilities?

Delays in social and communication functions?

Frontal

Occipital

Due et al, DOI 10.3389/fnagi.2023.1101879

Recent Advances in ASD Diagnosis

Early structural and functional brain imaging accurately predicts ASD diagnosis at 24 months! Nature. 2017 Feb 15;542(7641):348-351 Structural brain MRI at 6 (Blue) and 12 (Red) months accurately predicts (81%) which children will be diagnosed with ASD at 24 mo, especially social deficits. Early expansion of the cerebral cortex. In families with one child with autism, the next born sibling has a greater chance of autism – Established the Baby Siblings Study = higher risk of ASD

Functional connectivity MRI at 6 months accurately predicts which children will be diagnosed with ASD at 24 months. Sensitivity, 82%; Specificity, 100%. Red (-), Blue (+) connectivity

Sci Transl Med. 2017 Jun 7;9(393). pii: eaag2882.

THERE ARE MANY DIFFERENT GENETIC CAUSES OF ASD

(Autism Science Foundation)

Satterstrom et al, 102 Genes - Categories

Hypo- and Hyper-connectivity can be defined in mouse genetic models – Why Mice?

Hypo-connectivity in BLUE

Does hypoconnectivity cause delays in social and motor behaviors?

Hyper-connectivity in RED

Pagani et al, https://doi.org/10.1101/2025.03.04.641400

This gene-connectivity relationship is now being applied to people

Social behavior in mice – preference for another mouse Engrailed-2 mutants do not prefer another mouse!

The Role of Oxytocin in Social Behavior Select Neuronal Circuits?

Social Behavior in Voles - Monogamous vs Non-monogamous

The neuropeptide oxytocin (OT) regulates social behavior, including pair bonding and maternal-infant relations. OT acts via OT-Receptor and Arg-Vasopressin 1a-R.

Monogamous Prairie Vole

Non-Monogamous Montane Vole

Highly social Communal burrows Biparental child care 80% mate for life Separation-ultrasonic distress Not aggressive to intruders Isolated burrows No paternal child care Mate with multiple partners Separation-little distress Human clinical trials? Failed. But clinical subgroups? Yes DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

Our Approach

16p11.2 Deletion CNV

NJLAGS idiopathic ASD dataset

Deletes 28 genes

Genetically undefined

Goal: Use iPSC-derived neuronal precursor cells (NPCs) to study the developmental and signaling pathways that are abnormal in these individuals

DiCicco-Bloom, Rutgers Robert Wood Johnson Medical School

Connacher R et al., Stem Cell Reports. 2022 May 17;S2213-6711(22)00208-9

Stages in Neuron Production

Noctor & Kriegstein

Neurite

Cell body

Cell proliferation

Process/Neurite growth

Migration

Family-1: Different rates of proliferation

DAY 2

DAY 4

DAY 6

Connacher R et al., Stem Cell Reports. 2022 May 17;S2213-6711(22)00208-9

Idiopathic ASD Neuronal Processes

Smrithi Prem

Prem S et al., eLife, 2024;13:e82809

Idiopathic ASD Neuronal Migration

Smrithi Prem

Prem S et al., eLife, 2024;13:e82809

Autism genetic and environmental factors act during gestation and affect neural precursor cell proliferation, migration, and process outgrowth

The gut microbiome and brain have heightened sensitivity to environmental factors during early life Do Genes interact with Environmental factors to impact brain development?

CDC estimates that an infant will receive 2-3 courses of antibiotics Susceptible to infections

Immune Vagus nerve Endocrine Metabolites

Aversa et al., (2021) Slykerman et al., (2019) Lavebratt et al., (2019) Wimberley et al., (2018)

Postnatal antibiotic exposure

Antibiotic-induced perturbations

Courtney R. McDermott, PhD Columbia University

Aversa et al., (2021) Slykerman et al., (2019) Lavebratt et al., (2019) Wimberley et al., (2018)

Chung et al., (2021) Niarchou et al., (2019) Duyzend & Eichler et al., (2015) Blumenthal et al., (2014)

Postnatal antibiotic exposure

16p11.2 microdeletion copy number variation

Association with NDDs

Warrants investigation of underlying biological mechanisms

Accounts for ~1% ASD cases

Early life treatment with antibiotic altered the gut microbiome, brain development, and behavior, and these effects were magnified in mice that were carrying the 16p11.2 CNV

McDermott et al., bioRxiv: https://doi.org/10.1101/2025.02.25.639888

Acknowledgments:

Robert Wood Johnson Medical School

Rutgers University

DiCicco-Bloom lab Smrithi Prem Robert Connacher Madeline Williams Xiaofeng Zhou Courtney McDermott Cynthia Peng Bharati Dev Anna Markov

Linda M. Brzustowicz Judy Flax

Jinchuan Xing Rohan Alibutud

Child Health Institute Che Wei Lu

Percy L Yeung Zhiping Pang

Center for Advanced Biotechnology and Medicine

Nancy Lurie Marks Family Foundation NIMH

James H. Millonig Monal Mehta Paul Matteson

Lurie Family Foundation Autism Research Institute Simons Foundation RUCDR New Jersey Governor ’ s Council for Autism Research Mindworks Charitable Lead Trust Jewish Community Foundation of Greater MetroWest NJ The Families!

Questions? Call 800.4.AUTISM or visit www.autismnj.org

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