Table 2 Statistical analysis results

1

Number of USVs

rmANOVA

Genotype

F(1,42) = 5.669

.022

Sex

F(1,40) = 3.352

.075

Age

F(2,84) = 1.894

.157

Genotype* Age

F(2,84) = 0.863

.863

Mann–Whitney

Genotype

U(132) = 1373

.0011

Weight

rmANOVA

Genotype

F(1,42) = .016

.900

Sex

F(1,40) = .001

.977

Age

F(1.5,61) = 871.279

3.0E-44

Genotype* Age

F(1.5,61) = .347

.640

Latency to right

Mann–Whitney

Genotype

U(44) = 289.5

.148

Sex

U(44) = 249.5

.629

Temperature

rmANOVA

Genotype

F(1,42) = 5.706

.021

Sex

F(1,40) = .386

.538

Age

F(2,84) = 7.969

.001

Genotype* Age

F(2,84) = .411

.664

Number of USVs with temperature covariate

ANCOVA

Genotype

F(1,125) = 11.021

.001

Age

F(2,125) = .911

.405

Genotype* Age

F(2,125) = .111

.895

Number of phrases

Linear mixed model

Genotype

F(1,42.3) = 4.264

.045

Sex

F(1,39.9) = 2.935

.094

Age

F(2,52.6) = .686

.508

Genotype*Age

F(2,52.6) = 1.265

.291

Call duration

Linear mixed model

Genotype

F(1,28) = 11.198

.002

Sex

F(1,24.6) = 2.055

.164

Age

F(2,41.3) = 3.679

.034

Genotype*Age

F(2,41.3) = .282

.756

Pause duration

Mann–Whitney

Genotype

U(102) = 892

.048

Sound pressure level

Mann–Whitney

Genotype

U(102) = 972

.159

Mean frequency

Linear mixed model w/ simple main effects

Genotype

F(1,77.1) = 8.973

.004

Sex

F(1,68.7) = .156

.694

Age

F(2,96) = 4.364

.015

Genotype*Age

F(2,96) = 6.274

.003

P6, Genotype

F(1,96) = 20.942

.00001

Frequency range

Linear mixed model

Genotype

F(1,66.7) = 9.557

.003

Sex

F(1,76) = 3.623

.061

Age

F(1,96) = 5.084

.008

Genotype*Age

F(1,96) = .280

.757

% calls with frequency jump

Mann–Whitney

Genotype

U(127) = 1348.5

.006

2

Developmental data collection attempts

rmANOVA

Genotype

F(1,42) = .144

.706

Sex

F(1,42) = .144

.706

Age

F(1.5,68) = 2.545

.096

Genotype*Age

F(1.5,68) = .992

.361

Developmental body weight

rmANOVA

Genotype

F(1,42) = .007

.932

Sex

F(1,42) = 12.837

.0009

Age

F(1.9,80.3) = 1294.4

1.9E-94

Genotype*Age

F(1.9,80.3) = 3.599

.034^

Developmental body length

rmANOVA

Genotype

F(1,42) = .504

.481

Sex

F(1,42) = 16.008

.0003

Age

F(3,126) = 89.130

5.3E-31

Genotype*Age

F(3,126) = .214

.887

Developmental forelimb brake duration

Hierarchical mixed covariate model

Genotype

F(1,71.4) = 18.499

.00005 (.002*)

Sex

F(1,82.9) = .785

.378

Age

F(3,127.9) = .731

.536

Genotype*Age

F(3,130.6) = 2.734

.046 (.510*)

Developmental hindlimb absolute paw angle

Hierarchical mixed covariate model

Genotype

F(1,65.1) = 10.979

.002 (.033*)

Sex

F(1,79.5) = 2.581

.112

Age

F(3,124.8) = 3.604

.015 (.045*)

Genotype*Age

F(3,128.6) = 1.384

.251

Adult body length

ANOVA

Genotype

F(1,40) = 8.278

.006

Sex

F(1,40) = 18.846

.00009

Genotype*Sex

F(1,40) = .107

.745

Adult body weight

ANOVA

Genotype

F(1,40) = 10.809

.002

Sex

F(1,40) = 59.719

3.5E-9

Genotype*Sex

F(1,40) = .001

.972

30 cm/s data collection attempts

ANOVA

Genotype

F(1,42) = 2.261

.140

Sex

F(1,40) = 1.222

.276

Genotype*Sex

F(1,40) = .629

.433

40 cm/s data collection attempts

ANOVA

Genotype

F(1,36) = 6.746

.014

Sex

F(1,34) = .089

.767

Genotype*Sex

F(1,34) = .831

.368

Adult 30 cm/s forelimb brake duration

ANCOVA

Genotype

F(1,41) = 17.812

.0001 (.0006*)

Sex

F(1,39) = 2.067

.308

Genotype*Sex

F(1,39) = 1.473

.232

Adult 40 cm/s hindlimb % shared stance

ANCOVA

Genotype

F(1,35) = 12.092

.001 (.06*)

Sex

F(1,33) = .114

.738

Genotype*Sex

F(1,33) = .042

.838

Adult 40 cm/s hindlimb % swing

ANCOVA

Genotype

F(1,35) = 8.555

.006 (.088*)

Sex

F(1,33) = .814

.374

Genotype*Sex

F(1,33) = .030

.863

Adult 40 cm/s hindlimb % stance

ANCOVA

Genotype

F(1,35) = 8.555

.006 (.088*)

Sex

F(1,33) = .814

.374

Genotype*Sex

F(1,33) = .030

.863

3

Body weight

rmANOVA w/

simple main effects

Genotype

F(1,35) = 16.179

.0003

Sex

F(1,35) = 93.839

1.9E-11

Genotype*Sex

F(1,35) = .139

.712

Females, Genotype

F(1,35) = 6.484

.015

Males, Genotype

F(1,35) = 9.927

.003

Age

F(2,72.9) = 221.81

3.7E-32

Genotype*Age

F(2,72.9) = 9.667

.0002

Mean visual acuity

ANOVA

Genotype

F(1,37) = .573

.454

Sex

F(1,35) = 2.732

.107

Genotype*Sex

F(1,35) = .436

.513

Mean contrast

ANOVA

Genotype

F(1,37) = .003

.960

Sex

F(1,35) = 2.409

.130

Genotype*Sex

F(1,35) = 1.034

.316

4

Body weight during Acoustic Startle/PPI testing

ANOVA

Genotype

F(1,36) = 6.187

.018

Sex

F(1,36) = 27.528

7.1E-6

Genotype*Sex

F(1,36) = .136

.714

Startle magnitude – 120 dB

rmANCOVA

Genotype

F(1,37) = 4.282

.046

Sex

F(1,35) = .823

.371

Trial

F(3,111) = .338

.798

Genotype*Trial

F(3,111) = 1.216

.307

Startle magnitude – 80-120 dB

rmANCOVA

Genotype

F(1,37) = .921

.343

Sex

F(1,35) = .042

.839

dB

F(3.8,143.8) = 3.208

.016

Genotype*dB

F(3.8,143.8) = .192

.939

% inhibition of startle

rmANCOVA w/ simple main effects

Genotype

F(1,37) = 5.151

.029

Sex

F(1,35) = 2.035

.163

dB

F(2,74) = .046

.955

Genotype*dB

F(2,74) = 1.033

.361

4 dB, Genotype

F(1,113) = .712

.401

8 dB, Genotype

F(1,113) = 4.221

.042

16 dB, Genotype

F(1,113) = 8.444

.004

% freezing: tone + shock paring

rmANOVA

Genotype

F(1,35) = .027

.872

Sex

F(1,35) = 9.960

.003

Minute

F(2,70) = 10.981

.00007

Genotype*Minute

F(2,70) = .053

.949

% freezing: contextual fear

rmANOVA

Genotype

F(1,35) = .592

.447

Sex

F(1,35) = 5.635

.023

Minute

F(7,245) = 2.506

.017

Genotype*Minute

F(7,245) = 1.049

.397

% freezing: cued fear

rmANOVA

Genotype

F(1,35) = 4.210

.048

Sex

F(1,35) = 2.129

.153

Minute

F(7,245) = 18.164

2.1E-19

Genotype*Minute

F(7,245) = .670

.697

5

SA distance traveled

rmANOVA

Genotype

F(1,36) = .943

.338

Sex

F(1,36) = 1.674

.204

Genotype*Sex

F(1,36) = .051

.823

SA habituation investigation time

rmANOVA

Genotype

F(1,36) = .538

.468

Stimulus

F(1,36) = .645

.427

Sex

F(1,36) = 6.231

.017

Genotype*Stimulus

F(1,36) = .004

.950

Genotype*Sex

F(1,36) = .018

.893

SA sociability preference index

ANOVA

Genotype

F(1,36) = .886

.353

Sex

F(1,36) = 5.800

.021

Genotype*Sex

F(1,36) = 2.992

.092

SA sociability investigation time

rmANOVA w/

simple main effects

Genotype

F(1,36) = .233

.632

Stimulus

F(1,36) = 140.790

5.3E-14

Sex

F(1,36) = 12.039

.001

Genotype*Sex

F(1,36) = .059

.809

Genotype*Stimulus

F(1,36) = .429

.517

Controls, stimulus

F(1,36) = 78.384

1.4E-10

MPS IIIB, stimulus

F(1,36) = 62.836

2.1E-9

SA sociability preference index across time

rmANOVA

Genotype

F(1,36) = .143

.708

Genotype*Minute

F(8.9,322.9) = 1.867

.056

SA novelty preference index

ANOVA

Genotype

F(1,36) = .000

.991

Sex

F(1,36) = .094

.761

Genotype*Sex

F(1,36) = .301

.586

SA social novelty investigation time

rmANOVA w/

simple main effects

Genotype

F(1,36) = 2.784

.104

Stimulus

F(1,36) = 96.732

9.7E-12

Sex

F(1,36) = 12.258

.001

Genotype*Sex

F(1,36) = 2.390

.131

Genotype*Stimulus

F(1,36) = 1.479

.232

Controls, stimulus

F(1,36) = 37.144

5.2E-7

MPS IIIB, stimulus

F(1,36) = 61.067

2.9E-9

SA novelty preference index across time

rmANOVA

Genotype

F(1,36) = .058

.811

Genotype*Minute

F(9,324) = 1.651

.100

Tube Test percent wins

One-sample t-test (to 50%)

Female MPS IIIB

t(8) = -3.900

.005

Male MPS IIIB

t(9) = -.919

.382

RI bout with an attack

Fisher’s Exact Test

Genotype

OR = 1.25 [95% CI 1.045,1.495]

.024

RI anogenital sniff duration

Mann–Whitney

Genotype

U(20) = 22

.035

RI head sniff count

Mann–Whitney

Genotype

U(22) = 20

.023

6

Whole brain volume

ANOVA

Genotype

F(1,22) = 28.585

.00002

Sex

F(1,22) = 4.137

.054

Genotype*Sex

F(1,22) = .185

.671

Body weight

ANOVA

Genotype

F(1,22) = 61.441

8.3E-8

Sex

F(1,22) = 82.188

7.0E-9

Genotype*Sex

F(1,22) = 3.582

.072

Cerebellar volumetric ratio

ANOVA

Genotype

F(1,22) = 5.693

.026

Sex

F(1,22) = 2.131

.158

Genotype*Sex

F(1,22) = 1.144

.296

Cerebellar apparent diffusion coefficient

ANOVA

Genotype

F(1,22) = 2.883

.104

Sex

F(1,22) = .081

.779

Genotype*Sex

F(1,22) = .782

.386

Corpus callosal volumetric ratio

ANOVA

Genotype

F(1,22) = 10.219

.004

Sex

F(1,22) = .016

.900

Genotype*Sex

F(1,22) = .926

.346

Corpus callosal fractional anisotropy

ANOVA

Genotype

F(1,22) = 1.394

.250

Sex

F(1,22) = .010

.923

Genotype*Sex

F(1,22) = 3.241

.086

Cortical thickness

ANOVA

Genotype

F(1,22) = 1.399

.250

Sex

F(1,22) = .113

.740

Genotype*Sex

F(1,22) = .110

.743