Pos1299 long-term neurodevelopmental outcome of children born to systemic sclerosis women: pediatric neuropsychiatric assessment through a set of validated tools

Background The neurodevelopmental (ND) outcome of children born to women with autoimmune disorders might be affected by factors related to maternal disease ( e.g. the transplacental passage of maternal antibodies/drugs), as well as by mothers’ coping with their disabilities during growth. These aspects have been studied in rheumatoid arthritis (RA) and systemic lupus erythematosus (SLE) [1] ; no data are available for Systemic Sclerosis (SSc). Objectives To evaluate the long-term ND outcome of children born to SSc mothers. Methods A pediatric neuropsychiatric evaluation was proposed to SSc mothers with at least one child ≤18 years (August 2021-June 2022). Evaluations, performed by a child neuropsychiatrist, included the assessment of the following domains: Developmental/Cognitive skill (Griffiths Mental Development Scales III -GMDS-III- and Wechsler Scale for corrected age) Adaptive behavior (Vineland Adaptive Behavior Scales-II -VABS-II-) Social/Behavioral problems (Child Behavior CheckList and Youth Self Report -YSR- for children ≥11 years) Quality of life (Pediatric Quality of Life Inventory 4.0 -PedsQL-) A set of questions to investigate the child’s ND milestones were submitted to mothers. Data about maternal disease were retrieved from medical records. Results 20 children (median age 8 years; F/M 1:1) born to 12 SSc mothers were evaluated: 4 were born before and 16 after SSc onset. Four preterm births (<37 gestational weeks -GW-) (one twin pregnancy) and no small for gestational age babies were recorded. One child already had a diagnosis of autism spectrum disorder (ASD) at enrollment. All the children showed a normal score for developmental or intelligence quotient (IQ) and in all VABS-II domains, except for the child affected by ASD. YSR, administered to 6 children, highlighted an impairment in social abilities ( e.g. extra-academic/social activities). In CBCL, externalizing problems ( e.g. aggressive behavior, hyperactivity) were reported in 3 children (15%), while internalizing problems ( e.g. anxiety, somatic complaints) in 2 (10%). The lowest scores at PedsQL were observed in the emotional domain. Different types of sleep disorders were reported in 45% of children. No significant differences were found in maternal characteristics upon the comparison between children with/without ND abnormalities. Children and mothers’ features are shown in Table 1. Conclusion Children born to SSc mothers displayed normal intellectual functioning. The reported impairment in social abilities, suggesting an “adult profile”, possibly reflect the effect of coping with maternal disabilities. Our data did not show a specific pattern of ND alterations in the offspring of SSc mothers; however the inclusion of a child neuropsychiatrist in the multidisciplinary team dedicated to SSc patients and their families seems to be important to detect and correct any possible ND problems at an early stage. Reference [1]Nalli et al. Best Pract Res Clin Obstet Gynaecol.2020 Table 1. Characteristics of SSc women and their offspring Mother Antibodies Cutaneous involvement Digital Ulcers ILD Child GW at birth, pregnancy complications Cognitive domain Adaptive behavior Social and behavioral domain Sleep disorders 1 Scl-70 lcSSc 1 0 M, 12 yr 40 N N A 0 M, 10 yr 40 N N N 0 2 ANA+ lcSSc 0 0 M, 7 yr* 40 A A A 1 F, 5 yr 40 N N N 1 3 Th/To lcSSc 1 0 M, 13 yr 39 N N A 0 F, 9 yr 39 N N N 1 4 Scl-70 dcSSc 1 1 • F, 11 yr 39 N N A 1 M, 5yr 39 N N N 0 5 Scl-70 dcSSc 0 1 • F, 11 yr 39 N N A 1 M, 2 yr 35° N N N 1 6 ACA lcSSc 0 0 M, 3 yr 41 N N N 1 7 Scl-70 lcSSc 0 0 • F, 17 yr 32 ^ N N B 0 • F, 13 yr 39 N N N 0 M, 9 yr 39 N N N 0 8 ACA lcSSc 0 0 F, 1 yr 39 N N N 0 9 Scl-70 dcSSC 1 1 F, 8yr $ 31° N N N 1 M, 8yr $ 31° N N N 0 10 U1-RNP lcSSc 0 0 F, 5 yr 38 N N N 0 11 Scl-70 dcSSc 0 0 M, 3yr 39 N N N 1 12 U1-RNP lcSSc 1 0 F, 1 yr 40 N N N 0 ANA: anti-nuclear antibodies, ACA: anti-centromere antibodies, GW: gestational weeks; lcSSc: limited cutaneous SSc, dcSSc: diffuse cutaneous SSc, ILD: interstitial lung disease, 0: absent, 1: present, N: normal, B: borderline, A: abnormal *ASD diagnosis, • born before SSc onset $ Twins, °Gestational diabetes, ^ Preeclampsia/IUGR Acknowledgements he Authors would like to thank the Italian association of SSc patients ‘GILS’ (Gruppo Italiano Lotta Sclerodermia) for kindly supporting the project. Disclosure of Interests None Declared.