Session: (0460–0479) Reproductive Issues in Rheumatic Disorders Poster I
0478: The Long-term Neurodevelopmental Outcome of Children Born to Women with Systemic Sclerosis: Assessment Through a Self-reported Questionnaire by the Mothers and Nneuropsychiatric Evaluations of the Children
ASST Spedali Civili di Brescia Brescia, Brescia, Italy
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Maria Grazia Lazzaroni1, Eleonora Pedretti1, Liala Moschetti1, Marzia Bendoni2, Lucrezia Visconti2, Anna Molinaro2, Cecilia Nalli3, Franco Franceschini3, Paolo Airò1, Jessica Galli2, Elisa Fazzi2, Angela Tincani4 and Laura Andreoli5, 1Rheumatology and Clinical Immunology, ASST Spedali Civili of Brescia, University of Brescia, Brescia, Italy, 2Unit of Child and Adolescent Neuropsychiatry, ASST Spedali Civili of Brescia, University of Brescia, Brescia, Italy, 3Rheumatology and Clinical Immunology, ASST Spedali Civili of Brescia, Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy, 4Rheumatology and Clinical Immunology, ASST Spedali Civili of Brescia, University of Brescia, Gussago, Italy, 5University of Brescia, Brescia, Italy
Background/Purpose: The long-term neuropsychiatric (NP) outcome of children born to mothers affected by autoimmune diseases (AIDs) represents a controversial topic, with few studies1-3 reporting a possible increased prevalence of NP alterations. We focused on the neurodevelopment (ND) of children born to Systemic Sclerosis (SSc) mothers, starting with the creation of an ad-hoc questionnaire regarding different aspects of children’s ND administered to consecutive SSc mothers in 20214. We further investigated the long-term NP outcome of SSc offspring by offering a comprehensive pediatric NP evaluation.
Methods: SSc mothers (ACR/EULAR 2013 criteria) who previously completed the questionnaire4 were allowed to have their children aged < = 18 years undergo a clinical NP evaluation including:
(1) cognitive/adaptive functioning (Griffiths Mental Development Scales -GMDS-III-; Wechsler Scale for corrected age -WISC-III; Vineland Adaptive Behavior Scales -VABS-II-) (2) behavioral/social problems (Child Behavior CheckList -CBCL-, fulfilled by the mothers; Youth Self Report -YSR-, for children ³ >=11 years) (3) anxiety/depression/somatic disorders (Self-administered psychiatric scales for children/adolescents -SAFA-A/D/S- ³ >=8 years) (4) sleep disorders.
Cohen’s kappa coefficient was used to evaluate the agreement between questionnaire answers and NP test results.
Results: 23 SSc mothers reported 39 children aged < = 18 years, 37/39 were proposed for NP evaluation and 20 agreed to be evaluated (F/M 1:1; median age 8 [6-11] years). The mothers had reported at least one alteration in the questionnaire for 7 children (Table 1); one already had a diagnosis of autism spectrum disorder (ASD). At NP evaluations were recorded:
(1) normal scores for cognitive/adaptive functioning in all children, except the one with ASD (2) an increased risk of developing behavioral/social problems especially in extra-academic/social activities in 3/20 at CBCL and in 3/6 at YSR (3) an increased risk of developing depression in 4/11 (4) sleep disorders in 9/20
Features of mothers and their offspring are shown in Table 1. The agreement between questionnaires and NP evaluation was moderate for cognitive/adaptive functioning and sleep disorders (k 0.5 and 0.6 respectively) and weak for behavioural/social problems (k 0.2).
Conclusion: At NP evaluation, children born to SSc mothers showed normal cognitive skills but a tendency toward an impairment in the behavioral/social area. Mothers seldom recognized such a difficulty, according to the self-reported questionnaire.
In the setting of preconception counseling, it is important to inform SSc mothers about possible NP symptoms in their children and to seek for a specialist evaluation in case of doubts. Indeed, with the help of specialists, most NP problems can be overcome if detected early.
References.1Vinet E. et al. Arthritis Rheumatol.2015; 2Wojcik S. et al. Arthritis Care Re.2017; 3Nalli C. et al. Best Pract Res Clin Obstet Gynaecol.2020; 4Pedretti E. et al. [abstract]Arthritis Rheumatol.2022
The Authors would like to thank the Italian association of SSc patients ‘GILS’ (Gruppo Italiano Lotta Sclerodermia) for kindly supporting the project.
Table 1: Features of SSc mothers and their offspring. For each child different NP domains were evaluated at questionnaire and NP evaluations
M. Lazzaroni: None; E. Pedretti: None; L. Moschetti: None; M. Bendoni: None; L. Visconti: None; A. Molinaro: None; C. Nalli: None; F. Franceschini: None; P. Airò: None; J. Galli: None; E. Fazzi: None; A. Tincani: Galapagos, 6, GSK, 6, UCB, 6; L. Andreoli: None.