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

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

Meeting: ACR Convergence 2023

Keywords: gender, neuropsychiatric disorders, pregnancy, psychosocial factors, Systemic sclerosis

Session Information

Date: Sunday, November 12, 2023

Title: (0460–0479) Reproductive Issues in Rheumatic Disorders Poster I

Session Type: Poster Session A

Session Time: 9:00AM-11:00AM

Background/Purpose: The long-term neuropsychiatric (NP) outcome of children born to mothers affected by autoimmune diseases (AIDs) represents a controversial topic, with few studies^1-3 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

Disclosures: 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.

To cite this abstract in AMA style:

Lazzaroni M, Pedretti E, Moschetti L, Bendoni M, Visconti L, Molinaro A, Nalli C, Franceschini F, Airò P, Galli J, Fazzi E, Tincani A, Andreoli L. 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 [abstract]. Arthritis Rheumatol. 2023; 75 (suppl 9). https://acrabstracts.org/abstract/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-chi/. Accessed .

« Back to ACR Convergence 2023

ACR Meeting Abstracts - https://acrabstracts.org/abstract/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-chi/