Sertraline May Improve Language Developmental Trajectory in Young Children with Fragile X Syndrome: A Retrospective Chart Review

Abstract

Young children with fragile X syndrome (FXS) often experience anxiety, irritability, and hyperactivity related to sensory hyperarousal. However, there are no medication recommendations with documented efficacy for children under 5 years old of age with FXS. We examined data through a chart review for 45 children with FXS, 12–50 months old, using the Mullen Scales of Early Learning (MSEL) for baseline and longitudinal assessments. All children had clinical level of anxiety, language delays based on MSEL scores, and similar early learning composite (ELC) scores at their first visit to our clinic. Incidence of autism spectrum disorder (ASD) was similar in both groups. There were 11 children who were treated with sertraline, and these patients were retrospectively compared to 34 children who were not treated with sertraline by chart review. The baseline assessments were done at ages ranging from 18 to 44 months (mean 26.9, SD 7.99) and from 12 to 50 months (mean 29.94, SD 8.64) for treated and not treated groups, respectively. Mean rate of improvement in both expressive and receptive language development was significantly higher in the group who was treated with sertraline (P < 0.0001 and P = 0.0071, resp.). This data supports the need for a controlled trial of sertraline treatment in young children with FXS.

Serotonin dysregulation in Fragile X Syndrome: implications for treatment

“…Similarly, the consequences of FMRP expression depend on age (14). This is consistent with evidence from a retrospective chart review done by Winarni et al (65), which found that one to four year old children with FXS who received sertraline showed improved receptive and expressive language outcomes. It is critical that this therapeutic opportunity is further investigated with controlled trials, as it could lead to significant improvements in symptoms, cognition, and quality of life for patients with FXS.…”

“…Clinical results and theoretical knowledge support the usefulness of SSRIs in treating patients with FXS (65). Sertraline may be relatively more effective than other SSRIs for this patient population.…”

“…Anxiety is a classic feature in FXS throughout life and particularly in childhood (4,5). Recent evidence shows that SSRI treatment may confer non-classical benefits to patients with FXS as well (65). A core symptom in this patient population is difficulty in language acquisition and communication (1).…”

The neurobiology of the Prader-Willi phenotype of fragile X syndrome

“…Such intervention is also recommended in young children with FXS both with and without autism or the PWP (29,30). A variety of medication use can be helpful for those with FXS both with and without the PWP including; stimulants which can help with attention and appetite; Selective Serotonin Reuptake Inhibitors (SSRIs), such as sertraline for anxiety (31); and atypical antipsychotics, such as aripiprazole to stabilize mood, improve autism, aggression and/or tantrums (6). However, aripiprazole can cause an increase in weight gain and particularly for those with a CYP2D6 polymorphism that can slow down the metabolism of aripiprazole (32).…”

Assessment of efficacy of prenatal genetic diagnosis for fragile X syndrome using nested PCR

“…Genetic diagnosis shows that FXS is an X-linked genetic disorder that is characterized by behavioral problems and specific physical dysmorphisms (2). Child patients with FXS have ritualistic behaviors and social deficits and study has provided for early recognition and diagnosis for the children patients (3).…”