Assessment & Research

Incontinence and psychological symptoms in individuals with Mowat-Wilson Syndrome.

Niemczyk et al. (2017) · Research in developmental disabilities 2017
★ The Verdict

Nearly every person with Mowat-Wilson Syndrome remains incontinent into adulthood and half show clinical-level mood or anxiety signs—so always screen toileting and mental health.

✓ Read this if BCBAs working with teens or adults who have rare genetic syndromes.
✗ Skip if Clinicians serving only typically developing clients with no incontinence issues.

01Research in Context

01

What this study did

Justine et al. (2017) sent a survey to families of people with Mowat-Wilson Syndrome. They asked about toileting and mood, behavior, and anxiety problems. Forty-seven surveys came back, covering both kids and adults.

02

What they found

Almost every person—97.7%—still had incontinence past childhood. One quarter to nearly half scored above the worry line for psychological problems, no matter the age group. In short, toileting needs and mental-health risks stay high for life.

03

How this fits with other research

Doughty et al. (2002) looked at adult women with Rett syndrome and found fewer behavior problems than other adults with ID. Justine’s group saw more mood and anxiety flags in Mowat-Wilson adults. The picture flips: one syndrome shows lower irritability, the other shows higher psychological risk.

Emerson (2011) warned that adults with mild ID who skip specialist care carry hidden health risks. Justine’s data add a new hidden risk—lifelong incontinence—in a different genetic group. Together the papers say, "Screen everyone; problems you don’t see still hurt."

Johansson et al. (2010) showed autism tools miss kids with sensory and motor issues. People with Mowat-Wilson also have multiple needs at once. Both studies push clinicians to look past single labels and check toileting, mood, and development together.

04

Why it matters

If you serve adults with rare genetic diagnoses, add two quick boxes to your intake: "Still incontinent?" and "Mood/anxiety check done?" Catching these lifelong needs early saves dignity, hours of problem behavior, and caregiver stress.

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→ Action — try this Monday

Add a two-item caregiver questionnaire: "Daytime accidents? Night-time accidents? New worries or mood changes?"—then plan toileting or counseling referrals as needed.

02At a glance

Intervention
not applicable
Design
survey
Sample size
47
Population
other
Finding
not reported

03Original abstract

BACKGROUND: Mowat-Wilson Syndrome (MWS) is caused by deletion/mutation of the ZEB2 gene on chromosome 2q22. MWS is characterized by a distinctive facial appearance, severe intellectual disability and other anomalies, e.g. seizures and/or Hirschsprung disease (HSCR). Most individuals have a sociable demeanor, but one third show psychological problems. AIMS: The aim was to investigate incontinence and psychological problems in MWS. METHODS AND PROCEDURES: 26 children (4-12 years), 13 teens (13-17 years) and 8 adults (>18years) were recruited through a MWS support group. The Parental Questionnaire: Enuresis/Urinary Incontinence, as well as the Developmental Behaviour Checklist (DBC) were completed by parents or care-givers. OUTCOMES AND RESULTS: 97.7% of persons with MWS had incontinence (nocturnal enuresis 74.4%; daytime urinary incontinence 76.2%; fecal incontinence 81.4%). Incontinence remained high over age groups (children 95.8%, teens 100%, adults 100%). 46.2% of children, 25% of teens and 37.5% of adults exceeded the clinical cut-off on the DBC. The ability to use the toilet for micturition improved with age. CONCLUSIONS AND IMPLICATIONS: MWS incontinence rates are very high. All had physical disabilities including anomalies of the genitourinary and gastrointestinal tract. Due to the high prevalence rates, a screening for incontinence and psychological problems in MWS is recommended.

Research in developmental disabilities, 2017 · doi:10.1016/j.ridd.2017.01.006