A Systematic Review of the Psychosocial Adjustment of Children and Adolescents with Facial Palsy: The Impact of Moebius Syndrome
Abstract
:1. Introduction
Aims and Objectives
2. Methods
2.1. Protocol and Registration
2.2. Eligibility Criteria
2.2.1. Types of Studies
2.2.2. Types of Participants
2.2.3. Types of Intervention
2.2.4. Types of Outcome Measure
2.3. Information Sources
2.4. Search
2.5. Study Selection
2.6. Data Collection Process
2.7. Data Items
2.8. Quality Assessment
3. Results
3.1. Study Selection
3.2. Study Characteristics
3.3. Risk of Bias
3.4. Behaviour
3.5. Social Difficulties
3.6. Quality of Life
3.7. Anxiety
3.8. Depression
3.9. Personality
4. Discussion
4.1. Clinical Implications
4.2. Limitations and Future Directions
5. Conclusions
Author Contributions
Funding
Conflicts of Interest
References
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Subheading | Search Terms |
---|---|
Population | Facial palsy-facial paralys *, facial pals *, facial pares *, hemi-facial paralys *, hemi-facial pals *, hemi-facial pares *, Bell * Pals *, Ramsay Hunt, Mo?bius |
Outcome | Psychosocial- psychology, psychiatry, psych *, mental disorders, anxiety, anxious, depress *, distress *, mood, emotion *, confidence, self-concept, self-perception, self-esteem, self-image, self-worth, body image, appearance |
Outcome Measure | Construct Measured | Subscales | Parent-or Self-Report |
---|---|---|---|
AFS | Anxiety | Test Anxiety Manifest Anxiety Dislike of School Social Desirability | Self-report |
CBCL | Behaviour | Social Withdrawal Somatic Complaints Anxiety/Depression Social Problems Thought Problems Attention Problems Delinquent Behaviour Aggressive Behaviour | Parent-report |
DIKJ | Depression | N/A | Self-report |
KINDL | Quality of Life | Physical Wellbeing Emotional Wellbeing Self-Esteem Family Friends School | Parent- and self-report |
PFK 9–14 | Personality | General Anxiety Belief in Own Opinions Decisions and Plans Self-Perception of Impulsivity Tendency to Overestimate One’s own Capabilities Feeling of Inferiority | Self-report |
SDQ-Deu | Psychosocial Adjustment | Emotional Problems Conduct Problems Hyperactivity/Inattention Peer Problems Prosocial Behaviour | Parent-and self-report |
Authors | Country | Sample Size n (n Female) | Age in Years M (SD) | Diagnosis | Comorbidities | Design and Setting | Measures | Main Finding(s) |
---|---|---|---|---|---|---|---|---|
Briegel, Hofmann, and Schwab (2007) | Germany | 13 (6) | 3.83 (Range = 2.08– 5.92) |
Moebius syndrome:
|
| Cross-sectional; recruitment from the German Moebius Foundation | CBCL (parent-report) | 16.7% (n = 2) scored in clinical range on at least one scale Boys scored higher (more problems) than girls on: Aggressive Behaviour, Total Problems, Oppositional Defiant Problems and Anxiety Problems |
Briegel, Hofmann, and Schwab (2010) | Germany | 31 (19) | Median = 10.58 (Range = 4.58 – 17.00) |
Moebius syndrome:
|
| Cross-sectional; recruitment from the German Moebius Foundation | CBCL (parent-report) | 32.2% (n = 10) scored in clinical range on at least one scale No significant differences between genders on any scale Social Problems more common among older children |
Briegel (2012) | Germany | 17 (8) | 11.59 (1.87) |
Moebius syndrome:
|
| Cross-sectional; recruitment from the German Moebius Foundation | AFS | Lower Test Anxiety and Manifest Anxiety than normative data |
DIKJ | Lower Total Depression than normative data | |||||||
SDQ- Deu | 52.9% (n = 9) scored in the abnormal range on at least one scale Social Problems significantly increased with age | |||||||
Strobel and Renner (2016) | Germany | 26 (11) | 11.30 (4.50) |
Moebius syndrome:
|
| Cross-sectional; recruitment from the German Moebius Syndrome Foundation | KINDL | Significantly greater difficulties with Friends compared to normative data (parent- and self-report) Significantly greater difficulties with Friends and Emotional Well-Being with increased age |
SDQ- |Deu | Significantly higher Total Difficulties (parent-report) and Peer Problems (parent- and self-report) than normative data | |||||||
Briegel, Heinzel-Guttenbrunner, and Beate (2019) | Germany | 26 (14) | 15.20 (3.48) |
Moebius syndrome:
|
| Cohort; four-year follow-up to participants in study described by Briegel et al. (2010); German Moebius Syndrome Foundation | CBCL (parent report) | Significantly higher scores (more problems) than the general population on all scales apart from Externalising problems 55% (n = 11) scored in clinical range on at least one scale No significant changes between scores at T1 (Briegel et al. 2010) and T2 (Briegel et al. 2019) |
Study | |||||
---|---|---|---|---|---|
Briegel et al. (2007) [15] | Briegel et al. (2010) [16] | Briegel (2012) [21] | Strobel and Renner (2016) [17] | Briegel et al. (2019) [18] | |
1. Was the research question or objective in this paper clearly stated? | Yes | Yes | Yes | Yes | Yes |
2. Was the study population clearly specified and defined? | Yes | Yes | Yes | Yes | Yes |
3. Was the participation rate of eligible persons at least 50%? | Yes | Yes | NR | Yes | Yes |
4. Were all the subjects selected or recruited from the same or similar populations (including the same time period)? Were inclusion and exclusion criteria for being in the study prespecified and applied uniformly to all participants? | Yes | Yes | Yes | Yes | Yes |
5. Was a sample size justification, power description, or variance and effect estimates provided? | Yes | No | No | Yes | No |
6. For the analyses in this paper, were the exposure(s) of interest measured prior to the outcome(s) being measured? | No | No | No | No | No |
7. Was the timeframe sufficient so that one could reasonably expect to see an association between exposure and outcome if it existed? | Yes | Yes | Yes | Yes | Yes |
8. For exposures that can vary in amount or level, did the study examine different levels of the exposure as related to the outcome (e.g., categories of exposure, or exposure measured as continuous variable)? | No | No | No | No | No |
9. Were the exposure measures (independent variables) clearly defined, valid, reliable, and implemented consistently across all study participants? | Yes | Yes | Yes | Yes | Yes |
10. Was the exposure(s) assessed more than once over time? | No | No | No | No | Yes |
11. Were the outcome measures (dependent variables) clearly defined, valid, reliable, and implemented consistently across all study participants? | NR | NR | NR | Yes | NR |
12. Were the outcome assessors blinded to the exposure status of participants? | NR | NR | NR | NR | NR |
13. Was loss to follow-up after baseline 20% or less? | NA | NA | NA | NA | Yes |
14. Were key potential confounding variables measured and adjusted statistically for their impact on the relationship between exposure(s) and outcome(s)? | No | No | No | No | No |
Overall Score (0–14) | 7 | 6 | 5 | 8 | 8 |
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Hotton, M.; Huggons, E.; Hamlet, C.; Bogart, K.; Johnson, D.; Norris, J.H.; Kilcoyne, S.; Dalton, L. A Systematic Review of the Psychosocial Adjustment of Children and Adolescents with Facial Palsy: The Impact of Moebius Syndrome. Int. J. Environ. Res. Public Health 2020, 17, 5528. https://0-doi-org.brum.beds.ac.uk/10.3390/ijerph17155528
Hotton M, Huggons E, Hamlet C, Bogart K, Johnson D, Norris JH, Kilcoyne S, Dalton L. A Systematic Review of the Psychosocial Adjustment of Children and Adolescents with Facial Palsy: The Impact of Moebius Syndrome. International Journal of Environmental Research and Public Health. 2020; 17(15):5528. https://0-doi-org.brum.beds.ac.uk/10.3390/ijerph17155528
Chicago/Turabian StyleHotton, Matthew, Esme Huggons, Claire Hamlet, Kathleen Bogart, David Johnson, Jonathan H. Norris, Sarah Kilcoyne, and Louise Dalton. 2020. "A Systematic Review of the Psychosocial Adjustment of Children and Adolescents with Facial Palsy: The Impact of Moebius Syndrome" International Journal of Environmental Research and Public Health 17, no. 15: 5528. https://0-doi-org.brum.beds.ac.uk/10.3390/ijerph17155528