- Email: [email protected]
Self-perception of children and adolescents with Möbius sequence
Research in Developmental Disabilities 33 (2012) 54–59
Contents lists available at SciVerse ScienceDirect
Research in Developmental Disabilities
Self-perception of children and adolescents with Mo¨bius sequence Wolfgang Briegel * Department of Child and Adolescent Psychiatry and Psychotherapy, Leopoldina Hospital, Schweinfurt, Germany
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 August 2011 Accepted 15 August 2011 Available online 4 October 2011
Mo¨bius sequence is a rare congenital disorder usually defined as a combination of facial weakness with impairment of ocular abduction. Some studies suggest that psychosocial and psychiatric problems might be increased among affected persons. So far, there have been no studies on the self-perception of children and adolescents with the sequence. Seventeen participants with Mo¨bius sequence (9 male, 8 female) aged 9–15 (mean: 11.59) years were studied. None of the probands was mentally retarded or had a diagnosis of autism spectrum disorder. Participants filled out well standardized German questionnaires on depression (Depressionsinventar fu¨r Kinder und Jugendliche [DIKJ]), anxiety (Angstfragebogen fu¨r Schu¨ler [AFS]) and personality aspects (Perso¨nlichkeitsfragebogen fu¨r Kinder von 9–14 Jahren [PFK 9–14]). Additionally, their primary caregivers were asked to complete a special questionnaire to compile the probands’ personal, somatic and psychosocial history as well as the German version of the Strengths and Difficulties Questionnaire, Parent Form [SDQ-Deu]. According to the participants’ self-perception only one girl scored clinical (t-score 63) for manifest anxiety [AFS] and depression [DIKJ] (5.9% vs. 10% in the general population). Moreover, the whole sample reported significantly lower test anxiety (p = 0.000) and manifest anxiety (p = 0.005) [AFS] whereas general anxiety as a personality aspect [PFK 9–14] did not differ from the normative sample. Compared to normative data, subjects expressed significantly less depression (p = 0.023) and impulsivity (p = 0.042). One out of 17 subjects was rated abnormal for total problems on the SDQ-Deu (5.9% vs. 10% of the normative sample), five participants scored abnormal for social problems (29.4%) and three for emotional problems (17.6%). Social problems correlated significantly with the probands’ age (rho = 0.707; p = 0.002). As Mo¨bius patients have severe difficulties with facial expression of feelings, and others might therefore falsely recognize them as serious or even depressed, the subjects’ selfperception is crucial for assessment and diagnosis, especially if it differs considerably from that of the primary caregivers. ß 2011 Elsevier Ltd. All rights reserved.
Keywords: Mo¨bius sequence Self-perception Children and adolescents
1. Introduction Mo¨bius syndrome (Online Mendelian Inheritance in Man no. 157900), or better Mo¨bius sequence, is a rare, non-progressive condition usually defined as congenital facial weakness (uni- or bilateral) combined with impairment of ocular abduction (Verzijl, van der Zwaag, Cruysberg, & Padberg, 2003). As there is no central register for the syndrome in most countries, its true prevalence remains unclear (Briegel, Hofmann, & Schwab, 2010) with an estimated rate of 0.0002–0.002% of births (Kuklik, 2000; Verzijl et al., 2003). Etiology and pathogenesis are still not fully understood (Briegel, 2006; Kumar, 1990).
* Correspondence address: Klinik fu¨r Kinder- und Jugendpsychiatrie & Psychotherapie, Leopoldina-Krankenhaus, Gustav-Adolf-Str. 4, 97422 Schweinfurt, Germany. Fax: +49 9721 720 2901. E-mail address: [email protected]. 0891-4222/$ – see front matter ß 2011 Elsevier Ltd. All rights reserved. doi:10.1016/j.ridd.2011.08.013
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So far, most studies have focused on physical anomalies whereas neuropsychiatric or behavioural aspects have been less covered (Briegel, 2006). Disturbances in psychomotor and speech development are said to be very common (Sjo¨green, Andersson-Norinder, & Jacobsson, 2001; Verzijl et al., 2003), and intellectual disabilities seems to occur in about 10% of patients (Briegel, Schimek, Knapp, et al., 2009; Kuklik, 2000). As recent studies show, the association of autism spectrum disorders and Mo¨bius sequence is by far less frequent than formerly thought (Briegel, Schimek, Kamp-Becker, Hofmann, & Schwab, 2009; Briegel, Schimek, & Kamp-Becker, 2010). So far, two studies focused on behaviour problems of children and adolescents with the sequence (Briegel, Hofmann, & Schwab, 2007; Briegel, Hofmann, et al., 2010). One of them found that 1.5–5 year old participants with Mo¨bius sequence did not show essentially increased rates of clinical behaviour problems (Briegel et al., 2007). The other one confirmed this result for 4–17 year old probands but also showed that social problems seem to increase with age (Briegel, Hofmann, et al., 2010). Studies evaluating the self-perception of Mo¨bius patients of this age group are completely missing. 2. Aims The aim of this study was to assess 9–15 year old Mo¨bius patients’ self-perception of anxiety, depression and personality aspects as well as their primary caregivers’ perception of behavioural and emotional aspects. 3. Methods 3.1. Participants Participants aged 9–15 years who participated in a nationwide study on intellectual capacities of Mo¨bius patients (Briegel, Schimek, Knapp, et al., 2009). All participants underwent a physical examination, an intelligence testing with the WISC-III (Tewes, Rossmann, & Schallberger, 2000) and a special diagnostic algorithm for autism spectrum disorders (Briegel, Schimek, et al., 2010). Inclusion criteria were congenital facial weakness (uni- or bilateral) with concomitant impairment of ocular abduction (uni- or bilateral). Familiar cases of Mo¨bius sequence, cases with intellectual disabilities (defined as a Full Scale IQ < 70 in the WISC-III) and/or a diagnosis of autism spectrum disorder should be excluded. Prior to their inclusion in this study, written informed consent was obtained from all patients and their primary caregivers. The primary caregivers of all participants filled out: a special questionnaire to compile personal, somatic and psychosocial history of the probands the German version of the Strengths and Difficulties Questionnaire, Parent Form [SDQ-Deu] (Klasen, Woerner, Rothenberger, & Goodman, 2003). This well standardised 25 item questionnaire for parents of 4–16 year old children comprises five scales (emotional symptoms, conduct problems, hyperactivity, peer problems, prosocial) and a total difficulties score with classifications of normal (80%), borderline (10%) and abnormal (10%) (Woerner, Becker, & Rothenberger, 2004). All probands were asked to fill out the following questionnaires: The Angstfragebogen fu¨r Schu¨ler [AFS], a German anxiety questionnaire for students aged 9–16 years comprising 50 items (Wieczerkowski, Nickel, Janowski, Fittkau, & Rauer, 1998). Results of the scales test anxiety, manifest anxiety, dislike of school and social desirability are presented as t-values (mean t-value: 50, standard deviation: 10). The Depressionsinventar fu¨r Kinder und Jugendliche [DIKJ] (Stiensmeier-Pelster, Schu¨rmann, & Duda, 2000), the German version of the Children’s Depression Inventory (Kovacs, 1985). Results are given as t-values. The dimension self-perception of the Perso¨nlichkeitsfragebogen fu¨r Kinder und Jugendliche [PFK 9–14]. The PFK 9–14 is a well standardised German personality questionnaire for children and adolescents aged 9–14 years (Seitz & Rausche, 2004). The dimension self-perception comprises 60 items and 5 scales (general anxiety; belief in own opinions, decisions and plans; selfperception of impulsivity; tendency to overestimate one’s own capabilities; feeling of inferiority). Results are given as t-values. To have comparable criteria for instruments of self-perception and perception by others, t-scores 63 were defined as clinical for the AFS, the DIJK and the PFK 9–14 subscales. Statistical analysis of the results was done using PASW Statistics 18. To test the differences between the mean scores of the study sample and those of the AFS, the DIKJ and the PFK 9–14 normative samples, the one-sample t-test was used. Statistical significance was defined as p < 0.05 (two-sided). 4. Results 4.1. Participants Nine males and 8 females with Mo¨bius sequence aged 9–15 years (mean: 11.59 years, standard deviation: 1.87 years) could be included. In most cases, Mo¨bius sequence had been diagnosed by a geneticist or pediatrician. Mean Full score of the
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German version of the WISC-III (Tewes et al., 2000) was 93.88 (standard deviation: 14.03; range: 78–135). None of the probands had a diagnosis of autism spectrum disorder. Fifteen out of 17 probands had congenital bilateral facial paralysis, none of them had had facial reanimation. Sixteen of 17 showed bilateral impairment of ocular abduction, and 6/17 had impaired ocular adduction (in all cases bilateral). Fifteen of 17 probands wore glasses, 5 had had operative correction of congenital strabism, and 3 subjects showed ptosis. 2 probands had hearing impairment, but none of them needed a hearing aid. Fourteen of 17 suffered from congenital hypoglossal paralysis (all cases bilateral). Five of 17 probands had (had) congenital malformations of the upper extremities (mostly milder forms, e.g. syndactyly and brachyphalangy). Nine participants showed congenital lower extremity malformations, typically talipes. Three participants had a congenital heart defect (in two cases dextrocardia), another three had Poland’s syndrome, and one proband showed Kallmann’s syndrome. Familial cases of Mo¨bius sequence could not be found. Thirteen children lived with both parents, 4 with only one parent, and one with adoptive parents. Thirteen of 17 children had at least one sibling. Five children attended elementary school. After elementary school German students have to choose between three types of secondary schooling modalities: ‘‘Hauptschule’’ (grades 5–9), which is supposed to prepare for less-skilled jobs, ‘‘Realschule’’ (grades 5–10) as an intermediate school, which is supposed to prepare mainly practically and theoretically oriented students for trade, technical and administrative professions, and ‘‘Gymnasium’’ as academic secondary school, preparing students to tertiary education or university. Seven children or adolescents went to ‘‘Gymnasium’’, two to ‘‘Realschule’’, and one to ‘‘Hauptschule’’. Two subjects attended a school specialized in providing education for children with special needs, e.g. for physically handicapped students. Three participants had been already seen by a child and adolescent psychiatrist. Two children had attention deficit/ hyperactivity disorder and were on methylphenidate, and one subject had no ICD-10 diagnosis. Another child had lamotrigine because of seizures. 4.2. Self-perception of anxiety, depression and personality aspects For results of the AFS and the DIKJ see Table 1. Results of the PFK 9–14 are shown in Table 2. A 12 year old girl was rated both clinical for manifest anxiety, dislike of school, and depression. She also scored high for feelings of inferiority [PFK 9–14] (t-score = 63) and emotional problems [SDQ-Deu] (raw score: 7, abnormal). 4.3. Primary caregivers’ perception of children’s strengths and difficulties For results of the SDQ-Deu see Table 3. One out of 17 subjects, a 12 year old boy was rated clinical on the SDQ total score (5.9% vs. 10% of the nomative sample). This boy had a diagnosis of attention deficit/hyperactivity disorder, was treated with methylphenidate and had a Full scale IQ
Table 1 AFS and DIKJ: t-scores: means, standard deviations, range, comparison with normative sample, and number of subjects with a t-score 63. Test/scale AFS Test anxiety Manifest anxiety Dislike of school Social desirability DIKJ Total
Mean (standard deviation)
Range
Comparison with normative sample
t-Score 63
39.59 43.71 45.24 53.94
31–49 34–65 35–72 39–65
Lower (p = 0.000) Lower (p = 0.005) n.s. n.s.
– 1 girl 1 girl, 1 boy 4 girls, 2 boys
33–72
Lower (p = 0.023)
1 girl
(7.64) (8.10) (10.87) (9.60)
44.00 (5.94)
n.s., not significant.
Table 2 PFK 9–14: self-perception aspects. t-Scores: means, standard deviations, range, comparison with normative sample, and number of clinical cases (n = 16). Self-perception: subscales
t-Scores Mean (standard deviation)
Range
General anxiety Belief in own opinions, decisions and plans Self-perception of impulsivity Tendency to overestimate one’s own capabilities Feeling of inferiority
46.37 52.75 44.00 44.88 45.63
35–58 36–66 30–75 37–69 33–63
n.s., not significant.
(8.09) (9.91) (10.81) (9.69) (9.32)
Comparison with normative sample
t-Score 63
n.s. n.s. Lower (p = 0.042) n.s. n.s.
– 1 1 1 1
girl, 3 boys (25.0%) boy (6.3%) boy (6.3%) girl (6.3 &)
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Table 3 SDQ: minimum and maximum values, means, standard deviations, and clinical cases. Subscale
Minimum
Maximum
Mean
Standard deviation
Abnormal/clinical cases
Prosocial behaviour Emotional problems Behavioural problems Hyperactivity Social problems Total problem score
5 0 0 0 0 0
10 7 6 8 7 25
8.18 2.65 1.18 1.94 3.12 9.00
1.78 1.97 1.59 2.46 2.34 5.61
– 2 1 2 4 1
girls, 1 boy (17.6%) boy (5.9%) boys (11.8%) girls, 1 boy (29.4%) boy (5.9%)
of 79. He was scored clinical for behavioural problems, hyperactivity and social problems by his primary caregiver and had the highest scores of all participants for impulsivity (t-score = 75). Nine out of 17 probands (6 girls and 3 boys) were rated abnormal on at least one SDQ-Deu scale. Social problems were nearly three times as frequent among the subjects of this study as in the normative study (29.4% vs. 10%). There was also a tendency to more emotional problems among Mo¨bius subjects compared to the normal population (17.6% vs. 10%). Eight subjects (47.1% vs. 20%) were at least borderline cases for social problems, and five for emotional problems (29.4%). The SDQ subscale social problems correlated significantly with the probands’ age (rho = 0.707; p = 0.002). No sex differences could be found for any of the questionnaires.
5. Discussion Mo¨bius sequence is a very rare congenital condition. Therefore, the largest study on psychological aspects of adults comprised 37 participants (Bogart & Matsumoto, 2010), whereas the largest sample of children and adolescents consisted of 31 probands (Briegel, Hofmann, et al., 2010). To the author’s knowledge, this is the first study focusing on the self-perception of anxiety, depression and personality aspects. Compared to the study of Verzijl et al. (2003), participants showed similar rates of somatic problems. As intellectual disability and an age below 9 were exclusion criteria, all probands of this study were able to fill out questionnaires without major problems due to reading or understanding. According to their parents only one out of 17 participants (5.9% vs. 10% of the normative sample) was rated abnormal on the SDQ-Deu total score, a result which is similar to the findings of two previous studies. Assessing preschool children with the CBCL 1.5–5 (Achenbach & Rescorla, 2000; Arbeitsgruppe Deutsche Child Behavior Checklist, 2002). Briegel et al. (2007) found one out of 12 participants to score in the clinical range for total problems [t-score 64; 8.3%]. In another study (Briegel, Hofmann, et al., 2010) three out of 31 Mo¨bius patients aged 4–17 years were rated clinical [t-score 64; 9.7%] on the total problem scale of the CBCL/4–18 (Arbeitsgruppe Deutsche Child Behavior Checklist, 1998). In this study, eight participants (47.1 vs. 20% of the normative sample) were at least borderline cases for social problems, and five for emotional problems (29.4%). Social problems in the clinical range were nearly three times as frequent among the participants in this study as in the normative sample (29.4% vs. 10%). Moreover, social problems showed a significantly positive correlation with the probands’ age indicating an increase with age. Similar results have been already described for social problems in the above mentioned study of 31 subjects (Briegel, Hofmann, et al., 2010), of whom 14 subjects also participated in this study. Briegel, Hofmann, et al. (2010) concluded that it was not surprising that social problems represented the most important problems and that they showed an increase with age as various features associated with Mo¨bius sequence, especially masklike facies, could make it very difficult for affected participants to achieve normal interactions and relationships, especially if they were unknown to others. Another conclusion of the authors was that negative experiences like rejection and a lack of reinforcement by others, together with the patients’ own perception of being different might result in low self-esteem, behavioural problems, especially social ones, and even psychiatric disorders (Briegel, Hofmann, et al., 2010). Interestingly, this study found a considerable discrepancy between the parents’ perception of emotional problems and the participants’ self-perception. Three out of 17 participants (17.6%) were rated abnormal for emotional problems by their primary caregivers whereas only one girl scored clinical for manifest anxiety [AFS] and depression [DIKJ]. Moreover, the whole sample of subjects reported significantly lower depression, test anxiety and manifest anxiety whereas general anxiety as a personality aspect did not differ from the normative sample. The probands also showed less impulsivity than those of the normative sample. This result is similar to findings in adults with the sequence, who reported about more inhibitedness and introversion compared to the normative sample (Briegel, 2007). A possible explanation might be that children and adolescents with Mo¨bius syndrome have to face several neurologically defined problems, e.g. articulatory and communication problems, which forces them to develop alternative strategies to achieve an integration as good as possible. Obviously, these compensational efforts require a lot of self-control and discipline which are incompatible with impulsivity. Feelings of inferiority were similar to the normative sample indicating that the participants did not show low self-esteem – a finding which is in contrast to the suggestions of Briegel, Hofmann, et al. (2010).
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One possible explanation for the discrepancy between self-perception and perception by others might be that different questionnaires have been used to assess emotional problems (SDQ-Deu for primary caregivers, DIKJ and AFS for Mo¨bius patients). Another hypothesis is that subjects willing to be like their peers dissimulated emotional problems, but – of course – it is also possible that parents over-estimated their children’s problems. However, discrepancies between self-perception and perception by others seem to be quite normal (e.g.: CBCL vs. YSR: Seiffge-Krenke & Kollmar, 1998; SDQ parent report vs. self-report: van der Meer, Dixon, & Rose, 2008). There have also been divergent findings for the self-perception of depression and anxiety in adults. In his study on 21 adults Briegel (2007) found a tendency to more depression and anxiety among the patients compared to the normative sample, whereas Bogart and Matsumoto (2010) who compared 37 Mo¨bius patients with a matched control group could not find any significant differences between both groups. Bogart and Matsumoto suggested that cultural differences might be a possible explanation (Bogart & Matsumoto, 2010). To sum it up, this is the third study showing that primary caregivers of children with Mo¨bius sequence do not see increased total behaviour problems compared to the general population. However, they see more social problems which seem to increase with age. Additionally, primary caregivers report more emotional problems than the patients. In their selfperception affected children and adolescents show less anxiety, depression and impulsivity than normal subjects of the same age group. 6. Limitations This study has some limitations: It is unclear whether the study group is representative for the whole age group of subjects with Mo¨bius syndrome. As there is no central register for that syndrome in Germany and in most other countries, its true prevalence remains unclear. Therefore, questionable representativeness applies to nearly all studies on Mo¨bius sequence (Briegel, 2006). The fact that the study sample represents a self-selected sample of subjects might also limit generalization of the findings. A last limitation is that this study did not include a matched control group. 7. Conclusion As Mo¨bius patients have severe difficulties with facial expression of feelings, and others might therefore falsely recognize them as serious or even depressed, the participants’ self-perception is crucial for assessment and diagnosis, especially if it differs considerably from that of the primary caregivers. Acknowledgements The author is very grateful to Mo¨bius Syndrom Deutschland e.V., the German Mo¨bius association, and to all participating families. References Achenbach, T. M., & Rescorla, L. A. (2000). Manual for the ASEBA preschool forms & profiles. Burlington, VT: University of Vermont, Research Center for Children, Youth, & Families. Arbeitsgruppe Deutsche Child Behavior Checklist. (1998). Elternfragebogen u¨ber das Verhalten von Kindern und Jugendlichen; deutsche Bearbeitung der Child Behavior Checklist (CBCL/4-18). Einfu¨hrung und Anleitung der Handauswertung. 2. Auflage mit deutschen Normen bearbeitet von M. Do¨pfner, J. Plu¨ck, S. Bo¨lte, K. Lenz, P. Melchers und K. Heim. Ko¨ln: Arbeitsgruppe Kinder-, Jugend- und Familiendiagnostik (KJFD). Arbeitsgruppe Deutsche Child Behavior Checklist. (2002). Elternfragebogen fu¨r Klein- und Vorschulkinder (CBCL/1.5-5)PL Ko¨ln. Arbeitsgruppe Kinder-, Jugend- und Familiendiagnostik (KJFD). Bogart, K. R., & Matsumoto, D. (2010). Living with Moebius syndrome: Adjustment, social competence, and satisfaction with life. The Cleft Palate-Craniofacial Journal, 47(2), 134–142. Briegel, W. (2006). Neuropsychiatric findings of Mo¨bius sequence – A review. Clinical Genetics, 70, 91–97. Briegel, W. (2007). Psychopathology and personality aspects of adults with Mo¨bius sequence. Clinical Genetics, 71, 376–377. Briegel, W., Hofmann, C., & Schwab, K. O. (2007). Moebius sequence: Behaviour problems of preschool children and parental stress. Genetic Counseling, 18, 267– 275. Briegel, W., Hofmann, C., & Schwab, K. O. (2010). Behaviour problems of patients with Mo¨bius sequence and parental stress. Journal of Paediatrics and Child Health, 46, 144–148. Briegel, W., Schimek, M., & Kamp-Becker, I. (2010). Moebius sequence and autism spectrum disorders – Less frequently associated than formerly thought. Research in Developmental Disabilities, 31, 1462–1466. Briegel, W., Schimek, M., Kamp-Becker, I., Hofmann, C., & Schwab, K. O. (2009). Autism spectrum disorders in children and adolescents with Moebius Sequence. European Child and Adolescent Psychiatry, 18, 515–519. Briegel, W., Schimek, M., Knapp, D., Holderbach, R., Wenzel, P., & Knapp, E.-M. (2009). Cognitive evaluation in children and adolescents with Mo¨bius sequence. Child: Care, Health and Development, 35, 650–655. ¨ bersicht und Klasen, H., Woerner, W., Rothenberger, A., & Goodman, R. (2003). Die deutsche Fassung des Strengths and Difficulties Questionnaire (SDQ-Deu) – U Bewertung erster Validierungs- und Normierungsbefunde. Praxis der Kinderpsychologie und Kinderpsychiatrie, 52, 491–502. Kovacs, M. (1985). The Children’s Depression Inventory (CDI). American Psychologist, 44, 209–215. Kuklik, M. (2000). Poland-Mo¨bius syndrome and disruption spectrum affecting the face and extremities: A review paper and presentation of five cases. Acta Chirurgiae Plasticae, 42, 95–103. Kumar, D. (1990). Moebius syndrome. Journal of Medical Genetics, 27, 122–126. Seiffge-Krenke, I., & Kollmar, F. (1998). Discrepancies between mothers’ and fathers’ perceptions of sons’ and daughters’ problem behaviour: A longitudinal analysis of parent-adolescent agreement on internalising and externalising problem behaviour. Journal of Child Psychology and Psychiatry, 39, 687–697. Seitz, W., & Rausche, A. (2004). PFK 9–14, Perso¨nlichkeitsfragebogen fu¨r Kinder zwischen 9 und 14 Jahren. Manual (4th edition). Go¨ttingen: Hogrefe-Verlag.
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Sjo¨green, L., Andersson-Norinder, J., & Jacobsson, C. (2001). Development of speech, feeding, eating, and facial expression in Mo¨bius sequence. International Journal of Pediatric Otorhinolaryngology, 60, 197–204. Stiensmeier-Pelster, J., Schu¨rmann, M., & Duda, K. (2000). Depressions-Inventar fu¨r Kinder und Jugendliche (2nd edition). Go¨ttingen: Hogrefe-Verlag. Tewes, U., Rossmann, P., & Schallberger, U. (2000). Hamburg-Wechsler-Intelligenztest III. Go¨ttingen: Testzentrale, Hogrefe-Verlag. van der Meer, M., Dixon, A., & Rose, D. (2008). Parent and child agreement on reports of problem behaviour obtained from a screening questionnaire, the SDQ. European Child and Adolescent Psychiatry, 17, 491–497. Verzijl, H. T. F. M. , van der Zwaag, B., Cruysberg, J. R. M., & Padberg, G. W. (2003). Mo¨bius syndrome redefined. A syndrome of rhombencephalic maldevelopment. Neurology, 61, 327–333. Wieczerkowski, W., Nickel, H., Janowski, A., Fittkau, B., & Rauer, W. (1998). Angstfragebogen fu¨r Schu¨ler (AFS). Handanweisung fu¨r die Durchfu¨hrung, Auswertung und Interpretation. Go¨ttingen: Hogrefe-Verlag. Woerner, W., Becker, A., & Rothenberger, A. (2004). Normative data and scale properties of the German parent SDQ. European Child and Adolescent Psychiatry, 13 Suppl.(2), II11–II16.
Contents lists available at SciVerse ScienceDirect
Research in Developmental Disabilities
Self-perception of children and adolescents with Mo¨bius sequence Wolfgang Briegel * Department of Child and Adolescent Psychiatry and Psychotherapy, Leopoldina Hospital, Schweinfurt, Germany
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 August 2011 Accepted 15 August 2011 Available online 4 October 2011
Mo¨bius sequence is a rare congenital disorder usually defined as a combination of facial weakness with impairment of ocular abduction. Some studies suggest that psychosocial and psychiatric problems might be increased among affected persons. So far, there have been no studies on the self-perception of children and adolescents with the sequence. Seventeen participants with Mo¨bius sequence (9 male, 8 female) aged 9–15 (mean: 11.59) years were studied. None of the probands was mentally retarded or had a diagnosis of autism spectrum disorder. Participants filled out well standardized German questionnaires on depression (Depressionsinventar fu¨r Kinder und Jugendliche [DIKJ]), anxiety (Angstfragebogen fu¨r Schu¨ler [AFS]) and personality aspects (Perso¨nlichkeitsfragebogen fu¨r Kinder von 9–14 Jahren [PFK 9–14]). Additionally, their primary caregivers were asked to complete a special questionnaire to compile the probands’ personal, somatic and psychosocial history as well as the German version of the Strengths and Difficulties Questionnaire, Parent Form [SDQ-Deu]. According to the participants’ self-perception only one girl scored clinical (t-score 63) for manifest anxiety [AFS] and depression [DIKJ] (5.9% vs. 10% in the general population). Moreover, the whole sample reported significantly lower test anxiety (p = 0.000) and manifest anxiety (p = 0.005) [AFS] whereas general anxiety as a personality aspect [PFK 9–14] did not differ from the normative sample. Compared to normative data, subjects expressed significantly less depression (p = 0.023) and impulsivity (p = 0.042). One out of 17 subjects was rated abnormal for total problems on the SDQ-Deu (5.9% vs. 10% of the normative sample), five participants scored abnormal for social problems (29.4%) and three for emotional problems (17.6%). Social problems correlated significantly with the probands’ age (rho = 0.707; p = 0.002). As Mo¨bius patients have severe difficulties with facial expression of feelings, and others might therefore falsely recognize them as serious or even depressed, the subjects’ selfperception is crucial for assessment and diagnosis, especially if it differs considerably from that of the primary caregivers. ß 2011 Elsevier Ltd. All rights reserved.
Keywords: Mo¨bius sequence Self-perception Children and adolescents
1. Introduction Mo¨bius syndrome (Online Mendelian Inheritance in Man no. 157900), or better Mo¨bius sequence, is a rare, non-progressive condition usually defined as congenital facial weakness (uni- or bilateral) combined with impairment of ocular abduction (Verzijl, van der Zwaag, Cruysberg, & Padberg, 2003). As there is no central register for the syndrome in most countries, its true prevalence remains unclear (Briegel, Hofmann, & Schwab, 2010) with an estimated rate of 0.0002–0.002% of births (Kuklik, 2000; Verzijl et al., 2003). Etiology and pathogenesis are still not fully understood (Briegel, 2006; Kumar, 1990).
* Correspondence address: Klinik fu¨r Kinder- und Jugendpsychiatrie & Psychotherapie, Leopoldina-Krankenhaus, Gustav-Adolf-Str. 4, 97422 Schweinfurt, Germany. Fax: +49 9721 720 2901. E-mail address: [email protected]. 0891-4222/$ – see front matter ß 2011 Elsevier Ltd. All rights reserved. doi:10.1016/j.ridd.2011.08.013
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So far, most studies have focused on physical anomalies whereas neuropsychiatric or behavioural aspects have been less covered (Briegel, 2006). Disturbances in psychomotor and speech development are said to be very common (Sjo¨green, Andersson-Norinder, & Jacobsson, 2001; Verzijl et al., 2003), and intellectual disabilities seems to occur in about 10% of patients (Briegel, Schimek, Knapp, et al., 2009; Kuklik, 2000). As recent studies show, the association of autism spectrum disorders and Mo¨bius sequence is by far less frequent than formerly thought (Briegel, Schimek, Kamp-Becker, Hofmann, & Schwab, 2009; Briegel, Schimek, & Kamp-Becker, 2010). So far, two studies focused on behaviour problems of children and adolescents with the sequence (Briegel, Hofmann, & Schwab, 2007; Briegel, Hofmann, et al., 2010). One of them found that 1.5–5 year old participants with Mo¨bius sequence did not show essentially increased rates of clinical behaviour problems (Briegel et al., 2007). The other one confirmed this result for 4–17 year old probands but also showed that social problems seem to increase with age (Briegel, Hofmann, et al., 2010). Studies evaluating the self-perception of Mo¨bius patients of this age group are completely missing. 2. Aims The aim of this study was to assess 9–15 year old Mo¨bius patients’ self-perception of anxiety, depression and personality aspects as well as their primary caregivers’ perception of behavioural and emotional aspects. 3. Methods 3.1. Participants Participants aged 9–15 years who participated in a nationwide study on intellectual capacities of Mo¨bius patients (Briegel, Schimek, Knapp, et al., 2009). All participants underwent a physical examination, an intelligence testing with the WISC-III (Tewes, Rossmann, & Schallberger, 2000) and a special diagnostic algorithm for autism spectrum disorders (Briegel, Schimek, et al., 2010). Inclusion criteria were congenital facial weakness (uni- or bilateral) with concomitant impairment of ocular abduction (uni- or bilateral). Familiar cases of Mo¨bius sequence, cases with intellectual disabilities (defined as a Full Scale IQ < 70 in the WISC-III) and/or a diagnosis of autism spectrum disorder should be excluded. Prior to their inclusion in this study, written informed consent was obtained from all patients and their primary caregivers. The primary caregivers of all participants filled out: a special questionnaire to compile personal, somatic and psychosocial history of the probands the German version of the Strengths and Difficulties Questionnaire, Parent Form [SDQ-Deu] (Klasen, Woerner, Rothenberger, & Goodman, 2003). This well standardised 25 item questionnaire for parents of 4–16 year old children comprises five scales (emotional symptoms, conduct problems, hyperactivity, peer problems, prosocial) and a total difficulties score with classifications of normal (80%), borderline (10%) and abnormal (10%) (Woerner, Becker, & Rothenberger, 2004). All probands were asked to fill out the following questionnaires: The Angstfragebogen fu¨r Schu¨ler [AFS], a German anxiety questionnaire for students aged 9–16 years comprising 50 items (Wieczerkowski, Nickel, Janowski, Fittkau, & Rauer, 1998). Results of the scales test anxiety, manifest anxiety, dislike of school and social desirability are presented as t-values (mean t-value: 50, standard deviation: 10). The Depressionsinventar fu¨r Kinder und Jugendliche [DIKJ] (Stiensmeier-Pelster, Schu¨rmann, & Duda, 2000), the German version of the Children’s Depression Inventory (Kovacs, 1985). Results are given as t-values. The dimension self-perception of the Perso¨nlichkeitsfragebogen fu¨r Kinder und Jugendliche [PFK 9–14]. The PFK 9–14 is a well standardised German personality questionnaire for children and adolescents aged 9–14 years (Seitz & Rausche, 2004). The dimension self-perception comprises 60 items and 5 scales (general anxiety; belief in own opinions, decisions and plans; selfperception of impulsivity; tendency to overestimate one’s own capabilities; feeling of inferiority). Results are given as t-values. To have comparable criteria for instruments of self-perception and perception by others, t-scores 63 were defined as clinical for the AFS, the DIJK and the PFK 9–14 subscales. Statistical analysis of the results was done using PASW Statistics 18. To test the differences between the mean scores of the study sample and those of the AFS, the DIKJ and the PFK 9–14 normative samples, the one-sample t-test was used. Statistical significance was defined as p < 0.05 (two-sided). 4. Results 4.1. Participants Nine males and 8 females with Mo¨bius sequence aged 9–15 years (mean: 11.59 years, standard deviation: 1.87 years) could be included. In most cases, Mo¨bius sequence had been diagnosed by a geneticist or pediatrician. Mean Full score of the
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German version of the WISC-III (Tewes et al., 2000) was 93.88 (standard deviation: 14.03; range: 78–135). None of the probands had a diagnosis of autism spectrum disorder. Fifteen out of 17 probands had congenital bilateral facial paralysis, none of them had had facial reanimation. Sixteen of 17 showed bilateral impairment of ocular abduction, and 6/17 had impaired ocular adduction (in all cases bilateral). Fifteen of 17 probands wore glasses, 5 had had operative correction of congenital strabism, and 3 subjects showed ptosis. 2 probands had hearing impairment, but none of them needed a hearing aid. Fourteen of 17 suffered from congenital hypoglossal paralysis (all cases bilateral). Five of 17 probands had (had) congenital malformations of the upper extremities (mostly milder forms, e.g. syndactyly and brachyphalangy). Nine participants showed congenital lower extremity malformations, typically talipes. Three participants had a congenital heart defect (in two cases dextrocardia), another three had Poland’s syndrome, and one proband showed Kallmann’s syndrome. Familial cases of Mo¨bius sequence could not be found. Thirteen children lived with both parents, 4 with only one parent, and one with adoptive parents. Thirteen of 17 children had at least one sibling. Five children attended elementary school. After elementary school German students have to choose between three types of secondary schooling modalities: ‘‘Hauptschule’’ (grades 5–9), which is supposed to prepare for less-skilled jobs, ‘‘Realschule’’ (grades 5–10) as an intermediate school, which is supposed to prepare mainly practically and theoretically oriented students for trade, technical and administrative professions, and ‘‘Gymnasium’’ as academic secondary school, preparing students to tertiary education or university. Seven children or adolescents went to ‘‘Gymnasium’’, two to ‘‘Realschule’’, and one to ‘‘Hauptschule’’. Two subjects attended a school specialized in providing education for children with special needs, e.g. for physically handicapped students. Three participants had been already seen by a child and adolescent psychiatrist. Two children had attention deficit/ hyperactivity disorder and were on methylphenidate, and one subject had no ICD-10 diagnosis. Another child had lamotrigine because of seizures. 4.2. Self-perception of anxiety, depression and personality aspects For results of the AFS and the DIKJ see Table 1. Results of the PFK 9–14 are shown in Table 2. A 12 year old girl was rated both clinical for manifest anxiety, dislike of school, and depression. She also scored high for feelings of inferiority [PFK 9–14] (t-score = 63) and emotional problems [SDQ-Deu] (raw score: 7, abnormal). 4.3. Primary caregivers’ perception of children’s strengths and difficulties For results of the SDQ-Deu see Table 3. One out of 17 subjects, a 12 year old boy was rated clinical on the SDQ total score (5.9% vs. 10% of the nomative sample). This boy had a diagnosis of attention deficit/hyperactivity disorder, was treated with methylphenidate and had a Full scale IQ
Table 1 AFS and DIKJ: t-scores: means, standard deviations, range, comparison with normative sample, and number of subjects with a t-score 63. Test/scale AFS Test anxiety Manifest anxiety Dislike of school Social desirability DIKJ Total
Mean (standard deviation)
Range
Comparison with normative sample
t-Score 63
39.59 43.71 45.24 53.94
31–49 34–65 35–72 39–65
Lower (p = 0.000) Lower (p = 0.005) n.s. n.s.
– 1 girl 1 girl, 1 boy 4 girls, 2 boys
33–72
Lower (p = 0.023)
1 girl
(7.64) (8.10) (10.87) (9.60)
44.00 (5.94)
n.s., not significant.
Table 2 PFK 9–14: self-perception aspects. t-Scores: means, standard deviations, range, comparison with normative sample, and number of clinical cases (n = 16). Self-perception: subscales
t-Scores Mean (standard deviation)
Range
General anxiety Belief in own opinions, decisions and plans Self-perception of impulsivity Tendency to overestimate one’s own capabilities Feeling of inferiority
46.37 52.75 44.00 44.88 45.63
35–58 36–66 30–75 37–69 33–63
n.s., not significant.
(8.09) (9.91) (10.81) (9.69) (9.32)
Comparison with normative sample
t-Score 63
n.s. n.s. Lower (p = 0.042) n.s. n.s.
– 1 1 1 1
girl, 3 boys (25.0%) boy (6.3%) boy (6.3%) girl (6.3 &)
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Table 3 SDQ: minimum and maximum values, means, standard deviations, and clinical cases. Subscale
Minimum
Maximum
Mean
Standard deviation
Abnormal/clinical cases
Prosocial behaviour Emotional problems Behavioural problems Hyperactivity Social problems Total problem score
5 0 0 0 0 0
10 7 6 8 7 25
8.18 2.65 1.18 1.94 3.12 9.00
1.78 1.97 1.59 2.46 2.34 5.61
– 2 1 2 4 1
girls, 1 boy (17.6%) boy (5.9%) boys (11.8%) girls, 1 boy (29.4%) boy (5.9%)
of 79. He was scored clinical for behavioural problems, hyperactivity and social problems by his primary caregiver and had the highest scores of all participants for impulsivity (t-score = 75). Nine out of 17 probands (6 girls and 3 boys) were rated abnormal on at least one SDQ-Deu scale. Social problems were nearly three times as frequent among the subjects of this study as in the normative study (29.4% vs. 10%). There was also a tendency to more emotional problems among Mo¨bius subjects compared to the normal population (17.6% vs. 10%). Eight subjects (47.1% vs. 20%) were at least borderline cases for social problems, and five for emotional problems (29.4%). The SDQ subscale social problems correlated significantly with the probands’ age (rho = 0.707; p = 0.002). No sex differences could be found for any of the questionnaires.
5. Discussion Mo¨bius sequence is a very rare congenital condition. Therefore, the largest study on psychological aspects of adults comprised 37 participants (Bogart & Matsumoto, 2010), whereas the largest sample of children and adolescents consisted of 31 probands (Briegel, Hofmann, et al., 2010). To the author’s knowledge, this is the first study focusing on the self-perception of anxiety, depression and personality aspects. Compared to the study of Verzijl et al. (2003), participants showed similar rates of somatic problems. As intellectual disability and an age below 9 were exclusion criteria, all probands of this study were able to fill out questionnaires without major problems due to reading or understanding. According to their parents only one out of 17 participants (5.9% vs. 10% of the normative sample) was rated abnormal on the SDQ-Deu total score, a result which is similar to the findings of two previous studies. Assessing preschool children with the CBCL 1.5–5 (Achenbach & Rescorla, 2000; Arbeitsgruppe Deutsche Child Behavior Checklist, 2002). Briegel et al. (2007) found one out of 12 participants to score in the clinical range for total problems [t-score 64; 8.3%]. In another study (Briegel, Hofmann, et al., 2010) three out of 31 Mo¨bius patients aged 4–17 years were rated clinical [t-score 64; 9.7%] on the total problem scale of the CBCL/4–18 (Arbeitsgruppe Deutsche Child Behavior Checklist, 1998). In this study, eight participants (47.1 vs. 20% of the normative sample) were at least borderline cases for social problems, and five for emotional problems (29.4%). Social problems in the clinical range were nearly three times as frequent among the participants in this study as in the normative sample (29.4% vs. 10%). Moreover, social problems showed a significantly positive correlation with the probands’ age indicating an increase with age. Similar results have been already described for social problems in the above mentioned study of 31 subjects (Briegel, Hofmann, et al., 2010), of whom 14 subjects also participated in this study. Briegel, Hofmann, et al. (2010) concluded that it was not surprising that social problems represented the most important problems and that they showed an increase with age as various features associated with Mo¨bius sequence, especially masklike facies, could make it very difficult for affected participants to achieve normal interactions and relationships, especially if they were unknown to others. Another conclusion of the authors was that negative experiences like rejection and a lack of reinforcement by others, together with the patients’ own perception of being different might result in low self-esteem, behavioural problems, especially social ones, and even psychiatric disorders (Briegel, Hofmann, et al., 2010). Interestingly, this study found a considerable discrepancy between the parents’ perception of emotional problems and the participants’ self-perception. Three out of 17 participants (17.6%) were rated abnormal for emotional problems by their primary caregivers whereas only one girl scored clinical for manifest anxiety [AFS] and depression [DIKJ]. Moreover, the whole sample of subjects reported significantly lower depression, test anxiety and manifest anxiety whereas general anxiety as a personality aspect did not differ from the normative sample. The probands also showed less impulsivity than those of the normative sample. This result is similar to findings in adults with the sequence, who reported about more inhibitedness and introversion compared to the normative sample (Briegel, 2007). A possible explanation might be that children and adolescents with Mo¨bius syndrome have to face several neurologically defined problems, e.g. articulatory and communication problems, which forces them to develop alternative strategies to achieve an integration as good as possible. Obviously, these compensational efforts require a lot of self-control and discipline which are incompatible with impulsivity. Feelings of inferiority were similar to the normative sample indicating that the participants did not show low self-esteem – a finding which is in contrast to the suggestions of Briegel, Hofmann, et al. (2010).
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One possible explanation for the discrepancy between self-perception and perception by others might be that different questionnaires have been used to assess emotional problems (SDQ-Deu for primary caregivers, DIKJ and AFS for Mo¨bius patients). Another hypothesis is that subjects willing to be like their peers dissimulated emotional problems, but – of course – it is also possible that parents over-estimated their children’s problems. However, discrepancies between self-perception and perception by others seem to be quite normal (e.g.: CBCL vs. YSR: Seiffge-Krenke & Kollmar, 1998; SDQ parent report vs. self-report: van der Meer, Dixon, & Rose, 2008). There have also been divergent findings for the self-perception of depression and anxiety in adults. In his study on 21 adults Briegel (2007) found a tendency to more depression and anxiety among the patients compared to the normative sample, whereas Bogart and Matsumoto (2010) who compared 37 Mo¨bius patients with a matched control group could not find any significant differences between both groups. Bogart and Matsumoto suggested that cultural differences might be a possible explanation (Bogart & Matsumoto, 2010). To sum it up, this is the third study showing that primary caregivers of children with Mo¨bius sequence do not see increased total behaviour problems compared to the general population. However, they see more social problems which seem to increase with age. Additionally, primary caregivers report more emotional problems than the patients. In their selfperception affected children and adolescents show less anxiety, depression and impulsivity than normal subjects of the same age group. 6. Limitations This study has some limitations: It is unclear whether the study group is representative for the whole age group of subjects with Mo¨bius syndrome. As there is no central register for that syndrome in Germany and in most other countries, its true prevalence remains unclear. Therefore, questionable representativeness applies to nearly all studies on Mo¨bius sequence (Briegel, 2006). The fact that the study sample represents a self-selected sample of subjects might also limit generalization of the findings. A last limitation is that this study did not include a matched control group. 7. Conclusion As Mo¨bius patients have severe difficulties with facial expression of feelings, and others might therefore falsely recognize them as serious or even depressed, the participants’ self-perception is crucial for assessment and diagnosis, especially if it differs considerably from that of the primary caregivers. 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