Abstract
In the transition period between adolescence and young adulthood, individuals with 22q11.2DS are at an increased risk of developing severe psychiatric disorders. Various studies have focused on detecting risk factors, but until now protective factors are still understudied in 22q11.2DS. The current case–control study focuses on the role of resilience and quality of life (QoL) in young adults with 22q11.2DS and behavioural problems, in comparison with persons with an intellectual disability (ID) without a known genetic disorder. Self-report (and caregiver report) standardized questionnaires were used. Predictive general linear models were constructed to compare the resilience and quality of life across both groups (22q11.2DS vs ID-group) and to analyse the association between personal characteristics in both groups. Young adults with a 22q11.2DS show less resilience compared with both the general population norms and young adults with ID. Only a subscale of resilience (Acceptance of self and life) contributes to the reported level of QoL. Reported health problems are not related to resilience, but have an important effect on QoL. Our results suggest different factors are underlying resilience and the relation with QoL in 22q11.2DS and ID in general. These factors deserve more research and are important to take into account in clinical practice.
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McDonald-McGinn DM, Sullivan KE, Marino B, Philip N, Swillen A, Vorstman JAS, Zackai EH, Emanuel BS, Vermeesch JR, Morrow BE, Scambler PJ, Bassett AS (2015) 22q11.2 deletion syndrome. Nat Rev Dis Primers 1:1–19. https://doi.org/10.1038/nrdp.2015.71
Bassett AS, McDonald-McGinn DM, Devriendt K, Digilio MC, Goldenberg P, Habel A, Marino B, Oskardottir S, Philip N, Sullivan K, Swillen A, Vorstman J (2011) Practical guidelines for managing patients with 22q11.2 deletion syndrome. J Pediatr 159:332–339
Vo OK, McNeil A, Vogt KS (2018) The psychosocial impact of 22q11 deletion syndrome on patients and families: a systematic review. Am J Med Genet 176A:2215–2225. https://doi.org/10.1002/ajmg.a.38673
Swillen A, Moss E, Duijff S (2018) Neurodevelopmental outcome in 22q11.2 deletion syndrome and management. Am J Med Genet 176A:2160–2166. https://doi.org/10.1002/ajmg.a.38709
Schneider M, Debanné M, Bassett AS et al (2014) Psychiatric disorders from childhood to adulthood in 22q11.2 deletion syndrome: results from the International Consortium on Brain and Behavior in 22q11.2 deletion syndrome. Am J Psychiatry 171:627–639
Vorstman JAS, Breetvelt EJ, Duijff SN et al (2015) Cognitive decline preceding the onset of psychosis in patients with 22q11.2 deletion syndrome. JAMA Psychiat 72(4):377–385. https://doi.org/10.1001/jamapsychiatry.2014.2671
Rutter M (1987) Psychosocial resilience and protective mechanisms. Am J Orthopsychiatry 57(3):316–331
Rutter M (2012) Resilience as a dynamic concept. Dev Psychopathol 24:335–344. https://doi.org/10.1017/S0954579412000028
Portzky M, Wagnild G, De Bacquer D, Audenaert K (2010) Psychometric evaluation of the Dutch Resilience Scale RS-nl on 3265 healthy participants: a confirmation of the association between age and resilience found in the Swedish version. Scand J Caring Sci 24:86–92. https://doi.org/10.1111/j.1471-6712.2010.00841.x
Ten Hove J, Rosenbaum P (2018) The concept of resilience in childhood disability: does the international classification of functioning, disability and health help us? Child Care Health Development 44:730–735. https://doi.org/10.1111/cch.12590
Smith-Osborne A, Whitehill Bolton K (2013) Assessing resilience: a review of measures across the life course. J Evidence-Based Social Work 10(2):111–126. https://doi.org/10.1080/15433714.2011.597305
Tang SX, Moberg PJ, Yi JJ et al (2018) Olfactory deficits and psychosis-spectrum symptoms in 22q11.2 deletion syndrome. Schizophr Res 202:113–119. https://doi.org/10.1016/j.schres.2018.07.012
Tang SX, Gur RE (2018) Longitudinal perspectives on the psychosis spectrum in 22q11.2 deletion syndrome. Am J Med Genet 176A:2192–2202. https://doi.org/10.1002/ajmg.a.38500
The WHOQOL Group (1998) Development of the World Health Organization WHOQOL-BREF quality of life assessment. Psychol Med 28:551–558
Looman WS, Thurmes AK, O’Conner-Von SK (2010) Quality of life among children with velocardiofacial syndrome. Cleft Palate Craniofac J 47(3):273–283. https://doi.org/10.1597/09-009.1
Joyce P, O’Rourke C, McDermott B, Heussler H (2017) Health-related quality of life in 22q11.2 deletion syndrome: the child’s perspective. J Paediatr Child Health 54:311–315. https://doi.org/10.1111/jpc.13746
Fung WLA, Butcher NJ, Costain G et al (2015) Practical guidelines for managing adults with 22q11.2 deletion syndrome. Genet Med 17(8):599–609. https://doi.org/10.1038/gim.2014.175
Haase JE (2004) The adolescent resilience model as a guide to interventions. J Pediatr Oncol Nurs 21(5):289–299. https://doi.org/10.1177/1043454204267922
Hofer A, Mizuno Y, Wartelsteiner F et al (2017) Quality of life in schizophrenia and bipolar disorder: the impact of symptomatic remission and resilience. Eur Psychiatry 46:42–47. https://doi.org/10.1016/j.eurpsy.2017.08.005
Lee D, Cha B, Park CS et al (2017) Effects of resilience on quality of life in patients with bipolar disorder. J Affect Disord 207:434–441. https://doi.org/10.1016/j.jad.2016.08.075
Portzky M (2008) RS-nl: Resilience Scale—Nederlandse versie. Harcourt Test Publishers, Amsterdam
Wagnild GM, Young HM (1993) Development and psychometric evaluation of the Resilience Scale. J Nurs Meas 1(2):165–178
Power MJ, Green AM, The WHOQOL-Dis Group (2010) Development of the WHOQOL disabilities module. Qual Life Res 19:571–584
Achenbach TM (1997) Young adult behavior checklist. ASEBA research center for children. Youth and Families, Burlington
Achenbach TM (1997) Young Adult Self Report. ASEBA research center for children. Youth and Families, Burlington
Chatterjee S, Hadi A (2012) Regression analysis by example, 5th edn. Wiley, Somerset, p 248
Fiksinski AM, Schneider M, Murphy CM et al (2018) Understanding the pediatric psychiatric phenotype of 22q11.2 deletion syndrome. Am J Med Genet 176(10):2182–2191. https://doi.org/10.1002/ajmg.a.40387
Hawthorne G, Herrman H, Murphy B (2006) Interpreting the WHOQOL-Brèf: preliminary population norms and effect sizes. Soc Indic Res 77:37–59. https://doi.org/10.1007/s11205-005-5552-1
Vergaelen E, Claes S, Kempke S, Swillen A (2017) High prevalence of fatigue in adults with a 22q11.2 deletion syndrome. Am J Med Genet 173:858–867. https://doi.org/10.1002/ajmg.a.38094
Wagnild GM, Young HM (1993) Development and psychometric evaluation of the Resilience Scale. J Nurs Meas Winter 1(2):165–178
Swillen A, Moss E, Duijff S (2018) Neurodevelopmental outcome in 22q112 deletion syndrome and management. Am J Med Genet 176(10):2160–2166. https://doi.org/10.1002/ajmg.a.38709
Prince-Embury S, Saklofske DH (2014) Resilience interventions for youth in diverse populations. Springer, New York
Acknowledgements
We thank all the participants and their parents/caregivers for their willingness to participate in this study and Ine Verheyen for collecting the data.
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This study was approved by the Research Medical Ethics Committee UZ / KU Leuven (Belgium), S52418.
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Van de Woestyne, K., Vandensande, A., Vansteelandt, K. et al. Resilience and quality of life in young adults with a 22q11.2 deletion syndrome: a patient’s perspective. Eur Child Adolesc Psychiatry 31, 1885–1894 (2022). https://doi.org/10.1007/s00787-021-01822-6
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DOI: https://doi.org/10.1007/s00787-021-01822-6