Cooperative Parent-Mediated Therapy in Children with Fragile X Syndrome and Williams Beuren Syndrome: A Pilot RCT Study of a Transdiagnostic Intervention-Preliminary Data
Abstract
:1. Introduction
2. Materials and Methods
2.1. Participants
2.2. Procedure
2.3. Measures
2.3.1. Child Assessment
Socio-Communicative Skills
Language Skills
Adaptive Functioning
Emotional and Behavioral Problems
Clinical Global Impression
Cognitive/Developmental Assessment
2.3.2. Parent Assessment
Parenting Stress
Quality of Life
2.4. Interventions
2.4.1. Treatment as Usual
2.4.2. Cooperative Parent-Mediated Therapy—CPMT
2.5. Statistical Analysis
3. Results
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
References
- American Psychiatric Association. DSM 5; American Psychiatric Association: Washington, DC, USA, 2013; p. 70. [Google Scholar]
- Elsabbagh, M.; Divan, G.; Koh, Y.J.; Kim, Y.S.; Kauchali, S.; Marcín, C.; Fombonne, E. Global prevalence of autism and other pervasive developmental disorders. Autism Res. 2012, 5, 160–179. [Google Scholar] [CrossRef] [Green Version]
- Maenner, M.J.; Shaw, K.A.; Baio, J. Prevalence of autism spectrum disorder among children aged 8 years—Autism and developmental disabilities monitoring network, 11 sites, United States, 2016. MMWR Surveill. Summ. 2020, 69, 1. [Google Scholar] [CrossRef] [PubMed]
- Caglayan, A.O. Genetic causes of syndromic and non-syndromic autism. Dev. Med. Child Neurol. 2010, 52, 130–138. [Google Scholar] [CrossRef] [PubMed]
- Vicari, S.; Napoli, E.; Cordeddu, V.; Menghini, D.; Alesi, V.; Loddo, S.; Tartaglia, M. Copy number variants in autism spectrum disorders. Prog. Neuro-Psychopharmacol. Biol. Psychiatry 2019, 92, 421–427. [Google Scholar] [CrossRef]
- Gillberg, C.; Rasmussen, P. Brief report: Four case histories and a literature review of Williams syndrome and autistic behavior. J. Autism Dev. Disord. 1994, 24, 381–393. [Google Scholar] [CrossRef]
- Lincoln, A.J.; Searcy, Y.M.; Jones, W.; Lord, C. Social interaction behaviors discriminate young children With Autism and Williams syndrome. J. Am. Acad. Child Adolesc. Psychiatry 2007, 46, 323–331. [Google Scholar] [CrossRef] [Green Version]
- Klein-Tasman, B.P.; Mervis, C.B.; Lord, C.; Phillips, K.D. Socio-communicative deficits in young children with Williams syndrome: Performance on the Autism Diagnostic Observation Schedule. Child Neuropsychol. 2007, 13, 444–467. [Google Scholar] [CrossRef] [PubMed]
- Klein-Tasman, B.P.; Phillips, K.D.; Lord, C.E.; Mervis, C.B.; Gallo, F. Overlap with the autism spectrum in young children with Williams syndrome. J. Dev. Behav. Pediatr. 2009, 30, 289. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Klein-Tasman, B.P.; van der Fluit, F.; Mervis, C.B. Autism spectrum symptomatology in children with Williams syndrome who have phrase speech or fluent language. J. Autism Dev. Disord. 2018, 48, 3037–3050. [Google Scholar] [CrossRef] [PubMed]
- Stone, W.L.; Basit, H.; Los, E. Fragile X Syndrome; StatPearls Publishing: Treasure Island, FL, USA, 2021. [Google Scholar]
- Kaufmann, W.E.; Kidd, S.A.; Andrews, H.F.; Budimirovic, D.B.; Esler, A.; Haas-Givler, B.; Berry-Kravis, E. Autism spectrum disorder in fragile X syndrome: Cooccurring conditions and current treatment. Pediatrics 2017, 139, 194. [Google Scholar] [CrossRef] [Green Version]
- Crawford, D.C.; Acuña, J.M.; Sherman, S.L. FMR1 and the fragile X syndrome: Human genome epidemiology review. Genet. Med. 2001, 3, 359–371. [Google Scholar] [CrossRef] [Green Version]
- Tassone, F.; Greco, C.M.; Hunsaker, M.R.; Seritan, A.L.; Berman, R.F.; Gane, L.W.; Hagerman, R.J. Neuropathological, clinical and molecular pathology in female fragile X premutation carriers with and without FXTAS. Genes Brain Behav. 2012, 11, 577–585. [Google Scholar] [CrossRef] [Green Version]
- Boyle, L.; Kaufmann, W.E. The behavioral phenotype of FMR1 mutations. Am. J. Med. Genet. Part C Semin. Med Genet. 2010, 154, 469–476. [Google Scholar] [CrossRef] [PubMed]
- Hall, S.S.; Lightbody, A.A.; Hirt, M.; Rezvani, A.; Reiss, A.L. Autism in fragile X syndrome: A category mistake? J. Am. Acad. Child Adolesc. Psychiatry 2010, 49, 921–933. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Strømme, P.; Bjørnstad, P.G.; Ramstad, K. Prevalence estimation of Williams syndrome. J. Child Neurol. 2002, 17, 269–271. [Google Scholar] [CrossRef] [PubMed]
- Osborne, L.R.; Li, M.; Pober, B.; Chitayat, D.; Bodurtha, J.; Mandel, A.; Scherer, S.W. A 1.5 million–base pair inversion polymorphism in families with Williams-Beuren syndrome. Nat. Genet. 2001, 29, 321–325. [Google Scholar] [CrossRef] [PubMed]
- Järvinen-Pasley, A.; Bellugi, U.; Reilly, J.; Debra, L.; Galaburda, A.; Reiss, A.L.; Korenberg, J.R. Defining the social phenotype in Williams syndrome: A model for linking gene, the brain, and behavior. Dev. Psychopathol. 2008, 20, 1–35. [Google Scholar] [CrossRef] [Green Version]
- Alfieri, P.; Menghini, D.; Marotta, L.; De Peppo, L.; Ravà, L.; Salvaguardia, F.; Vicari, S. A comparison between linguistic skills and socio-communicative abilities in Williams syndrome. J. Intellect. Disabil. Res. 2017, 61, 866–868. [Google Scholar] [CrossRef]
- Vivanti, G.; Hamner, T.; Lee, N.R. Neurodevelopmental disorders affecting sociability: Recent research advances and future directions in autism spectrum disorder and Williams syndrome. Curr. Neurol. Neurosci. Rep. 2018, 18, 94. [Google Scholar] [CrossRef]
- Royston, R.; Howlin, P.; Waite, J.; Oliver, C. Anxiety disorders in Williams syndrome contrasted with intellectual disability and the general population: A systematic review and meta-analysis. J. Autism Dev. Disord. 2017, 20, 3765–3777. [Google Scholar] [CrossRef] [Green Version]
- Järvinen, A.; Korenberg, J.R.; Bellugi, U. The social phenotype of Williams syndrome. Curr. Opin. Neurobiol. 2013, 23, 414–422. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Serrano-Juárez, C.A.; Prieto-Corona, B.; Rodríguez-Camacho, M.; Venegas-Vega, C.A.; Yáñez-Téllez, M.G.; Silva-Pereyra, J.; Miranda, M.A.D.L. An Exploration of Social Cognition in Children with Different Degrees of Genetic Deletion in Williams Syndrome. J. Autism Dev. Disord. 2021, 51, 1695–1704. [Google Scholar] [CrossRef] [PubMed]
- Schultz, R.T.; Grelotti, D.J.; Pober, B. Genetics of childhood disorders: XXVI. Williams syndrome and brain–behavior relationships. J. Am. Acad. Child Adolesc. Psychiatry 2001, 40, 606–609. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Alfieri, P.; Scibelli, F.; Digilio, M.C.; Novello, R.L.; Caciolo, C.; Valeri, G.; Vicari, S. Comparison of Adaptive Functioning in Children with Williams Beuren Syndrome and Autism Spectrum Disorder: A Cross-Syndrome Study. Autism Res. 2021, 14, 748–758. [Google Scholar] [CrossRef]
- Hamner, T.; Raitano Lee, N.; Hocking, D.R.; Vivanti, G. Shared and syndrome-specific adaptive difficulties in preschoolers with Williams syndrome and autism spectrum disorder: A cross-syndrome study. J. Intellect. Disabil. Res. 2019, 63, 1305–1311. [Google Scholar] [CrossRef]
- Monlux, K.D.; Pollard, J.S.; Rodriguez, A.Y.B.; Hall, S.S. Telehealth delivery of function-based behavioral treatment for problem behaviors exhibited by boys with fragile X syndrome. J. Autism Dev. Disord. 2019, 49, 246. [Google Scholar] [CrossRef]
- Hall, S.S.; Monlux, K.D.; Rodriguez, A.B.; Jo, B.; Pollard, J.S. Telehealth-enabled behavioral treatment for problem behaviors in boys with fragile X syndrome: A randomized controlled trial. J. Neurodev. Disord. 2020, 12, 31. [Google Scholar] [CrossRef] [PubMed]
- Moskowitz, L.J.; Jones, E.A. Uncovering the evidence for behavioral interventions with individuals with fragile X syndrome: A systematic review. Res. Dev. Disabil. 2015, 38, 223–241. [Google Scholar] [CrossRef]
- Vismara, L.A.; McCormick, C.E.; Shields, R.; Hessl, D. Extending the parent-delivered Early Start Denver Model to young children with fragile X syndrome. J. Autism Dev. Disord. 2019, 49, 1250–1266. [Google Scholar] [CrossRef]
- Bearss, K.; Burrell, T.L.; Stewart, L.; Scahill, L. Parent training in autism spectrum disorder: What’s in a name? Clin. Child Family Psychol. Rev. 2015, 18, 170–182. [Google Scholar] [CrossRef]
- Oono, I.P.; Honey, E.J.; McConachie, H. Parent-mediated early intervention for young children with autism spectrum disorders (ASD). Evid.-Based Child Health A Cochrane Rev. J. 2013, 8, 2380–2479. [Google Scholar] [CrossRef]
- Green, J.; Charman, T.; McConachie, H.; Aldred, C.; Slonims, V.; Howlin, P.; PACT Consortium. Parent-mediated communication-focused treatment in children with autism (PACT): A randomised controlled trial. Lancet 2010, 375, 2152–2160. [Google Scholar] [CrossRef] [Green Version]
- Pickles, A.; Le Couteur, A.; Leadbitter, K.; Salomone, E.; Cole-Fletcher, R.; Tobin, H.; Green, J. Parent-mediated social communication therapy for young children with autism (PACT): Long-term follow-up of a randomised controlled trial. Lancet 2016, 388, 2501–2509. [Google Scholar] [CrossRef] [Green Version]
- Nevill, R.E.; Lecavalier, L.; Stratis, E.A. Meta-analysis of parent-mediated interventions for young children with autism spectrum disorder. Autism 2018, 22, 84–98. [Google Scholar] [CrossRef] [PubMed]
- Althoff, C.E.; Dammann, C.P.; Hope, S.J.; Ausderau, K.K. Parent-mediated interventions for children with autism spectrum disorder: A systematic review. Am. J. Occup. Ther. 2019, 73, 7303205010. [Google Scholar] [CrossRef] [PubMed]
- Valeri, G.; Casula, L.; Menghini, D.; Amendola, F.A.; Napoli, E.; Pasqualetti, P.; Vicari, S. Cooperative parent-mediated therapy for Italian preschool children with autism spectrum disorder: A randomized controlled trial. Eur. Child Adolesc. Psychiatry 2020, 29, 935–946. [Google Scholar] [CrossRef] [PubMed]
- Thurman, A.J.; Potter, L.A.; Kim, K.; Tassone, F.; Banasik, A.; Potter, S.N.; Abbeduto, L. Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome. J. Neurodev. Disord. 2020, 12, 12. [Google Scholar] [CrossRef]
- Schreibman, L.; Dawson, G.; Stahmer, A.C.; Landa, R.; Rogers, S.J.; McGee, G.G.; Halladay, A. Naturalistic developmental behavioral interventions: Empirically validated treatments for autism spectrum disorder. J. Autism Dev. Disord. 2015, 45, 2411–2428. [Google Scholar] [CrossRef] [Green Version]
- Tomasello, M. Origins of Human Communication; MIT Press: Cambridge, MA, USA, 2008. [Google Scholar]
- Mundy, P.C. Autism and Joint Attention: Development, Neuroscience, and Clinical Fundamentals; Guilford Publications: New York, NY, USA, 2016. [Google Scholar]
- Tomasello, M. Why We Cooperate; MIT Press: Cambridge, MA, USA, 2009. [Google Scholar]
- Tomasello, M. Becoming Human: A Theory of Ontogeny; Harvard University Press: Cambridge, MA, USA, 2019. [Google Scholar]
- Colombi, C.; Liebal, K.; Tomasello, M.; Young, G.; Warneken, F.; Rogers, S.J. Examining correlates of cooperation in autism: Imitation, joint attention, and understanding intentions. Autism 2009, 13, 143–163. [Google Scholar] [CrossRef] [Green Version]
- Liebal, K.; Colombi, C.; Rogers, S.J.; Warneken, F.; Tomasello, M. Helping and cooperation in children with autism. J. Autism Dev. Disord. 2008, 38, 224–238. [Google Scholar] [CrossRef] [Green Version]
- Astle, D.E.; Holmes, J.; Kievit, R.; Gathercole, S.E. Annual Research Review: The transdiagnostic revolution in neurodevelopmental disorders. J. Child Psychol. Psychiatry 2021. Epub ahead of print. [Google Scholar] [CrossRef] [PubMed]
- Stessman, H.A.; Xiong, B.; Coe, B.P.; Wang, T.; Hoekzema, K.; Fenckova, M.; Kvarnung, M.; Gerdts, J.; Trinh, S.; Cosemans, N.; et al. Targeted sequencing identifies 91 neurodevelopmental-disorder risk genes with autism and developmental-disability biases. Nat. Genet. 2017, 49, 515. [Google Scholar] [CrossRef] [PubMed]
- Myers, S.M.; Challman, T.D.; Bernier, R.; Bourgeron, T.; Chung, W.K.; Constantino, J.N.; Eichler, E.E.; Jacquemont, S.; Miller, D.T.; Mitchell, K.J.; et al. Insufficient evidence for “autism-specific” genes. Am. J. Hum. Genet. 2020, 106, 587–595. [Google Scholar] [CrossRef] [PubMed]
- Georgiades, S.; Szatmari, P.; Zwaigenbaum, L.; Duku, E.; Bryson, S.; Roberts, W.; Goldberg, J.; Mahoney, W. Structure of the autism symptom phenotype: A proposed multidimensional model. J. Am. Acad. Child Adolesc. Psychiatry 2007, 46, 188–196. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Bonifacio, S.; Girolametto, L.; Bulligan, M.; Callegari, M.; Vignola, S.; Zocconi, E. Assertive and responsive conversational skills of Italian—speaking late talkers. Int. J. Lang. Commun. Disord. 2007, 42, 607–623. [Google Scholar] [CrossRef]
- Guy, W. CGI. Clinical global impressions. In ECDEU Assessment Manual for Psychopharmacology; US Department of Heath, Education, and Welfare Public Health Service Alcohol, Drug Abuse, and Mental Health Administration: Rockville, MD, USA, 1976. [Google Scholar]
- Mundy, P.; Delgado, C.; Block, J.; Venezia, M.; Hogan, A.; Seibert, J. Early Social Communication Scales (ESCS); University of Miami: Coral Gables, FL, USA, 2003. [Google Scholar]
- Caselli, M.C.; Bello, A.; Rinaldi, P.; Stefanini, S.; Pasqualetti, P. Il primo vocabolario del bambino: Gesti, parole e frasi. In Forme Lunghe E Forme Brevi Del Questionario E Valori Di Riferimento Per La Fascia; Franco Angeli: Milano, Italy, 2015. [Google Scholar]
- Fenson, L. MacArthur-Bates Communicative Development Inventories; Paul H. Brookes Publishing Company: Baltimore, MD, USA, 2007. [Google Scholar]
- Sparrow, S.S.; Cicchetti, D.V.; Balla, D.A. Vineland Adaptive Behavior Scales, Second Edition—Survey Forms Manual; American Guidance Service: Circle Pines, MN, USA, 2005. [Google Scholar]
- Achenbach, T.M.; Ruffle, T.M. The Child Behavior Checklist and related forms for assessing behavioral/emotional problems and competencies. Pediatrics Rev. 2000, 21, 265–271. [Google Scholar] [CrossRef]
- Hoffmann, W.; Weber, L.; König, U.; Becker, K.; Kamp-Becker, I. The role of the CBCL in the assessment of autism spectrum disorders: An evaluation of symptom profiles and screening characteristics. Res. Autism Spectr. Disord. 2016, 27, 44–53. [Google Scholar] [CrossRef]
- Roid, G.H.; Koch, C. Leiter-3: Nonverbal cognitive and neuropsychological assessment. In Handbook of Nonverbal Assessment; Springer: Cham, Switzerland, 2017; pp. 127–150. [Google Scholar]
- Green, E.; Stroud, L.; Bloomfield, S.; Cronje, J.; Foxcroft, C.; Hurter, K.; McAlinden, P. Griffith III-Griffths Scale of Child Developmental; Hogrefe: Florence, Italy, 2016. [Google Scholar]
- Abidin, R.; Flens, J.R.; Austin, W.G. The Parenting Stress Index; Lawrence Erlbaum Associates Publishers: Mahwah, NJ, USA, 2006. [Google Scholar]
- Whoqol Group. Development of the World Health Organization WHOQOL-BREF quality of life assessment. Psychol. Med. 1998, 28, 551–558. [Google Scholar] [CrossRef] [Green Version]
- Goedeke, S.; Shepherd, D.; Landon, J.; Taylor, S. How perceived support relates to child autism symptoms and care-related stress in parents caring for a child with autism. Res. Autism Spectr. Disord. 2019, 60, 36–47. [Google Scholar] [CrossRef]
- Conner, C.M.; White, S.W. Stress in mothers of children with autism: Trait mindfulness as a protective factor. Res. Autism Spectr. Disord. 2014, 8, 617–624. [Google Scholar] [CrossRef]
- Scibelli, F.; Fucà, E.; Guerrera, S.; Lupi, E.; Alfieri, P.; Valeri, G.; Vicari, S. Clinical and individual features associated with maternal stress in young adolescents with autism spectrum disorder. Autism Res. 2021, 14, 1935–1947. [Google Scholar] [CrossRef] [PubMed]
- Cuthbert, B.N.; Insel, T.R. Toward the future of psychiatric diagnosis: The seven pillars of RDoC. BMC Med. 2013, 11, 126. [Google Scholar] [CrossRef] [PubMed] [Green Version]
Variable | Control Group Median (IQR) (Min–Max Range) | Experimental Group Median (IQR) (Min–Max Range) | p-Value |
---|---|---|---|
Sex (male:female) | 3:4 | 3:2 | 1.000 |
Age | 4.9 (3.1–5.6) [3.1–6.3] | 5.4 (4.9–6.1) [3.2–6.5] | 0.329 |
IQ/DQ | 49 (49–71) [49–78] | 56 (53–76) [50–78] | 0.160 |
Clinical Global Impression-Severity | 5 (4–6) [4–7] | 5 (4–5) [4–6] | 0.669 |
SCSRS Assertivity | 37 (27–51) [23–65] | 41 (34–56) [30–68] | 0.723 |
SCSRS Responsivity | 18 (15–41) [14–56] | 34 (24–37) [20–45] | 0.258 |
Level of education (mother) | 4 (3–5) [2–5] | 5 (4–5) [4–6] | 0.202 |
Employment (mother) | 28.6% (Employed) 71.4% (Unemployed) | 80.0% (Employed) 20.0% (Unemployed) | 0.242 |
Level of education (father) | 4 (2–5) [2–5] | 5 (4–5) [4–5] | 0.144 |
Employment (father) | 85.7% (Employed) 14.3% (Unemployed) | 100.0% (Employed) - (Unemployed) | 1.000 |
Variable | Control Group Median (IQR) (Min–Max Range) | Experimental Group Median (IQR) (Min–Max Range) | |||||
---|---|---|---|---|---|---|---|
T0 | T1 | p-Value | T0 | T1 | p-Value | ||
SCSRS | Assertivity | 37 (27–51) [23–65] | 53 (37–56) [34–67] | 0.091 | 41 (34–56) [30–68] | 65 (49–66) [39–67] | 0.080 |
Responsivity | 18 (15–41) [14–56] | 37 (25–41) [17–49] | 0.237 | 34 (24–37) [20–45] | 39 (30–43) [22–48] | 0.042 * | |
ESCS | Frequence Initiate Joint Attention (IJA) | 21 (0–30) [0–66] | 30 (13–36) [10–85] | 0.063 | 12 (11–30) [2–62] | 48 (12–52) [5–62] | 0.225 |
Ratio Higher/Total Level IJA | 0.14 (0–0.46) [0–0.75] | 0.33 (0.21–0.47) [0.00–0.92] | 0.203 | 0.31 (0.08–0.48) [0.00–0.50] | 0.27 (0.20–0.33) [0.16–0.38] | 0.715 | |
Higher Level Response Joint Attention | 100 (0–100) [0–100] | 87.5 (87.5–100) [0–100] | 0.856 | 87.5 (75–100) [12.5–100] | 100 (100–100) [37.5–100] | 0.572 | |
Frequence Initiate Behavioral Request (IBR) | 20 (15–38) [5–38] | 29 (29–34) [19–39] | 0.128 | 30 (24–31) [2–33] | 32 (31–34) [31–37] | 0.104 | |
Ratio Higher/Total Level IBR | 0.37 (0.16–0.4) [0.14–0.71] | 0.31 (0.24–0.34) [0.05–0.52] | 0.499 | 0.36 (0.29–0.42) [0–0.5] | 0.41 (0.34–0.48) [0.32–0.56] | 0.686 | |
Responding Behavioral Requests | 75 (18–100) [0.7–100] | 100 (60–100) [56–100] | 0.212 | 100 (0–100) [0–100] | 100 (84–100) [0–100] | 0.317 | |
Initiate Social Interaction | 2 (0–2) [0–4] | 3 (2–3) [0–3] | 0.094 | 2 (2–4) [0–4] | 3 (2–4) [2–4] | 0.160 | |
Total Responding social interaction | 24 (10–45) [8–46] | 38 (10–43) [6–49] | 0.612 | 26 (20–39) [17–49] | 37 (32–46) [25–48] | 0.144 | |
VABS II | Communication | 51 (32–62) [32–67] | 50 (28–54) [21–62] | 0.248 | 54 (52–57) [20–62] | 53 (43–65) [20–65] | 0.586 |
Daily Living | 58 (49–66) [48–66] | 56 (50–68) [42–73] | 0.916 | 62 (57–74) [37–77] | 58 (52–65) [35–65] | 0.080 | |
Socialization | 64.5 (58–70) [58–83] | 66 (55–72) [48–75] | 0.035 * | 73 (73–81) [42–82] | 72 (61–74) [38–88] | 0.588 | |
Expressive | 6.5 (3–9) [3–9] | 8 (7–9) [2–9] | 0.665 | 7 (6–7) [3–7] | 8 (8–8) [2–10] | 0.131 | |
Receptive | 3.5 (2–7) [1–10] | 5 (3–6) [1–9] | 0.831 | 7 (5–9) [1–11] | 9 (2–10) [1–10] | 0.891 | |
CBCL | Withdrawal | 63 (52–79) [50–85] | 63 (52–79) [50–85] | 0.914 | 62 (52–63) [51–67] | 51 (50–60) [50–68] | 0.225 |
Internalizing Problems | 62 (43–73) [29–79] | 56 (45–71) [37–80] | 0.459 | 62 (49–63) [33–69] | 58 (37–62) [29–62] | 0.131 | |
Externalizing problems | 63 (51–67) [44–73] | 57 (51–67) [48–73] | 0.398 | 53 (50–60) [40–65] | 57 (51–67) [48–73] | 0.586 | |
Total problems | 59 (51–74) [39–79] | 57 (50–74) [43–77] | 0.866 | 63 (50–66) [35–68] | 59 (40–67) [37–67] | 0.686 | |
PVB | Word Production | 26 (1–258) [0–661] | 111 (39–425) [0–676] | 0.031 * | 295 (229–243) [0–664] | 517 (287–642) [0–644] | 0.586 |
CGI-I | Clinical Global Impression—Improvement | - | 4 (3–4) [1–4] | - | 2 (1–2) [1, 2] | 0.030 *,° | |
PSI-SF Mother | Parental Distress | 65 (10–75) [5–90] | 55 (25–85) [12–95] | 0.351 | 80 (25–80) [5–100] | 85 (55–100) [5–100] | 0.339 |
Parent-Child Dysfunctional Interaction | 75 (45–95) [10–95] | 65 (30–95) [25–100] | 0.307 | 45 (40–65) [10–100] | 75 (75–80) [35–100] | 0.174 | |
Difficult Child | 95 (80–100) [25–100] | 90 (70–95) [25–100] | 0.193 | 40 (25–90) [10–100] | 80 (25–95) [5–95] | 0.891 | |
PSI-SF Father | Parental Distress | 60 (5–75) [1–90] | 20 (5–45) [1–95] | 0.344 | 50 (10–55) [1–100] | 65 (25–85) [5–100] | 0.174 |
Parent-Child Dysfunctional Interaction | 75 (30–95) [25–95] | 60 (35–95) [5–100] | 0.732 | 60 (20–85) [1–95] | 70 (40–80) [35–95] | 0.174 | |
Difficult Child | 90 (35–100) [30–100] | 95 (30–95) [10–95] | 0.146 | 50 (15–95) [1–100] | 30 (30–95) [5–95] | 0.586 | |
WhoQol Mother | Physical health | 29 (26–30) [20–33] | 27 (24–29) [23–29] | 0.444 | 24 (24–25) [20–27] | 23 (22–28) [19–30] | 1.000 |
Psychological health | 23 (19–26) [16–29] | 20 (18–22) [18–23] | 0.149 | 18 (18–19) [15–23] | 18 (15–19) [14–22] | 0.160 | |
Social relationships | 13 (12–14) [11–15] | 12 (11–12) [10–16] | 0.227 | 11 (10–13) [6–14] | 10 (8–10) [7–12] | 0.172 | |
Environment | 26 (21–33) [20–36] | 26 (24–31) [21–31] | 0.796 | 27 (25–28) [21–28] | 24 (21–32) [20–32] | 1.000 | |
WhoQol Father | Physical health | 31 (30–32) [22–33] | 29 (29–32) [27–33] | 0.798 | 25 (24–29) [21–34] | 25 (23–29) [18–31] | 0.172 |
Psychological health | 25 (21–27) [17–28] | 20 (18–22) [18–23] | 0.669 | 20 (19–21) [17–25] | 21 (20–23) [15–24] | 0.786 | |
Social relationships | 11 (10–13) [8–14] | 13 (12–14) [11–15] | 0.073 | 11 (10–12) [8–14] | 9 (9–10) [9–12] | 0.098 | |
Environment | 29 (27–32) [23–36] | 31 (27–33) [26–35] | 0.444 | 28 (25–28) [24–29] | 25 (25–26) [20–29] | 0.174 |
Publisher’s Note: MDPI stays neutral with regard to jurisdictional claims in published maps and institutional affiliations. |
© 2021 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
Share and Cite
Alfieri, P.; Scibelli, F.; Casula, L.; Piga, S.; Napoli, E.; Valeri, G.; Vicari, S. Cooperative Parent-Mediated Therapy in Children with Fragile X Syndrome and Williams Beuren Syndrome: A Pilot RCT Study of a Transdiagnostic Intervention-Preliminary Data. Brain Sci. 2022, 12, 8. https://doi.org/10.3390/brainsci12010008
Alfieri P, Scibelli F, Casula L, Piga S, Napoli E, Valeri G, Vicari S. Cooperative Parent-Mediated Therapy in Children with Fragile X Syndrome and Williams Beuren Syndrome: A Pilot RCT Study of a Transdiagnostic Intervention-Preliminary Data. Brain Sciences. 2022; 12(1):8. https://doi.org/10.3390/brainsci12010008
Chicago/Turabian StyleAlfieri, Paolo, Francesco Scibelli, Laura Casula, Simone Piga, Eleonora Napoli, Giovanni Valeri, and Stefano Vicari. 2022. "Cooperative Parent-Mediated Therapy in Children with Fragile X Syndrome and Williams Beuren Syndrome: A Pilot RCT Study of a Transdiagnostic Intervention-Preliminary Data" Brain Sciences 12, no. 1: 8. https://doi.org/10.3390/brainsci12010008
APA StyleAlfieri, P., Scibelli, F., Casula, L., Piga, S., Napoli, E., Valeri, G., & Vicari, S. (2022). Cooperative Parent-Mediated Therapy in Children with Fragile X Syndrome and Williams Beuren Syndrome: A Pilot RCT Study of a Transdiagnostic Intervention-Preliminary Data. Brain Sciences, 12(1), 8. https://doi.org/10.3390/brainsci12010008