Literature DB >> 35930004

Sleep disturbance is a common feature of Kabuki syndrome.

Tyler Rapp1, Allison J Kalinousky2, Jennifer Johnson3, Hans Bjornsson2,4,5, Jacqueline Harris2,3.   

Abstract

Kabuki syndrome (KS) is a rare epigenetic disorder caused by heterozygous loss of function variants in either KMT2D (90%) or KDM6A (10%), both involved in regulation of histone methylation. While sleep disturbance in other Mendelian disorders of the epigenetic machinery has been reported, no study has been conducted on sleep in KS. This study assessed sleep in 59 participants with KS using a validated sleep questionnaire. Participants ranged in age from 4 to 43 years old with 86% of participants having a pathogenic variant in KMT2D. In addition, data on adaptive function, behavior, anxiety, and quality of life were collected using their respective questionnaires. Some form of sleep issue was present in 71% of participants, with night-waking, daytime sleepiness, and sleep onset delay being the most prevalent. Sleep dysfunction was positively correlated with maladaptive behaviors, anxiety levels, and decreasing quality of life. Sleep issues were not correlated with adaptive function. This study establishes sleep disturbance as a common feature of KS. Quantitative sleep measures may be a useful outcome measure for clinical trials in KS. Further, clinicians caring for those with KS should consider sleep dysfunction as an important feature that impacts overall health and well being in these patients.
© 2022 Wiley Periodicals LLC.

Entities:  

Keywords:  KDM6A; KMT2D; MLL2; epigenetics; neurodevelopmental

Mesh:

Substances:

Year:  2022        PMID: 35930004      PMCID: PMC9474613          DOI: 10.1002/ajmg.a.62921

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.578


  25 in total

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Authors:  Krista Specketer; Cyrus P Zabetian; Karen L Edwards; Lu Tian; Joseph F Quinn; Amie L Peterson-Hiller; Kathryn A Chung; Shu-Ching Hu; Thomas J Montine; Brenna A Cholerton
Journal:  J Clin Exp Neuropsychol       Date:  2019-06-10       Impact factor: 2.475

2.  Histone deacetylase inhibition rescues structural and functional brain deficits in a mouse model of Kabuki syndrome.

Authors:  Hans T Bjornsson; Joel S Benjamin; Li Zhang; Jacqueline Weissman; Elizabeth E Gerber; Yi-Chun Chen; Rebecca G Vaurio; Michelle C Potter; Kasper D Hansen; Harry C Dietz
Journal:  Sci Transl Med       Date:  2014-10-01       Impact factor: 17.956

3.  The Screen for Child Anxiety Related Emotional Disorders (SCARED): scale construction and psychometric characteristics.

Authors:  B Birmaher; S Khetarpal; D Brent; M Cully; L Balach; J Kaufman; S M Neer
Journal:  J Am Acad Child Adolesc Psychiatry       Date:  1997-04       Impact factor: 8.829

4.  Prevalence of sleep disorders and sleep behaviors in children and adolescents.

Authors:  J F Simonds; H Parraga
Journal:  J Am Acad Child Psychiatry       Date:  1982-07

5.  Sleep and behavior problems in school-aged children.

Authors:  M A Stein; J Mendelsohn; W H Obermeyer; J Amromin; R Benca
Journal:  Pediatrics       Date:  2001-04       Impact factor: 7.124

6.  Developmental Pathways among Adaptive Functioning and Externalizing and Internalizing Behavioral Problems: Cascades from Childhood into Adolescence.

Authors:  Marc H Bornstein; Chun-Shin Hahn; Joan T D Suwalsky
Journal:  Appl Dev Sci       Date:  2013

7.  Sleep behaviors and disorders in children and adolescents evaluated at psychiatric clinics.

Authors:  J F Simonds; H Parraga
Journal:  J Dev Behav Pediatr       Date:  1984-02       Impact factor: 2.225

8.  Characterization of sleep habits of children with Sotos syndrome.

Authors:  Christine F Stafford; Carrie Ward; Sally L D Ward; Pedro A Sanchez-Lara
Journal:  Am J Med Genet A       Date:  2021-04-24       Impact factor: 2.802

9.  Expanding the genotypic and phenotypic spectrum in a diverse cohort of 104 individuals with Wiedemann-Steiner syndrome.

Authors:  Sarah E Sheppard; Ian M Campbell; Margaret H Harr; Nina Gold; Dong Li; Hans T Bjornsson; Julie S Cohen; Jill A Fahrner; Ali Fatemi; Jacqueline R Harris; Catherine Nowak; Cathy A Stevens; Katheryn Grand; Margaret Au; John M Graham; Pedro A Sanchez-Lara; Miguel Del Campo; Marilyn C Jones; Omar Abdul-Rahman; Fowzan S Alkuraya; Jennifer A Bassetti; Katherine Bergstrom; Elizabeth Bhoj; Sarah Dugan; Julie D Kaplan; Nada Derar; Karen W Gripp; Natalie Hauser; A Micheil Innes; Beth Keena; Neslida Kodra; Rebecca Miller; Beverly Nelson; Malgorzata J Nowaczyk; Zuhair Rahbeeni; Shay Ben-Shachar; Joseph T Shieh; Anne Slavotinek; Andrew K Sobering; Mary-Alice Abbott; Dawn C Allain; Louise Amlie-Wolf; Ping Yee Billie Au; Emma Bedoukian; Geoffrey Beek; James Barry; Janet Berg; Jonathan A Bernstein; Cheryl Cytrynbaum; Brian Hon-Yin Chung; Sarah Donoghue; Naghmeh Dorrani; Alison Eaton; Josue A Flores-Daboub; Holly Dubbs; Carolyn A Felix; Chin-To Fong; Jasmine Lee Fong Fung; Balram Gangaram; Amy Goldstein; Rotem Greenberg; Thoa K Ha; Joseph Hersh; Kosuke Izumi; Staci Kallish; Elijah Kravets; Pui-Yan Kwok; Rebekah K Jobling; Amy E Knight Johnson; Jessica Kushner; Bo Hoon Lee; Brooke Levin; Kristin Lindstrom; Kandamurugu Manickam; Rebecca Mardach; Elizabeth McCormick; D Ross McLeod; Frank D Mentch; Kelly Minks; Colleen Muraresku; Stanley F Nelson; Patrizia Porazzi; Pavel N Pichurin; Nina N Powell-Hamilton; Zoe Powis; Alyssa Ritter; Caleb Rogers; Luis Rohena; Carey Ronspies; Audrey Schroeder; Zornitza Stark; Lois Starr; Joan Stoler; Pim Suwannarat; Milen Velinov; Rosanna Weksberg; Yael Wilnai; Neda Zadeh; Dina J Zand; Marni J Falk; Hakon Hakonarson; Elaine H Zackai; Fabiola Quintero-Rivera
Journal:  Am J Med Genet A       Date:  2021-03-30       Impact factor: 2.578

Review 10.  Memory consolidation during sleep and adult hippocampal neurogenesis.

Authors:  Iyo Koyanagi; Katherine G Akers; Pablo Vergara; Sakthivel Srinivasan; Takeshi Sakurai; Masanori Sakaguchi
Journal:  Neural Regen Res       Date:  2019-01       Impact factor: 5.135

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