Literature DB >> 23810129

47,XYY syndrome: clinical phenotype and timing of ascertainment.

Martha Zeger Bardsley1, Karen Kowal, Carly Levy, Ania Gosek, Natalie Ayari, Nicole Tartaglia, Najiba Lahlou, Breanna Winder, Shannon Grimes, Judith L Ross.   

Abstract

OBJECTIVE: To describe auxologic, physical, and behavioral features in a large cohort of males with 47,XYY (XYY), ages newborn to young adult. STUDY
DESIGN: This is a cross-sectional descriptive study of male subjects with XYY who were evaluated at 1 of 2 specialized academic sites. Subjects underwent a history, physical examination, laboratory testing, and cognitive/behavioral evaluation.
RESULTS: In 90 males with XYY (mean age 9.6 ± 5.3 years [range 0.5-36.5]), mean height SD was above average (1.0 ± 1.2 SD). Macrocephaly (head circumference >2 SD) was noted in 28/84 (33%), hypotonia in 57/90 (63%), clinodactyly in 47/90 (52%), and hypertelorism in 53/90 (59%). There was testicular enlargement for age (>2 SD) in 41/82 (50%), but no increase in genital anomalies. No physical phenotypic differences were seen in boys diagnosed prenatally vs postnatally. Testosterone, luteinizing hormone, and follicle stimulating hormone levels were in the normal range in most boys. There was an increased incidence of asthma, seizures, tremor, and autistic spectrum disorder (ASD) compared with the general population rates. Prenatally diagnosed boys scored significantly better on cognitive testing and were less likely to be diagnosed with ASD (P < .01).
CONCLUSIONS: The XYY phenotype commonly includes tall stature, macrocephaly, macroorchidism, hypotonia, hypertelorism, and tremor. Physical phenotypic features were similar in boys diagnosed prenatally vs postnatally. Prenatal diagnosis was associated with higher cognitive function and less likelihood of an ASD diagnosis.
Copyright © 2013 Mosby, Inc. All rights reserved.

Entities:  

Keywords:  47,XYY; ADHD; ADI-R; AMH; ASD; Anti-mullerian hormone; Attention deficit hyperactivity disorder; Autism Diagnostic Interview–Revised; Autistism spectrum disorder; BMI; Body mass index; CBCL; CDI-2; Child Behavior Checklist; Children's Depression Inventory–Second Edition; FSH; FSIQ; Follicle stimulating hormone; Full scale IQ; HC; Head circumference; LH; Luteinzing hormone; SCQ; SES; SRS; Social Communicative Questionnaire; Social Responsiveness Scale; Socioeconomic status; WC; Waist circumference; XYY

Mesh:

Year:  2013        PMID: 23810129      PMCID: PMC4097881          DOI: 10.1016/j.jpeds.2013.05.037

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  37 in total

1.  Psychological studies of ten patients with the XYY syndrome.

Authors:  A L Christensen; J Nielsen
Journal:  Br J Psychiatry       Date:  1973-08       Impact factor: 9.319

Review 2.  Behavioral implications of the human XYY genotype.

Authors:  E B Hook
Journal:  Science       Date:  1973-01-12       Impact factor: 47.728

3.  XYY with abnormal genitalia.

Authors:  G Carakushansky; R L Neu; L I Gardner
Journal:  Lancet       Date:  1968-11-23       Impact factor: 79.321

4.  Testosterone excretion rates in normal males and males with an XYY complement.

Authors:  B T Rudd; O M Galal; M D Casey
Journal:  J Med Genet       Date:  1968-12       Impact factor: 6.318

5.  Sex chromosomes and the brain: a study of neuroanatomy in XYY syndrome.

Authors:  Daniel M Bryant; Fumiko Hoeft; Song Lai; John Lackey; David Roeltgen; Judith Ross; Allan L Reiss
Journal:  Dev Med Child Neurol       Date:  2012-10-12       Impact factor: 5.449

6.  Autism, language and communication in children with sex chromosome trisomies.

Authors:  Dorothy V M Bishop; Patricia A Jacobs; Katherine Lachlan; Diana Wellesley; Angela Barnicoat; Patricia A Boyd; Alan Fryer; Prisca Middlemiss; Sarah Smithson; Kay Metcalfe; Deborah Shears; Victoria Leggett; Kate Nation; Gaia Scerif
Journal:  Arch Dis Child       Date:  2010-07-23       Impact factor: 3.791

7.  Behavioral and social phenotypes in boys with 47,XYY syndrome or 47,XXY Klinefelter syndrome.

Authors:  Judith L Ross; David P Roeltgen; Harvey Kushner; Andrew R Zinn; Allan Reiss; Martha Zeger Bardsley; Elizabeth McCauley; Nicole Tartaglia
Journal:  Pediatrics       Date:  2012-03-12       Impact factor: 7.124

8.  The XYY syndrome: a follow-up study on 38 boys.

Authors:  M Geerts; J Steyaert; J P Fryns
Journal:  Genet Couns       Date:  2003

9.  Fifty-one prenatally diagnosed children and adolescents with sex chromosome abnormalities.

Authors:  Mary G Linden; Bruce G Bender
Journal:  Am J Med Genet       Date:  2002-06-01

10.  Early manifestations in a cohort of children prenatally diagnosed with 47,XYY. Role of multidisciplinary counseling for parental guidance and prevention of aggressive behavior.

Authors:  Faustina Lalatta; Emanuela Folliero; Ugo Cavallari; Marina Di Segni; Barbara Gentilin; Roberto Fogliani; Donatella Quagliarini; Paola Vizziello; Federico Monti; Luigi Gargantini
Journal:  Ital J Pediatr       Date:  2012-10-03       Impact factor: 2.638

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  45 in total

1.  A non-mosaic isodicentric Y chromosome resulting from breakage and fusion at the Yq pseudo-autosomal region in a fetus.

Authors:  Long-Ching Kuan; Mei-Tsz Su; Ming Chen; Pao-Lin Kuo; Tsung-Cheng Kuo
Journal:  J Assist Reprod Genet       Date:  2013-10-25       Impact factor: 3.412

2.  Y chromosome gene copy number and lack of autism phenotype in a male with an isodicentric Y chromosome and absent NLGN4Y expression.

Authors:  Judith L Ross; Luke Bloy; Timothy P L Roberts; Judith Miller; Chao Xing; Lawrence A Silverman; Andrew R Zinn
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2019-06-03       Impact factor: 3.568

3.  Auditory evoked response delays in children with 47,XYY syndrome.

Authors:  Luke Bloy; Matthew Ku; J Christopher Edgar; Judith S Miller; Lisa Blaskey; Judith Ross; Timothy P L Roberts
Journal:  Neuroreport       Date:  2019-05-01       Impact factor: 1.837

Review 4.  A review of neurocognitive functioning and risk for psychopathology in sex chromosome trisomy (47,XXY, 47,XXX, 47, XYY).

Authors:  Sophie van Rijn
Journal:  Curr Opin Psychiatry       Date:  2019-03       Impact factor: 4.741

5.  XYY syndrome: a 13-year-old boy with tall stature.

Authors:  Won Ha Jo; Mo Kyung Jung; Ki Eun Kim; Hyun Wook Chae; Duk Hee Kim; Ah Reum Kwon; Ho-Seong Kim
Journal:  Ann Pediatr Endocrinol Metab       Date:  2015-09-30

Review 6.  Genomics-based non-invasive prenatal testing for detection of fetal chromosomal aneuploidy in pregnant women.

Authors:  Mylène Badeau; Carmen Lindsay; Jonatan Blais; Leon Nshimyumukiza; Yemisi Takwoingi; Sylvie Langlois; France Légaré; Yves Giguère; Alexis F Turgeon; William Witteman; François Rousseau
Journal:  Cochrane Database Syst Rev       Date:  2017-11-10

7.  Tremor and Dystonia in Jacob's Syndrome (47,XYY).

Authors:  Jennie L Davis; Julie A Kurek; John C Morgan; Kapil D Sethi
Journal:  Mov Disord Clin Pract       Date:  2019-12-17

8.  Behavioral phenotypes in males with XYY and possible role of increased NLGN4Y expression in autism features.

Authors:  J L Ross; N Tartaglia; D E Merry; M Dalva; A R Zinn
Journal:  Genes Brain Behav       Date:  2015-02-01       Impact factor: 3.449

9.  Behavioral phenotype in a child with Prader-Willi syndrome and comorbid 47, XYY.

Authors:  Pooja Palkar; Anahid Kabasakalian; Bonnie Taylor; Ellen Doernberg; Casara Jean Ferretti; Genoveva Uzunova; Eric Hollander
Journal:  Intractable Rare Dis Res       Date:  2016-08

Review 10.  Genetics of Male Infertility.

Authors:  Filipe Tenorio Lira Neto; Phil Vu Bach; Bobby Baback Najari; Philip Shihua Li; Marc Goldstein
Journal:  Curr Urol Rep       Date:  2016-10       Impact factor: 3.092

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