Literature DB >> 9350820

Macrocephaly, epilepsy, autism, dysmorphic features, and mental retardation in two sisters: a new autosomal recessive syndrome?

K H Orstavik1, P Strømme, J Ek, A Torvik, O H Skjeldal.   

Abstract

We report two sisters with macrocephaly, epilepsy, and severe mental retardation. The first child was a 14 year old girl born at term after a normal pregnancy, with birth weight 3600 g and occipitofrontal circumference (OFC) 36 cm (75th centile). Her head size increased markedly during the first six months of life, and was later stable at 2-3 cm above the 97.5th centile. Her development was characterised by psychomotor delay, epilepsy, and autistic features. Her face appeared mildly dysmorphic with a large forehead, short philtrum, and bushy eyebrows. Her younger sister was also born at term with birth weight 2600 g and OFC 34 cm (25th centile). She also developed postnatal macrocephaly with OFC 2 cm above the 97.5th centile and the same mild dysmorphic facial features as her sister. Her development was also characterised by psychomotor delay, autistic features, and epilepsy. In addition, she suffered from coeliac disease. She died unexpectedly at the age of 5 years, probably from an epileptic attack. Necropsy confirmed megalencephaly but no other pathological changes were found. The clinical features in these two sisters do not fit with any known syndrome and may represent a previously unrecognised autosomal recessive disorder.

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Year:  1997        PMID: 9350820      PMCID: PMC1051094          DOI: 10.1136/jmg.34.10.849

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  9 in total

1.  Centiles for adult head circumference.

Authors:  K M Bushby; T Cole; J N Matthews; J A Goodship
Journal:  Arch Dis Child       Date:  1992-10       Impact factor: 3.791

2.  Sotos syndrome: a study of the diagnostic criteria and natural history.

Authors:  T R Cole; H E Hughes
Journal:  J Med Genet       Date:  1994-01       Impact factor: 6.318

3.  An apparently new autosomal recessive syndrome with facial dysmorphism, macrocephaly, myopia and Dandy-Walker malformation.

Authors:  M Buttiens; J P Fryns; H van den Berghe
Journal:  Clin Genet       Date:  1989-12       Impact factor: 4.438

4.  Mental retardation, macrocephaly, short stature and craniofacial dysmorphism in three sisters. A new entity among the mental retardation-macrocephaly syndromes?

Authors:  J P Fryns; A M Dereymaeker; J Haegeman; H van den Berghe
Journal:  Clin Genet       Date:  1988-04       Impact factor: 4.438

5.  A prospective study on sudden unexpected death in epilepsy.

Authors:  J E Leestma; T Walczak; J R Hughes; M B Kalelkar; S S Teas
Journal:  Ann Neurol       Date:  1989-08       Impact factor: 10.422

6.  Macrocephaly, distinct craniofacial appearance and spastic paraplegia: an autosomal recessive subtype of complicated spastic paraplegia.

Authors:  J P Fryns; M Hellemans; H Van den Berghe
Journal:  Clin Genet       Date:  1994-05       Impact factor: 4.438

7.  Epilepsy, cerebral calcifications, and coeliac disease.

Authors:  W Dickey
Journal:  Lancet       Date:  1994-12-10       Impact factor: 79.321

8.  Body Length and Organ Weights of Infants and Children: A Study of the Body Length and Normal Weights of the More Important Vital Organs of the Body between Birth and Twelve Years of Age.

Authors:  J M Coppoletta; S B Wolbach
Journal:  Am J Pathol       Date:  1933-01       Impact factor: 4.307

9.  Apnoea and bradycardia during epileptic seizures: relation to sudden death in epilepsy.

Authors:  L Nashef; F Walker; P Allen; J W Sander; S D Shorvon; D R Fish
Journal:  J Neurol Neurosurg Psychiatry       Date:  1996-03       Impact factor: 10.154

  9 in total
  4 in total

1.  Mutation screening of the PTEN gene in patients with autism spectrum disorders and macrocephaly.

Authors:  Joseph D Buxbaum; Guiqing Cai; Pauline Chaste; Gudrun Nygren; Juliet Goldsmith; Jennifer Reichert; Henrik Anckarsäter; Maria Rastam; Christopher J Smith; Jeremy M Silverman; Eric Hollander; Marion Leboyer; Christopher Gillberg; Alain Verloes; Catalina Betancur
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2007-06-05       Impact factor: 3.568

2.  Temporal lobe, autism, and macrocephaly.

Authors:  Erin D Bigler; David F Tate; E Shannon Neeley; Lara J Wolfson; Michael J Miller; Sara A Rice; Howard Cleavinger; Carol Anderson; Hilary Coon; Sally Ozonoff; Michael Johnson; Elena Dinh; Jeff Lu; William Mc Mahon; Janet E Lainhart
Journal:  AJNR Am J Neuroradiol       Date:  2003 Nov-Dec       Impact factor: 3.825

3.  Mutation analysis of the NSD1 gene in patients with autism spectrum disorders and macrocephaly.

Authors:  Joseph D Buxbaum; Guiqing Cai; Gudrun Nygren; Pauline Chaste; Richard Delorme; Juliet Goldsmith; Maria Råstam; Jeremy M Silverman; Eric Hollander; Christopher Gillberg; Marion Leboyer; Catalina Betancur
Journal:  BMC Med Genet       Date:  2007-11-14       Impact factor: 2.103

4.  The Neuropathology of Gluten-Related Neurological Disorders: A Systematic Review.

Authors:  Maxine D Rouvroye; Panagiotis Zis; Anne-Marie Van Dam; Annemieke J M Rozemuller; Gerd Bouma; Marios Hadjivassiliou
Journal:  Nutrients       Date:  2020-03-20       Impact factor: 5.717

  4 in total

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