Literature DB >> 26023614

Absence of Nails, Deaf-mutism, Seizures, and Intellectual Disability: A Case Report.

Lal Devayanivasudevan Nair1, Benjamin Sagayaraj2, Radha Kumar3.   

Abstract

Seizures coexists in children with intellectual disability and are often attributed to neural dysfunction associated with it. Often a careful clinical examination will unravel many diagnostic pointers as in this 8-year-old child with global development delay, deaf-mutism and moderate intellectual disability (mental retardation) who presented with seizures in the emergency department. General examination revealed dysmorphic features like anonychia, low set ears, long philtrum, large lower lips and abnormal dermatoglyphics with features of osteodystrophy on radiology. She was diagnosed as a case of DOORS syndrome, an extremely rare genetic condition affecting the TCA cycle, with just over 40 cases reported, worldwide till date, since its first description in 1961. Her genetic analysis did not reveal the common TBC1D24 mutation in 16p13.3 resulting often from substitutions affecting the arginine at position 242, in spite of all classical clinical features associated with it, suggesting genetic heterogeneity in DOORS syndrome. Though four year follow-up revealed changes in seizure pattern, there was no optic atrophy, change in IQ or peripheral nerve problem. This probably suggests that children with typical clinical features and TBC1D24 mutations may have more progressive deterioration than those without it and newer molecular techniques may identify unexplained phenotypic expressions.

Entities:  

Keywords:  Absent nails; Developmental delay; Door syndrome; Dysmorphia; Onychodystrophy

Year:  2015        PMID: 26023614      PMCID: PMC4437130          DOI: 10.7860/JCDR/2015/12086.5763

Source DB:  PubMed          Journal:  J Clin Diagn Res        ISSN: 0973-709X


  8 in total

1.  Congenital deafness associated with onychodystrophy.

Authors:  M FEINMESSER; S ZELIG
Journal:  Arch Otolaryngol       Date:  1961-11

2.  Congenital sensori-neural deafness associated with onycho-osteo dystrophy and mental retardation (D.O.O.R. syndrome).

Authors:  R J Cantwell
Journal:  Humangenetik       Date:  1975

3.  DOOR syndrome.

Authors:  Meenakshi Girish; Nilofer Mujawar; Atul Salodkar
Journal:  Indian Pediatr       Date:  2011-06       Impact factor: 1.411

4.  2-ketoglutarate dehydrogenase deficiency with intermittent 2-ketoglutaric aciduria.

Authors:  R J Dunckelmann; F Ebinger; A Schulze; R J Wanders; D Rating; E Mayatepek
Journal:  Neuropediatrics       Date:  2000-02       Impact factor: 1.947

5.  Further delineation of the DOOR syndrome.

Authors:  A Rajab; A Riaz; G Paul; S Al-Khusaibi; R Chalmers; M A Patton
Journal:  Clin Dysmorphol       Date:  2000-10       Impact factor: 0.816

6.  DOOR syndrome: clinical report, literature review and discussion of natural history.

Authors:  Aaron W James; Suzette G Miranda; Kathy Culver; Bryan D Hall; Mahin Golabi
Journal:  Am J Med Genet A       Date:  2007-12-01       Impact factor: 2.802

7.  [DOOR (deafness, onychodystrophy, osteodystrophy, mental retardation) syndrome].

Authors:  László Szabó; Katalin Kocsis; Katalin Hollódy; Márta Soroncz; Wolfgang Erwa; Miklós Andits
Journal:  Orv Hetil       Date:  2004-05-30       Impact factor: 0.540

8.  The genetic basis of DOORS syndrome: an exome-sequencing study.

Authors:  Philippe M Campeau; Dalia Kasperaviciute; James T Lu; Lindsay C Burrage; Choel Kim; Mutsuki Hori; Berkley R Powell; Fiona Stewart; Têmis Maria Félix; Jenneke van den Ende; Marzena Wisniewska; Hülya Kayserili; Patrick Rump; Sheela Nampoothiri; Salim Aftimos; Antje Mey; Lal D V Nair; Michael L Begleiter; Isabelle De Bie; Girish Meenakshi; Mitzi L Murray; Gabriela M Repetto; Mahin Golabi; Edward Blair; Alison Male; Fabienne Giuliano; Ariana Kariminejad; William G Newman; Sanjeev S Bhaskar; Jonathan E Dickerson; Bronwyn Kerr; Siddharth Banka; Jacques C Giltay; Dagmar Wieczorek; Anna Tostevin; Joanna Wiszniewska; Sau Wai Cheung; Raoul C Hennekam; Richard A Gibbs; Brendan H Lee; Sanjay M Sisodiya
Journal:  Lancet Neurol       Date:  2013-11-29       Impact factor: 44.182

  8 in total
  1 in total

1.  Doors Syndrome: Case Report.

Authors:  Dua Cebeci; Didem Rıfkı
Journal:  Indian J Dermatol       Date:  2022 Mar-Apr       Impact factor: 1.757

  1 in total

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