Literature DB >> 7294061

A comprehensive scoring system for evaluating Noonan syndrome.

W J Duncan, R S Fowler, L G Farkas, R B Ross, A W Wright, K R Bloom, D J Huot, H M Sondheimer, R D Rowe.   

Abstract

A multidisciplinary team assessed 23 patients with various manifestations of the Noonan syndrome, including pulmonary valve stenosis (with leaflet dysplasia), "typical" facial appearance (including hypertelorism, epicanthic folds, flat nasal bridge, and apparently low-set ears), short stature, and mental retardation. Seven patients had a family history of the syndrome. A comprehensive scoring system was devised on the basis of frequency and severity of manifestations and results of invasive and noninvasive tests in these patients and those reported in the literature. The scoring system was condensed into a score card for clinical use and validated by "blind" application to patients with isolated pulmonary valve stenosis or suspected Noonan syndrome. Use of a scoring system to diagnose a syndrome for which there is no specific diagnostic test facilitates accuracy and decreases observer bias. In the case of unusual congenital disorders it is particularly valuable for a pediatrician in general practice.

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Year:  1981        PMID: 7294061     DOI: 10.1002/ajmg.1320100106

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  12 in total

1.  Absence of linkage of Noonan syndrome to the neurofibromatosis type 1 locus.

Authors:  M Sharland; R Taylor; M A Patton; S Jeffery
Journal:  J Med Genet       Date:  1992-03       Impact factor: 6.318

2.  A clinical study of Noonan syndrome.

Authors:  M Sharland; M Burch; W M McKenna; M A Paton
Journal:  Arch Dis Child       Date:  1992-02       Impact factor: 3.791

3.  Noonan syndrome: clinical aspects and molecular pathogenesis.

Authors:  M Tartaglia; G Zampino; B D Gelb
Journal:  Mol Syndromol       Date:  2010-01-15

4.  Phenotypic spectrum of 80 Greek patients referred as Noonan syndrome and PTPN11 mutation analysis: the value of initial clinical assessment.

Authors:  Anna Papadopoulou; Michalis Issakidis; Evangelia Gole; Konstantina Kosma; Helen Fryssira; Andreas Fretzayas; Polyxeni Nicolaidou; Sophia Kitsiou-Tzeli
Journal:  Eur J Pediatr       Date:  2011-05-18       Impact factor: 3.183

5.  Short stature in Noonan syndrome: response to growth hormone therapy.

Authors:  J M Kirk; P R Betts; G E Butler; M D Donaldson; D B Dunger; D I Johnston; C J Kelnar; D A Price; P Wilton
Journal:  Arch Dis Child       Date:  2001-05       Impact factor: 3.791

Review 6.  Stunted growth with more or less normal appearance.

Authors:  J R Bierich; H Enders; U Heinrich; R Huenges; M B Ranke; D Schoenberg
Journal:  Eur J Pediatr       Date:  1982-12       Impact factor: 3.183

7.  Are ECG abnormalities in Noonan syndrome characteristic for the syndrome?

Authors:  R Raaijmakers; C Noordam; J A Noonan; E A Croonen; C J A M van der Burgt; J M T Draaisma
Journal:  Eur J Pediatr       Date:  2008-02-13       Impact factor: 3.183

8.  The dysplastic pulmonary valve: echocardiographic features and results of balloon dilatation.

Authors:  N N Musewe; M A Robertson; L N Benson; J F Smallhorn; P E Burrows; R M Freedom; C A Moes; R D Rowe
Journal:  Br Heart J       Date:  1987-04

9.  Noonan syndrome: growth and clinical manifestations in 144 cases.

Authors:  M B Ranke; P Heidemann; C Knupfer; H Enders; A A Schmaltz; J R Bierich
Journal:  Eur J Pediatr       Date:  1988-12       Impact factor: 3.183

10.  Mutation Spectrum and Phenotypic Features in Noonan Syndrome with PTPN11 Mutations: Definition of Two Novel Mutations.

Authors:  Tahir Atik; Ayca Aykut; Filiz Hazan; Huseyin Onay; Damla Goksen; Sukran Darcan; Ajlan Tukun; Ferda Ozkinay
Journal:  Indian J Pediatr       Date:  2016-01-28       Impact factor: 1.967

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