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A clinical study of Noonan syndrome.
Open accessAbstract
Clinical details are presented on 151 individuals with Noonan syndrome (83 males and 68 females, mean age 12.6 years). Polyhydramnios complicated 33% of affected pregnancies. The commonest cardiac lesions were pulmonary stenosis (62%), and hypertrophic cardiomyopathy (20%), with a normal echocardiogram present in only 12.5% of all cases. Significant feeding difficulties during infancy were present in 76% of the group. Although the children were short (50% with a height less than 3rd centile), and underweight (43% with a weight less than 3rd centile), the mean head circumference of the group was on the 50th centile. Motor milestone delay was usual, the cohort having a mean age of sitting unsupported of 10 months and walking of 21 months. Abnormal vision (94%) and hearing (40%) were frequent findings, but 89% of the group were attending normal primary or secondary schools. Other associations included undescended testicles (77%), hepatosplenomegaly (50%), and evidence of abnormal bleeding (56%). The mean age at diagnosis of Noonan syndrome in this group was 9.0 years. Earlier diagnosis of this common condition would aid both clinical management and genetic counselling.
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Citations
Cited by
Gain-of-function SOS1 mutations cause a distinctive form of Noonan syndrome
Tartaglia, M., Pennacchio, L. A., Zhao, C., Yadav, K. K., Fodale, V., Sarkozy, A., Pandit, B., Oishi, K., Martinelli, S., Schackwitz, W., et al. (2007) · Nature Genetics · doi:10.1038/ng1939 · Cited by 586
Goriely, A., & Wilkie, A. O. M. (2012) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2011.12.017 · Cited by 365
van der Burgt, I. (2007) · Orphanet Journal of Rare Diseases · doi:10.1186/1750-1172-2-4 · Cited by 365
NOONAN SYNDROME AND RELATED DISORDERS: Genetics and Pathogenesis
Tartaglia, M., & Gelb, B. D. (2005) · Annual Review of Genomics and Human Genetics · doi:10.1146/annurev.genom.6.080604.162305 · Cited by 355
Meulepas, J. M., Ronckers, C. M., Merks, J., Weijerman, M. E., Lubin, J. H., & Hauptmann, M. (2016) · Journal of Radiological Protection · doi:10.1088/0952-4746/36/4/953 · Cited by 29
Metadata
- Authors
- Sharland, M., Burch, M., McKenna, W. M., & Paton, M. A.
- Venue
- Archives of Disease in Childhood
- Publisher
- BMJ
- Published
- 1992-02-01
- Volume
- 67
- Issue
- 2
- Pages
- 178-183
- ISSN
- 0003-9888 (ISSN-L)
- 1468-2044
- OpenAlex
- W2033157019
- Cited by
- 475
- References
- 0
- FWCI
- 3.05
- Type
- journal article
- Language
- en
- Open access
- bronze
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- Sharland, M., Burch, M., McKenna, W. M., & Paton, M. A. (1992). A clinical study of Noonan syndrome. Archives of Disease in Childhood. https://doi.org/10.1136/adc.67.2.178
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