There is no specific treatment for Noonan syndrome. Treatment focuses on the individual symptom, and may include surgery for undescended testes in boys, optimisation of cardiac function, and growth hormone (GH) treatment for short stature.[60]Romano AA, Allanson JE, Dahlgren J, et al. Noonan syndrome: clinical features, diagnosis, and management guidelines. Pediatrics. 2010 Oct;126(4):746-59.
http://www.ncbi.nlm.nih.gov/pubmed/20876176?tool=bestpractice.com
It is important to note specific medical conditions that are associated with NS. Some patients may not have the constellation of facial characteristics, but may present with associated medical conditions. These include: skeletal abnormalities, including chest deformity; variable degrees of developmental delay; ophthalmic and hearing deficits; coagulation defects; autoimmune disease; and lymphatic dysplasia. Appropriate investigations and specialist consults should be obtained.
Cryptorchidism
Undescended testes are presenting features in most boys (60% to 69%) and may be associated with delayed puberty.[1]Jorge AA, Malaquias AC, Arnhold IJ, et al. Noonan syndrome and related disorders: a review of clinical features and mutations in genes of the RAS/MAPK pathway. Horm Res. 2009;71(4):185-93.
https://www.karger.com/Article/FullText/201106
http://www.ncbi.nlm.nih.gov/pubmed/19258709?tool=bestpractice.com
The surgical approach is determined by surgeon preference, and is generally through an inguinal or scrotal incision. The optimal timing for surgical therapy is debated, but recent data suggest that there may be better preservation of spermatogenesis and hormonal production, with decreased risk of testicular cancer, when surgery is performed early, ideally prior to 2 years of age, with some suggesting before 1 year of age.[65]Ritzén EM, Bergh A, Bjerknes R, et al. Nordic consensus on treatment of undescended testes. Acta Paediatr. 2007 May;96(5):638-43.
http://onlinelibrary.wiley.com/doi/10.1111/j.1651-2227.2006.00159.x/full
http://www.ncbi.nlm.nih.gov/pubmed/17326760?tool=bestpractice.com
Congenital heart anomalies
These defects may occur in up to 75% of cases and are the most common cause of death for people with the syndrome.[1]Jorge AA, Malaquias AC, Arnhold IJ, et al. Noonan syndrome and related disorders: a review of clinical features and mutations in genes of the RAS/MAPK pathway. Horm Res. 2009;71(4):185-93.
https://www.karger.com/Article/FullText/201106
http://www.ncbi.nlm.nih.gov/pubmed/19258709?tool=bestpractice.com
They include pulmonary valve stenosis and/or dysplasia, hypertrophic cardiomyopathy, septal defects, and tetralogy of Fallot.[30]Shaw AC, Kalidas K, Crosby AH, et al. The natural history of Noonan syndrome: a long-term follow-up study. Arch Dis Child. 2007 Feb;92(2):128-32.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2083343
http://www.ncbi.nlm.nih.gov/pubmed/16990350?tool=bestpractice.com
[55]Burch M, Sharland M, Shinebourne E, et al. Cardiologic abnormalities in Noonan syndrome: phenotypic diagnosis and echocardiographic assessment of 118 patients. J Am Coll Cardiol. 1993 Oct;22(4):1189-92.
http://www.sciencedirect.com/science/article/pii/0735109793904365?via%3Dihub
http://www.ncbi.nlm.nih.gov/pubmed/8409059?tool=bestpractice.com
[56]Ishizawa A, Oho S, Dodo H, et al. Cardiovascular abnormalities in Noonan syndrome: the clinical findings and treatments. Acta Paediatr Jpn. 1996 Feb;38(1):84-90.
http://www.ncbi.nlm.nih.gov/pubmed/8992869?tool=bestpractice.com
[57]Noonan JA, O'Connor W. Noonan syndrome: a clinical description emphasizing the cardiac findings. Acta Paediatr Jpn. 1996 Feb;38(1):76-83.
http://www.ncbi.nlm.nih.gov/pubmed/8992867?tool=bestpractice.com
Comprehensive cardiovascular evaluation at the time of diagnosis is imperative. Subsequent treatment will be guided by the specific anomaly identified. Pulmonary valve dysplasia may respond to balloon valvuloplasty, but may require valve replacement.[56]Ishizawa A, Oho S, Dodo H, et al. Cardiovascular abnormalities in Noonan syndrome: the clinical findings and treatments. Acta Paediatr Jpn. 1996 Feb;38(1):84-90.
http://www.ncbi.nlm.nih.gov/pubmed/8992869?tool=bestpractice.com
Hypertrophic cardiomyopathy may be treated by beta-blockade or calcium-channel blockade, but occasionally requires surgical myomectomy.[56]Ishizawa A, Oho S, Dodo H, et al. Cardiovascular abnormalities in Noonan syndrome: the clinical findings and treatments. Acta Paediatr Jpn. 1996 Feb;38(1):84-90.
http://www.ncbi.nlm.nih.gov/pubmed/8992869?tool=bestpractice.com
[57]Noonan JA, O'Connor W. Noonan syndrome: a clinical description emphasizing the cardiac findings. Acta Paediatr Jpn. 1996 Feb;38(1):76-83.
http://www.ncbi.nlm.nih.gov/pubmed/8992867?tool=bestpractice.com
Cardiac care should continue into adulthood, as there can be ongoing morbidity and mortality associated with the defects.[30]Shaw AC, Kalidas K, Crosby AH, et al. The natural history of Noonan syndrome: a long-term follow-up study. Arch Dis Child. 2007 Feb;92(2):128-32.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2083343
http://www.ncbi.nlm.nih.gov/pubmed/16990350?tool=bestpractice.com
[56]Ishizawa A, Oho S, Dodo H, et al. Cardiovascular abnormalities in Noonan syndrome: the clinical findings and treatments. Acta Paediatr Jpn. 1996 Feb;38(1):84-90.
http://www.ncbi.nlm.nih.gov/pubmed/8992869?tool=bestpractice.com
In some centres, children with cardiac anomalies should be transferred to an Adult Congenital Heart Disease clinic at age 18 years, while in other centres, patients will continue to see paediatric cardiologists.
Poor growth
Poor growth, short stature, and an attenuated pubertal growth spurt are common features and may be the presenting symptoms of NS. Children with documented poor growth should be referred to an endocrinologist. Early initiation of GH therapy may facilitate growth optimization, but risks and benefits of comorbidities of NS such as hypertrophic cardiomyopathy and scoliosis must be considered.
Studies of growth hormone (GH) treatment in Noonan syndrome report height gains of 13 cm (5 inches) for males.[66]Romano AA. Growth and Growth Hormone Treatment in Noonan Syndrome. Pediatr Endocrinol Rev. 2019 May;16(suppl 2):459-464.
https://www.doi.org/10.17458/per.vol16.2019.r.growthhormonenoonan
http://www.ncbi.nlm.nih.gov/pubmed/31115197?tool=bestpractice.com
[67]Raaijmakers R, Noordam C, Karagiannis G, et al. Response to growth hormone treatment and final height in Noonan syndrome in a large cohort of patients in the KIGS database. J Pediatr Endocrinol Metab. 2008 Mar;21(3):267-73.
http://www.ncbi.nlm.nih.gov/pubmed/18540254?tool=bestpractice.com
[68]Osio D, Dahlgren J, Wikland KA, et al. Improved final height with long-term growth hormone treatment in Noonan syndrome. Acta Paediatr. 2005 Sep;94(9):1232-7.
http://www.ncbi.nlm.nih.gov/pubmed/16203673?tool=bestpractice.com
The US National Cooperative Growth study demonstrated near-adult height gains of up to 15 cm (6 inches) in patients (mean starting age of 11.6 years) treated for an average of 5 years.[69]Romano AA, Dana K, Bakker B, et al. Growth response, near-adult height, and patterns of growth and puberty in patients with Noonan syndrome treated with growth hormone. J Clin Endocrinol Metab. 2009 Jul;94(7):2338-44.
http://www.ncbi.nlm.nih.gov/pubmed/19401366?tool=bestpractice.com
The duration of prepubertal treatment was an important contributor to change in height, suggesting greater growth potential with earlier treatment initiation.[69]Romano AA, Dana K, Bakker B, et al. Growth response, near-adult height, and patterns of growth and puberty in patients with Noonan syndrome treated with growth hormone. J Clin Endocrinol Metab. 2009 Jul;94(7):2338-44.
http://www.ncbi.nlm.nih.gov/pubmed/19401366?tool=bestpractice.com
[66]Romano AA. Growth and Growth Hormone Treatment in Noonan Syndrome. Pediatr Endocrinol Rev. 2019 May;16(suppl 2):459-464.
https://www.doi.org/10.17458/per.vol16.2019.r.growthhormonenoonan
http://www.ncbi.nlm.nih.gov/pubmed/31115197?tool=bestpractice.com
Treatment effect is monitored by the growth response and insulin-like growth factor 1 (IGF-1) levels.
Pathogenic variants in the PTPN11 gene have been associated with reduced growth response to GH in NS.[25]Binder G, Neuer K, Ranke MB, et al. PTPN11 mutations are associated with mild growth hormone resistance in individuals with Noonan syndrome. J Clin Endocrinol Metab. 2005 Sep;90(9):5377-81.
https://academic.oup.com/jcem/article-lookup/doi/10.1210/jc.2005-0995
http://www.ncbi.nlm.nih.gov/pubmed/15985475?tool=bestpractice.com
[15]Limal JM, Parfait B, Cabrol S, et al. Noonan syndrome: relationships between genotype, growth, and growth factors. J Clin Endocrinol Metab. 2006 Jan;91(1):300-6.
https://academic.oup.com/jcem/article-lookup/doi/10.1210/jc.2005-0983
http://www.ncbi.nlm.nih.gov/pubmed/16263833?tool=bestpractice.com
[70]Ferreira LV, Souza SA, Arnhold IJ, et al. PTPN11 (protein tyrosine phosphatase, nonreceptor type 11) mutations and response to growth hormone therapy in children with Noonan syndrome. J Clin Endocrinol Metab. 2005 Sep;90(9):5156-60.
https://academic.oup.com/jcem/article/90/9/5156/2838677
http://www.ncbi.nlm.nih.gov/pubmed/15956085?tool=bestpractice.com
[71]Jeong I, Kang E, Cho JH, et al. Long-term efficacy of recombinant human growth hormone therapy in short-statured patients with Noonan syndrome. Ann Pediatr Endocrinol Metab. 2016 Mar;21(1):26-30.
https://www.doi.org/10.6065/apem.2016.21.1.26
http://www.ncbi.nlm.nih.gov/pubmed/27104176?tool=bestpractice.com
However, other studies report that response to GH therapy does not differ significantly according to genotype.[72]Choi JH, Lee BH, Jung CW, et al. Response to growth hormone therapy in children with Noonan syndrome: correlation with or without PTPN11 gene mutation. Horm Res Paediatr. 2012;77(6):388-93.
https://www.doi.org/10.1159/000339677
http://www.ncbi.nlm.nih.gov/pubmed/22777296?tool=bestpractice.com
[73]Malaquias AC, Noronha RM, Souza TTO, et al. Impact of Growth Hormone Therapy on Adult Height in Patients with PTPN11 Mutations Related to Noonan Syndrome. Horm Res Paediatr. 2019;91(4):252-261.
https://www.doi.org/10.1159/000500264
http://www.ncbi.nlm.nih.gov/pubmed/31132774?tool=bestpractice.com
[74]Noordam C, Peer PG, Francois I, et al. Long-term GH treatment improves adult height in children with Noonan syndrome with and without mutations in protein tyrosine phosphatase, non-receptor-type 11. Eur J Endocrinol. 2008 Sep;159(3):203-8.
https://www.doi.org/10.1530/EJE-08-0413
http://www.ncbi.nlm.nih.gov/pubmed/18562489?tool=bestpractice.com
[75]Seok EM, Park HK, Rho JG, et al. Effectiveness of growth hormone therapy in children with Noonan syndrome. Ann Pediatr Endocrinol Metab. 2020 Sep;25(3):182-186.
https://www.doi.org/10.6065/apem.1938154.077
http://www.ncbi.nlm.nih.gov/pubmed/32871657?tool=bestpractice.com
Large prospective studies are required to determine whether genotype influences response to GH therapy.