Blood phenylalanine (phe) levels should be obtained at periodic intervals to monitor the efficacy of therapy in patients with Phenylketonuria (PKU). The recommended minimum interval is:[18]van Wegberg AMJ, MacDonald A, Ahring K, et al. The complete European guidelines on phenylketonuria: diagnosis and treatment. Orphanet J Rare Dis. 2017 Oct 12;12(1):162.
https://ojrd.biomedcentral.com/articles/10.1186/s13023-017-0685-2
http://www.ncbi.nlm.nih.gov/pubmed/29025426?tool=bestpractice.com
Weekly for infants <1 year of age, whose nutritional requirements change frequently as a result of rapid growth
Every 2 weeks from 1 to 12 years of age
Monthly after 12 years of age
Weekly during preconception
Twice weekly during pregnancy
The target therapeutic range for blood phe levels varies across guidelines. In Europe, the European Society of Phenylketonuria and Allied Disorders stratifies recommended targets by age: 2-6 mg/dL for individuals under 12 years and 2-10 mg/dL for those ages 12 and older.[18]van Wegberg AMJ, MacDonald A, Ahring K, et al. The complete European guidelines on phenylketonuria: diagnosis and treatment. Orphanet J Rare Dis. 2017 Oct 12;12(1):162.
https://ojrd.biomedcentral.com/articles/10.1186/s13023-017-0685-2
http://www.ncbi.nlm.nih.gov/pubmed/29025426?tool=bestpractice.com
In the US, the American College of Medical Genetics and Genomics recommends that all individuals with PKU maintain a mean lifetime phe level of ≤6 mg/dL.[9]Smith WE, Berry SA, Bloom K, et al. Phenylalanine hydroxylase deficiency diagnosis and management: a 2023 evidence-based clinical guideline of the American College of Medical Genetics and Genomics (ACMG). Genet Med. 2025 Jan;27(1):101289.
https://www.gimjournal.org/article/S1098-3600(24)00223-5/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/39630157?tool=bestpractice.com
Increasing evidence supports stricter recommendations by the US guidelines, as studies have reported that adolescents and young adults with blood phe levels above 4-6 mg/dL tend to experience poorer social-cognition and executive functioning, and more severe mental health challenges.[10]Burton BK, Hermida Á, Bélanger-Quintana A, et al. Management of early treated adolescents and young adults with phenylketonuria: development of international consensus recommendations using a modified Delphi approach. Mol Genet Metab. 2022 Sep-Oct;137(1-2):114-26.
https://www.sciencedirect.com/science/article/pii/S1096719222003729
http://www.ncbi.nlm.nih.gov/pubmed/36027720?tool=bestpractice.com
Levels above or below the therapeutic range should prompt an adjustment in therapy.
A diet diary should be submitted with each blood sample so that the adequacy of the diet can be assessed and adjustments can be made when blood phe levels are not within range. Screening for physical health comorbidities should begin early and continue throughout life, as these comorbidities may occur regardless of age and are influenced by blood phe control.[10]Burton BK, Hermida Á, Bélanger-Quintana A, et al. Management of early treated adolescents and young adults with phenylketonuria: development of international consensus recommendations using a modified Delphi approach. Mol Genet Metab. 2022 Sep-Oct;137(1-2):114-26.
https://www.sciencedirect.com/science/article/pii/S1096719222003729
http://www.ncbi.nlm.nih.gov/pubmed/36027720?tool=bestpractice.com
In addition to physical exams, assessments should include anthropometric measurements, nutritional intake reviews, lipid panels, allergy history (e.g., eczema and rhinitis), and micronutrient levels, such as vitamins B12, D, folate, and zinc.[10]Burton BK, Hermida Á, Bélanger-Quintana A, et al. Management of early treated adolescents and young adults with phenylketonuria: development of international consensus recommendations using a modified Delphi approach. Mol Genet Metab. 2022 Sep-Oct;137(1-2):114-26.
https://www.sciencedirect.com/science/article/pii/S1096719222003729
http://www.ncbi.nlm.nih.gov/pubmed/36027720?tool=bestpractice.com
A DEXA scan for bone density should be performed initially by age 12, with follow-up scans as dictated by findings.[10]Burton BK, Hermida Á, Bélanger-Quintana A, et al. Management of early treated adolescents and young adults with phenylketonuria: development of international consensus recommendations using a modified Delphi approach. Mol Genet Metab. 2022 Sep-Oct;137(1-2):114-26.
https://www.sciencedirect.com/science/article/pii/S1096719222003729
http://www.ncbi.nlm.nih.gov/pubmed/36027720?tool=bestpractice.com
Vitamin D levels should be monitored regularly, and supplementation should be provided for any deficiencies. If osteopenia is identified, serum calcium, phosphorus, and parathyroid hormone levels should also be obtained. Neuropsychologic assessment is recommended if cognitive or behavioral issues are suspected.[18]van Wegberg AMJ, MacDonald A, Ahring K, et al. The complete European guidelines on phenylketonuria: diagnosis and treatment. Orphanet J Rare Dis. 2017 Oct 12;12(1):162.
https://ojrd.biomedcentral.com/articles/10.1186/s13023-017-0685-2
http://www.ncbi.nlm.nih.gov/pubmed/29025426?tool=bestpractice.com
The recommended minimum frequency of outpatient visits for patients with good clinical and metabolic control is:[18]van Wegberg AMJ, MacDonald A, Ahring K, et al. The complete European guidelines on phenylketonuria: diagnosis and treatment. Orphanet J Rare Dis. 2017 Oct 12;12(1):162.
https://ojrd.biomedcentral.com/articles/10.1186/s13023-017-0685-2
http://www.ncbi.nlm.nih.gov/pubmed/29025426?tool=bestpractice.com
Every 2 months for infants ages <1 year
Every 6 months from ages 1-18 years
Yearly after ages 12 years
Once per trimester during pregnancy
Pregnant women with PKU should be monitored closely by physicians familiar with the condition, with ongoing follow-up from a metabolic geneticist and high-risk pregnancy specialists. Additionally, fetal growth assessments and a detailed anatomic survey are recommended to detect potential anomalies or growth restrictions, and fetal echocardiography should be performed to screen for congenital heart defects.[19]American College of Obstetricians and Gynocologists. Practice bulletin no. 802: management of women with phenylalanine hydroxylase deficiency (phenylketonuria). Apr 2020 [internet publication].