TABLE 1.
Characteristics of enrolled participants1
Subject no. | BMI, kg/m2 | Classical/ variant2 | Mutation3 | Phe, μmol/L | Tyr, μmol/L |
1 | 21.7 | Variant4 | L348V; 165T | 201 | 25 |
2 | 20.3 | Variant | R68S; IVS12+1G>T | 224 | 36 |
3 | 39.7 | Variant | L48S; 195_K96delinsK | 297 | 53 |
4 | 25.1 | Classical | R408W; V338M | 301 | 26 |
5 | 27.3 | Classical | R408W; IVS12+1G>T | 367 | 36 |
6 | 21.1 | Variant4 | R241H; Q304Q | 380 | 29 |
7 | 24.6 | Variant | 165T; IVS10-11G>A | 461 | 34 |
8 | 24.6 | Classical | R158Q; R261Q | 481 | 30 |
9 | 25.3 | Classical | E280K; L348V | 520 | 56 |
10 | 34.1 | Classical | R408W; IVS7+3G>C | 535 | 42 |
11 | 29.2 | Variant | E280K; E390G | 541 | 20 |
12 | 22.3 | Classical | MIV; A246fsde1C | 606 | 36 |
13 | 26.8 | Variant4 | R158Q; R408W | 640 | 33 |
14 | 23.8 | Variant4 | R157N; L348V | 643 | 36 |
15 | 23.5 | Classical | R408W; IVS12 | 649 | 23 |
16 | 18.4 | Variant4 | P416Q; 664delGA | 704 | 26 |
17 | 23.4 | Classical | Y356X; R408W | 743 | 31 |
18 | 27.0 | Classical | R408W; R261Q | 754 | 47 |
19 | 31.7 | Classical | IVS1+5G>T; IVS12+1G>A | 800 | 38 |
20 | 25.3 | Variant | R408W; IVS12+1G>A | 821 | 37 |
21 | 26.9 | Classical | L242F; R408W | 829 | 29 |
22 | 28.1 | Classical | R408W; Y356X | 843 | 38 |
23 | 26.3 | Classical | E280K; IVS12+1G>T | 852 | 20 |
24 | 29.5 | Classical | Y277D; IVS10-11G>A | 893 | 17 |
25 | 32.0 | Classical | E280K; L348V | 937 | 40 |
26 | 23.9 | Classical | R261Q; IVS10-11G>A | 1059 | 30 |
27 | 23.8 | Classical | F55>Lfs; IVS5+1G>A | 1086 | 39 |
28 | 20.1 | Classical | F55>Lfs; R408W | 1160 | 30 |
29 | 26.9 | Classical | R408W; IVS12+1G>A | 1360 | 26 |
30 | 23.0 | Classical | IVS4+5G>T; IVS12+1G>A | 1418 | 37 |
Values were obtained at the first study visit when participants were consuming their usual low-Phe amino acid diet, and a fasting blood sample was obtained for baseline analysis of amino acids in plasma.
Subjects classified as having a variant form of phenylketonuria displayed a phenylalanine hydroxylase genotype and/or response to the drug sapropterin dihydrochloride, a synthetic form of the tetrahydrobiopterin cofactor for phenylalanine hydroxylase, that was consistent with a milder or variant form of phenylketonuria.
Mutation names are defined at http://www.pahdb.mcgill.ca and http://www.biopku.org.
Stable dose of sapropterin dihydrochloride (KUVAN, BioMarin Pharmaceutical Inc.) throughout the study.