Table 1.
Genotypes, biochemical and clinical phenotypes
Pat. No. | Mutation 1 | Mutation severity | Mutation 2 | Mutation severity | Genotype group | C8 NBS | C8 confirmation | HG at confirmation (0 = not detectable 1 = detectable) | Number of symptomatic episodes | If symptomatic episode: hypoglycaemia (1 = yes, 0 = no) | If symptomatic episode: impaired consciousness (1 = yes, 0 = no) | Clinical status (1 = abnormal, 0 = normal) | IQ | Age at evaluation (years) |
---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
1 | c.985A>G | S | c.985A>G | S | 1 | 1.73 | 1.91 | 0 | 0 | 0 | 128 | 14.4 | ||
2 | c.985A>G | S | c.985A>G | S | 1 | 7.14 | 1.3 | 1 | 1a | 1 | 1 | 0 | 109 | 13.7 |
3 | c.985A>G | S | c.985A>G | S | 1 | 1.05 | 5.33 | 0 | 0 | 0 | 102 | 13.7 | ||
4 | c.985A>G | S | c.985A>G | S | 1 | 7.58 | 10.1 | 1 | 1 | 0 | 1 | 0 | 79 | 12.3 |
5 | c.985A>G | S | c.985A>G | S | 1 | 6.49 | mv | mv | 1a | mv | 1 | 0 | 105 | 12.3 |
6 | c.985A>G | S | c.985A>G | S | 1 | 22.46 | 2.31 | mv | 0 | nd | nd | 11.3 | ||
7 | c.985A>G | S | c.985A>G | S | 1 | 23.19 | 5.5 | mv | 0 | 0 | 106 | 11.2 | ||
8 | c.985A>G | S | c.985A>G | S | 1 | 3.05 | 4.03 | mv | 0 | 0 | 104 | 9.9 | ||
9 | c.985A>G | S | c.985A>G | S | 1 | 4.69 | 1.73 | 1 | 0 | 0 | 104 | 6.4 | ||
10 | c.985A>G | S | c.985A>G | S | 1 | 1.87 | 3.1 | 1 | 0 | nd | 76 | 6.2 | ||
11 | c.985A>G | S | c.985A>G | S | 1 | 4.9 | 3.39 | 1 | 2 | mv | 1 | 0 | 119 | 6.8 |
12 | c.985A>G | S | c.985A>G | S | 1 | 6.43 | 3.96 | 1 | 0 | 0 | 118 | 5.9 | ||
13 | c.985A>G | S | c.985A>G | S | 1 | 3.92 | 3.06 | 1 | 0 | 0 | 120 | 5.3 | ||
14 | c.985A>G | S | c.985A>G | S | 1 | 6.54 | 1.59 | 1 | 0 | 0 | AA | 2.4 | ||
15 | c.985A>G | S | c.985A>G | S | 1 | 40.22 | 0.97 | 1 | 1a | 1 | 0 | 0 | AA | 1.8 |
16 | c.985A>G | S | c.985A>G | S | 1 | 7.89 | 1.46 | 1 | 0 | 0 | nd | 1.2 | ||
17 | c.799G>A | Sb | c.199T>C | M | 2 | 2.15 | 6.51c | 1 | 1a | 1 | 1 | 0 | 93 | 4.3 |
18 | c.403_405delATT | Sd | c.199T>C | M | 2 | 2.51 | 0.39 | 1 | 0 | 0 | 114 | 9.9 | ||
19 | c.985A>G | S | c.199T>C | M | 2 | 1.7 | 1.34 | 0 | 0 | 0 | 108 | 14.1 | ||
20 | c.985A>G | S | c.199T>C | M | 2 | 2.2 | 0.5 | 1 | 0 | 0 | 127 | 8.4 | ||
21 | c.985A>G | S | c.199T>C | M | 2 | 2.21 | 1.13 | 1 | 1a | 1 | 0 | 0 | 100 | 8.4 |
22 | c.985A>G | S | c.199T>C | M | 2 | 3.1 | 0.53 | 0 | 0 | 0 | 92 | 7.8 | ||
23 | c.985A>G | S | c.199T>C | M | 2 | 1.36 | 0.35 | 1 | 0 | 0 | 122 | 7.7 | ||
24 | c.985A>G | S | c.199T>C | M | 2 | 0.97 | 1.94 | mv | 0 | 0 | 122 | 7.8 | ||
25 | c.985A>G | S | c.199T>C | M | 2 | 1.17 | 0.37 | 1 | 0 | 0 | AA | 1.9 | ||
26 | c.985A>G | S | c.199T>C | M | 2 | 1.94 | 0.28 | 1 | 0 | 0 | AA | 1.5 | ||
27 | c.1140_1141insG | Ue/S | c.199T>C | M | 2 | 0.78 | 0.48 | mv | 0 | 0 | AA | 7.8 | ||
28 | c.985A>G | S | c.424_426delAAG | Sf | 3 | 6.17 | 4.6 | mv | 0 | 0 | nd | 4.3 | ||
29 | c.985A>G | S | c.721G>A | Ug | 3 | 14.38 | 5.72 | 1 | 0 | 0 | AA | 3.2 | ||
30 | c.985A>G | S | c.799G>A | Sb | 3 | 6.47 | 1.52 | 1 | 0 | 0 | nd | 1.6 | ||
31 | c.985A>G | S | c.387+1G>A | Sh | 3 | 4.97 | 1.06 | 1 | 0 | 1 | NAA | 3.9 | ||
32 | c.985A>G | S | c.362C>T | Si | 3 | 2.15 | 3.86 | 1 | 0 | nd | 107 | 7.1 | ||
33 | c.985A>G | S | c.244_245insT | Si | 3 | 10.82 | 3.88 | 1 | 0 | 0 | nd | 1.7 | ||
34 | c.985A>G | S | c.244_245insT | Si | 3 | 0.57 | 1.67 | 1 | 0 | 0 | nd | 0.6 | ||
35 | c.583G>A | Sj | c.583G>A | Sj | 4 | 9.27 | 3.82 | 1 | 0 | 0 | AA | 2.7 | ||
36 | c.799G>A | Sb | c.799G>A | Sb | 4 | 4.17 | 2.67 | 1 | 0 | 0 | 107 | 11.0 | ||
37 | c.799G>A | Sb | c.799G>A | Sb | 4 | 6.01 | 1.24 | mv | 0 | 0 | 90 | 7.7 |
aDecompensation in neonatal period
bSmith et al. (2010)
cSample was taken in period of decompensation
dSturm et al. (2012) (Nomenclature c.397_399delATT, enzyme activity 3%)
eSequence variant of unknown clinical significance; theoretically causing frameshift and premature stop codon, therefore presumably severe mutation; enzyme activity in this patient 13.9% according to ter Veld et al. (2009)
fEnzyme activity in lymphocytes 0% (performed at the University Children’s Hospital, Duesseldorf)
gMutation not previously described
hYusupov et al. (2010)
iAndresen et al. (2001)
jWaddell et al. (2006)
AA result of Denver test appropriate for age, C8 octanoylcarnitine, HG hexanoylglycine in urine, mv missing value, NAA result of Denver test not appropriate for age, NBS newborn screening, nd not done, S severe (assumed mutation severity), M mild (assumed mutation severity), U mutation severity unknown