Table 2 Incorporation of [2‐14C]acetate into cellular sterols and gas chromatography–mass spectrometry sterol analysis of lymphoblastoid cell lines from a patient with known Smith–Lemli–Opitz syndrome and from 22 patients with holoprosencephaly showing an abnormal sterol pattern in the [2‐14C]acetate loading test.
| Cholesterol | Desmosterol | Lathosterol | 7‐DHC | 8‐DHC | Lanosterol | DHCR7 mutation | |||||||
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
| 14C acetate loading | GC‐MS | 14C acetate loading | GC‐MS | 14C acetate loading | GC‐MS | 14C acetate loading | GC‐MS | 14C acetate loading | GC‐MS | 14C acetate loading | GC‐MS | ||
| Controls | 72–91 | 91–98 | 11.7 | 1.7 | 11.7 | 2.4 | 3.5 | 0.64 | 11.7 | 0.4 | 8.5 | 1.3 | |
| Patient | |||||||||||||
| SLOS | 4.9 | 60 | 25 | 0.1 | 25 | 4.7 | 61 | 32 | 25 | 3.7 | 8.0 | 0.1 | R352W/IVS8‐1G>C |
| 144 | 63 | 91 | 15 | 2.3 | 15 | 2.1 | 4.8 | 0.6 | 15 | 0.3 | 8.9 | 1.9 | ND |
| 329 | 60 | 95 | 16 | 0.9 | 16 | 1.9 | 9.6 | 0.4 | 16 | 0.2 | 7.4 | 0.3 | −/− |
| 331 | 36 | 94 | 37 | 0.7 | 37 | 3.6 | 13 | 0.9 | 37 | 0.4 | 8.4 | 0.1 | −/− |
| 383 | 68 | 93 | 13 | 0.4 | 13 | 5.1 | 6.9 | 0.8 | 13 | 0.4 | 3.3 | < dl | −/− |
| 613 | 19 | 72 | 23 | 0.1 | 23 | 3 | 47 | 22 | 23 | 1.8 | 6.3 | 0.2 | T154R/G410S |
| 646 | 34 | 97 | 38 | < dl | 38 | 1.6 | 11 | 0.8 | 38 | 0.2 | 8.6 | < dl | ND |
| 719 | 62 | 95 | 17 | 0.3 | 17 | 2.0 | 5.4 | 0.5 | 17 | 0.3 | 8.0 | < dl | −/− |
| 811 | 76 | 94 | 11 | 0.4 | 11 | 1.8 | 5.8 | 1.4 | 11 | 0.2 | 3 | < dl | −/− |
| 859 | 37 | 92 | 21 | 5.4 | 21 | 1 | 11 | 0.7 | 21 | 0.7 | 22 | < dl | ND |
| 887 | 56 | 94 | 19 | 0.8 | 19 | 2.1 | 6.2 | 0.4 | 19 | 0.2 | 7 | 0.4 | −/− |
| 893 | 64 | 91 | 18 | 0.3 | 18 | 5.8 | 2.8 | 0.9 | 18 | 0.5 | 9.2 | 0.1 | ND |
| 1265 | 63 | 93 | 14 | 0.2 | 14 | 0.9 | 14 | 0.9 | 14 | 1.5 | 6.9 | < dl | −/− |
| 6006 | 48 | 94 | 18 | 0.4 | 18 | 1.3 | 19 | 1.6 | 18 | 0.4 | 3.9 | < dl | −/− |
| 6833 | 75 | 95 | 11 | 0.4 | 11 | 2.2 | 7.9 | 0.4 | 11 | 0.2 | 3.2 | 0.4 | −/− |
| 7304 | 58 | 94 | 21 | 0.9 | 21 | 1.9 | 11 | 0.5 | 21 | 0.2 | 5.4 | 0.5 | −/− |
| 7334 | 77 | 94 | 7.6 | 0.4 | 7.6 | 2.1 | 5.6 | 0.5 | 7.6 | 0.4 | 5.9 | 0.1 | R242H/− |
| 7835 | 66 | 96 | 14 | 0.6 | 14 | 1.6 | 12 | 0.9 | 14 | 0.3 | 4.9 | 0.4 | −/− |
| 7858 | 74 | 93 | 10 | 0.4 | 10 | 0.8 | 11 | 0.3 | 10 | 0.2 | 2.6 | 0.2 | −/− |
| 7880 | 51 | 91 | 18 | 0.5 | 18 | 1.7 | 12 | 0.3 | 18 | 0.2 | 5.1 | 0.3 | −/− |
| 8064 | 61 | 94 | 18 | 0.9 | 18 | 1.5 | 5.9 | 0.7 | 18 | 0.4 | 8.6 | 0.5 | ND |
| 8103 | 42 | 97 | 22 | 1.1 | 22 | 0.7 | 14 | 0.3 | 22 | 0.2 | 8.6 | 0.3 | ND |
| 8121 | 66 | 93 | 14 | 0.3 | 14 | 1.3 | 13 | 0.3 | 14 | 0.1 | 2.7 | 0.3 | −/− |
DHC, dehydrocholesterol; DHCR, 7‐dehydrosterol reductase; GC‐MS, gas chromatography‐mass spectrometry; ND, not detected; SLOS, Smith–Lemli–Opitz syndrome.
Indicated is the percentage of total sterols, mean of three independent experiments ([2‐14C]acetate‐loading) or two independent experiments (GC‐MS). The control values represent mean (2 SD) of nine controls (cholesterol) or mean (2 SD) (cholesterol precursors). Data are printed in bold if they fell below this range (cholesterol) or exceeded this range (cholesterol precursors).