Summary of studies investigating the prevalence of central adrenal insufficiency in patients with Prader–Willi syndrome
| Study . | N . | Median Age, Years (Range) . | GH Treatment (%) . | Testing Method . | Prevalence (%) . |
|---|---|---|---|---|---|
| Lind van Wijngaarden, et al (2008) (12) | 25 | 9.7 (3.7–18.6) | 100 | sMTP | 60 |
| Connell, et al (2010) (17) | 4 | 7.16 (0.43–16.27) | N/A | LDSST | 4 |
| 6 | HDSST | ||||
| 15 | ITT | ||||
| Nyunt, et al (2010) (16) | 41 | 7.68 (±5.23) a | 46 | LDSST | 0 |
| Farholt, et al (2011) (18) | 58 | 22 (0.42–48.0) | 62 | HDSST | 0 |
| 8 | ITT | 0 | |||
| Corrias, et al (2012) (10) | 84 | 7.7 (±5.0) a | 63 | LDSST | 14.2 |
| 9 b | HDSST | 4.8 | |||
| Grugni, et al (2013) (15) | 53 | 27.9 (18.0–45.2) | 30 | LDSST | 15 |
| 6 b | HDSST | 7.5 | |||
| Beauloye, et al (2015) (21) | 14 | 4.55 (0.8–14.7) | 25 | GT | 5 |
| 7 c | 5.6 (3.5–14.4) | ITT | |||
| Obrynba, et al (2018) (19) | 21 d | 13.9 (±10.9) a | 76 | LDSST | 29 |
| sMTP | 0 | ||||
| Oto, et al (2018) (20) | 36 | 2.0 (0.6–12.0) | 0 | ITT | 0 |
| This study (2019) | 46 | 25.3 (18.1–55.5) | 28 | MTP | 0 |
| 36 | 24.9 (18.0–55.3) | ITT | 2.8 |
| Study . | N . | Median Age, Years (Range) . | GH Treatment (%) . | Testing Method . | Prevalence (%) . |
|---|---|---|---|---|---|
| Lind van Wijngaarden, et al (2008) (12) | 25 | 9.7 (3.7–18.6) | 100 | sMTP | 60 |
| Connell, et al (2010) (17) | 4 | 7.16 (0.43–16.27) | N/A | LDSST | 4 |
| 6 | HDSST | ||||
| 15 | ITT | ||||
| Nyunt, et al (2010) (16) | 41 | 7.68 (±5.23) a | 46 | LDSST | 0 |
| Farholt, et al (2011) (18) | 58 | 22 (0.42–48.0) | 62 | HDSST | 0 |
| 8 | ITT | 0 | |||
| Corrias, et al (2012) (10) | 84 | 7.7 (±5.0) a | 63 | LDSST | 14.2 |
| 9 b | HDSST | 4.8 | |||
| Grugni, et al (2013) (15) | 53 | 27.9 (18.0–45.2) | 30 | LDSST | 15 |
| 6 b | HDSST | 7.5 | |||
| Beauloye, et al (2015) (21) | 14 | 4.55 (0.8–14.7) | 25 | GT | 5 |
| 7 c | 5.6 (3.5–14.4) | ITT | |||
| Obrynba, et al (2018) (19) | 21 d | 13.9 (±10.9) a | 76 | LDSST | 29 |
| sMTP | 0 | ||||
| Oto, et al (2018) (20) | 36 | 2.0 (0.6–12.0) | 0 | ITT | 0 |
| This study (2019) | 46 | 25.3 (18.1–55.5) | 28 | MTP | 0 |
| 36 | 24.9 (18.0–55.3) | ITT | 2.8 |
Abbreviations: GT, glucose tolerance test; HDSST, high-dose synacthen test; ITT, insulin tolerance test; LDSST, low-dose synacthen test; MTP, multiple-dose metyrapone test; N/A, not available; sMTP, single-dose metyrapone test.
a Age expressed as mean ± SD. b Number of subjects who failed the LDSST and underwent HDSST confirmation test. c 1 subject was tested by GT and ITT. d All subjects were tested by LDSST and sMTP.
Summary of studies investigating the prevalence of central adrenal insufficiency in patients with Prader–Willi syndrome
| Study . | N . | Median Age, Years (Range) . | GH Treatment (%) . | Testing Method . | Prevalence (%) . |
|---|---|---|---|---|---|
| Lind van Wijngaarden, et al (2008) (12) | 25 | 9.7 (3.7–18.6) | 100 | sMTP | 60 |
| Connell, et al (2010) (17) | 4 | 7.16 (0.43–16.27) | N/A | LDSST | 4 |
| 6 | HDSST | ||||
| 15 | ITT | ||||
| Nyunt, et al (2010) (16) | 41 | 7.68 (±5.23) a | 46 | LDSST | 0 |
| Farholt, et al (2011) (18) | 58 | 22 (0.42–48.0) | 62 | HDSST | 0 |
| 8 | ITT | 0 | |||
| Corrias, et al (2012) (10) | 84 | 7.7 (±5.0) a | 63 | LDSST | 14.2 |
| 9 b | HDSST | 4.8 | |||
| Grugni, et al (2013) (15) | 53 | 27.9 (18.0–45.2) | 30 | LDSST | 15 |
| 6 b | HDSST | 7.5 | |||
| Beauloye, et al (2015) (21) | 14 | 4.55 (0.8–14.7) | 25 | GT | 5 |
| 7 c | 5.6 (3.5–14.4) | ITT | |||
| Obrynba, et al (2018) (19) | 21 d | 13.9 (±10.9) a | 76 | LDSST | 29 |
| sMTP | 0 | ||||
| Oto, et al (2018) (20) | 36 | 2.0 (0.6–12.0) | 0 | ITT | 0 |
| This study (2019) | 46 | 25.3 (18.1–55.5) | 28 | MTP | 0 |
| 36 | 24.9 (18.0–55.3) | ITT | 2.8 |
| Study . | N . | Median Age, Years (Range) . | GH Treatment (%) . | Testing Method . | Prevalence (%) . |
|---|---|---|---|---|---|
| Lind van Wijngaarden, et al (2008) (12) | 25 | 9.7 (3.7–18.6) | 100 | sMTP | 60 |
| Connell, et al (2010) (17) | 4 | 7.16 (0.43–16.27) | N/A | LDSST | 4 |
| 6 | HDSST | ||||
| 15 | ITT | ||||
| Nyunt, et al (2010) (16) | 41 | 7.68 (±5.23) a | 46 | LDSST | 0 |
| Farholt, et al (2011) (18) | 58 | 22 (0.42–48.0) | 62 | HDSST | 0 |
| 8 | ITT | 0 | |||
| Corrias, et al (2012) (10) | 84 | 7.7 (±5.0) a | 63 | LDSST | 14.2 |
| 9 b | HDSST | 4.8 | |||
| Grugni, et al (2013) (15) | 53 | 27.9 (18.0–45.2) | 30 | LDSST | 15 |
| 6 b | HDSST | 7.5 | |||
| Beauloye, et al (2015) (21) | 14 | 4.55 (0.8–14.7) | 25 | GT | 5 |
| 7 c | 5.6 (3.5–14.4) | ITT | |||
| Obrynba, et al (2018) (19) | 21 d | 13.9 (±10.9) a | 76 | LDSST | 29 |
| sMTP | 0 | ||||
| Oto, et al (2018) (20) | 36 | 2.0 (0.6–12.0) | 0 | ITT | 0 |
| This study (2019) | 46 | 25.3 (18.1–55.5) | 28 | MTP | 0 |
| 36 | 24.9 (18.0–55.3) | ITT | 2.8 |
Abbreviations: GT, glucose tolerance test; HDSST, high-dose synacthen test; ITT, insulin tolerance test; LDSST, low-dose synacthen test; MTP, multiple-dose metyrapone test; N/A, not available; sMTP, single-dose metyrapone test.
a Age expressed as mean ± SD. b Number of subjects who failed the LDSST and underwent HDSST confirmation test. c 1 subject was tested by GT and ITT. d All subjects were tested by LDSST and sMTP.
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