Leukocyte Telomere Length in Children With Congenital Adrenal Hyperplasia
© The Author(s) 2022. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissionsoup.com..
CONTEXT: Exposure to chronic stress and hypercortisolism is associated with decreased leukocyte telomere length (LTL), a marker for biological aging and cardiovascular disease. Children with congenital adrenal hyperplasia (CAH) are treated with glucocorticoids.
OBJECTIVE: To investigate LTL in children with CAH.
METHODS: In this prospective observational cohort study, conducted at 4 academic pediatric endocrinology outpatient clinics, children with genetically confirmed CAH were assessed at 2 follow-up visits (mean 4.1 ± 0.7 months apart). At each visit, LTL was determined by quantitative real-time PCR. All subjects underwent detailed clinical and endocrinologic evaluation and were classified as undertreated, optimally treated, or overtreated, accordingly. The influence of clinical factors on LTL was investigated using linear mixed models adjusted for age, sex, and BMI-z.
RESULTS: We studied 76 patients, of whom 31 (41%) were girls, 63 (83%) had classic CAH, 67 (88%) received hydrocortisone, and 8 (11%) prednisolone. Median age at first visit was 12.0 years (IQR, 6.3-15.1), and median BMI-z was 0.51 (IQR, -0.12 to 1.43). LTL was shorter in patients with classic vs nonclassic CAH (-0.29, P = 0.012), in overtreated than in optimally treated patients (-0.07, P = 0.002), and patients receiving prednisolone compared with hydrocortisone (-0.34, P < 0.001). LTL was not associated with undertreatment or daily hydrocortisone-equivalent dose (P > 0.05).
CONCLUSION: LTL is shorter in patients with classic than nonclassic CAH, and in those who are overtreated with hydrocortisone or treated with long-acting glucocorticoids. These findings may be attributed to chronic exposure to supraphysiologic glucocorticoid concentrations and indicate that LTL may be used as a biomarker for monitoring glucocorticoid treatment.
| Medienart: |
E-Artikel |
|---|
| Erscheinungsjahr: |
2023 |
|---|---|
| Erschienen: |
2023 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:108 |
|---|---|
| Enthalten in: |
The Journal of clinical endocrinology and metabolism - 108(2023), 2 vom: 17. Jan., Seite 443-452 |
| Sprache: |
Englisch |
|---|
| Beteiligte Personen: |
Raftopoulou, Christina [VerfasserIn] |
|---|
| Links: |
|---|
| Anmerkungen: |
Date Completed 19.01.2023 Date Revised 16.02.2023 published: Print Citation Status MEDLINE |
|---|
| doi: |
10.1210/clinem/dgac560 |
|---|
| funding: |
|
|---|---|
| Förderinstitution / Projekttitel: |
|
| PPN (Katalog-ID): |
NLM346969751 |
|---|
| LEADER | 01000naa a22002652 4500 | ||
|---|---|---|---|
| 001 | NLM346969751 | ||
| 003 | DE-627 | ||
| 005 | 20231226032721.0 | ||
| 007 | cr uuu---uuuuu | ||
| 008 | 231226s2023 xx |||||o 00| ||eng c | ||
| 024 | 7 | |a 10.1210/clinem/dgac560 |2 doi | |
| 028 | 5 | 2 | |a pubmed24n1156.xml |
| 035 | |a (DE-627)NLM346969751 | ||
| 035 | |a (NLM)36181470 | ||
| 040 | |a DE-627 |b ger |c DE-627 |e rakwb | ||
| 041 | |a eng | ||
| 100 | 1 | |a Raftopoulou, Christina |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Leukocyte Telomere Length in Children With Congenital Adrenal Hyperplasia |
| 264 | 1 | |c 2023 | |
| 336 | |a Text |b txt |2 rdacontent | ||
| 337 | |a ƒaComputermedien |b c |2 rdamedia | ||
| 338 | |a ƒa Online-Ressource |b cr |2 rdacarrier | ||
| 500 | |a Date Completed 19.01.2023 | ||
| 500 | |a Date Revised 16.02.2023 | ||
| 500 | |a published: Print | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © The Author(s) 2022. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissionsoup.com. | ||
| 520 | |a CONTEXT: Exposure to chronic stress and hypercortisolism is associated with decreased leukocyte telomere length (LTL), a marker for biological aging and cardiovascular disease. Children with congenital adrenal hyperplasia (CAH) are treated with glucocorticoids | ||
| 520 | |a OBJECTIVE: To investigate LTL in children with CAH | ||
| 520 | |a METHODS: In this prospective observational cohort study, conducted at 4 academic pediatric endocrinology outpatient clinics, children with genetically confirmed CAH were assessed at 2 follow-up visits (mean 4.1 ± 0.7 months apart). At each visit, LTL was determined by quantitative real-time PCR. All subjects underwent detailed clinical and endocrinologic evaluation and were classified as undertreated, optimally treated, or overtreated, accordingly. The influence of clinical factors on LTL was investigated using linear mixed models adjusted for age, sex, and BMI-z | ||
| 520 | |a RESULTS: We studied 76 patients, of whom 31 (41%) were girls, 63 (83%) had classic CAH, 67 (88%) received hydrocortisone, and 8 (11%) prednisolone. Median age at first visit was 12.0 years (IQR, 6.3-15.1), and median BMI-z was 0.51 (IQR, -0.12 to 1.43). LTL was shorter in patients with classic vs nonclassic CAH (-0.29, P = 0.012), in overtreated than in optimally treated patients (-0.07, P = 0.002), and patients receiving prednisolone compared with hydrocortisone (-0.34, P < 0.001). LTL was not associated with undertreatment or daily hydrocortisone-equivalent dose (P > 0.05) | ||
| 520 | |a CONCLUSION: LTL is shorter in patients with classic than nonclassic CAH, and in those who are overtreated with hydrocortisone or treated with long-acting glucocorticoids. These findings may be attributed to chronic exposure to supraphysiologic glucocorticoid concentrations and indicate that LTL may be used as a biomarker for monitoring glucocorticoid treatment | ||
| 650 | 4 | |a Observational Study | |
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Research Support, Non-U.S. Gov't | |
| 650 | 4 | |a 21-hydroxylase | |
| 650 | 4 | |a LTL | |
| 650 | 4 | |a adolescents | |
| 650 | 4 | |a congenital adrenal hyperplasia | |
| 650 | 4 | |a glucocorticoid | |
| 650 | 4 | |a telomeres | |
| 650 | 7 | |a Hydrocortisone |2 NLM | |
| 650 | 7 | |a WI4X0X7BPJ |2 NLM | |
| 650 | 7 | |a Glucocorticoids |2 NLM | |
| 650 | 7 | |a Prednisolone |2 NLM | |
| 650 | 7 | |a 9PHQ9Y1OLM |2 NLM | |
| 700 | 1 | |a Abawi, Ozair |e verfasserin |4 aut | |
| 700 | 1 | |a Sommer, Grit |e verfasserin |4 aut | |
| 700 | 1 | |a Binou, Maria |e verfasserin |4 aut | |
| 700 | 1 | |a Paltoglou, George |e verfasserin |4 aut | |
| 700 | 1 | |a Flück, Christa E |e verfasserin |4 aut | |
| 700 | 1 | |a van den Akker, Erica L T |e verfasserin |4 aut | |
| 700 | 1 | |a Charmandari, E |e verfasserin |4 aut | |
| 773 | 0 | 8 | |i Enthalten in |t The Journal of clinical endocrinology and metabolism |d 1945 |g 108(2023), 2 vom: 17. Jan., Seite 443-452 |w (DE-627)NLM00001821X |x 1945-7197 |7 nnns |
| 773 | 1 | 8 | |g volume:108 |g year:2023 |g number:2 |g day:17 |g month:01 |g pages:443-452 |
| 856 | 4 | 0 | |u http://dx.doi.org/10.1210/clinem/dgac560 |3 Volltext |
| 912 | |a GBV_USEFLAG_A | ||
| 912 | |a GBV_NLM | ||
| 951 | |a AR | ||
| 952 | |d 108 |j 2023 |e 2 |b 17 |c 01 |h 443-452 | ||