Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage

Standard

Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage. / Beckhaus, Julia; Friedrich, Carsten; Boekhoff, Svenja; Calaminus, Gabriele; Bison, Brigitte; Eveslage, Maria; Timmermann, Beate; Flitsch, Jörg; Müller, Hermann L.

In: EUR J ENDOCRINOL, Vol. 188, No. 3, 02.03.2023, p. 300–309.

Research output: SCORING: Contribution to journalSCORING: Journal articleResearchpeer-review

Harvard

Beckhaus, J, Friedrich, C, Boekhoff, S, Calaminus, G, Bison, B, Eveslage, M, Timmermann, B, Flitsch, J & Müller, HL 2023, 'Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage', EUR J ENDOCRINOL, vol. 188, no. 3, pp. 300–309. https://doi.org/10.1093/ejendo/lvad027

APA

Beckhaus, J., Friedrich, C., Boekhoff, S., Calaminus, G., Bison, B., Eveslage, M., Timmermann, B., Flitsch, J., & Müller, H. L. (2023). Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage. EUR J ENDOCRINOL, 188(3), 300–309. https://doi.org/10.1093/ejendo/lvad027

Vancouver

Beckhaus J, Friedrich C, Boekhoff S, Calaminus G, Bison B, Eveslage M et al. Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage. EUR J ENDOCRINOL. 2023 Mar 2;188(3):300–309. https://doi.org/10.1093/ejendo/lvad027

Bibtex

@article{44d7ec5167274502842a0db74e5a834c,
title = "Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage",
abstract = "OBJECTIVE: Craniopharyngiomas (CP) are rare malformational tumors. Clinical presentation and outcome of pediatric patients with CP with specific regard to age at diagnosis is not clear. The aim of this cohort study was to determine clinical presentation and outcome in these patients diagnosed at different ages at diagnosis.DESIGN: Seven hundred and nine patients diagnosed with CP were recruited from 1999 to 2021 in HIT-Endo and KRANIOPHARYNGEOM 2000/2007/Registry 2019 and prospectively observed.METHODS: Age at diagnosis was categorized as infants and toddlers (<2 years), early childhood (2-6 years), middle childhood (6-12 years), and early adolescence (12-18 years). Overall and event-free survival (EFS), functional capacity (FMH), and quality of life (QoL) (PEDQOL) were assessed.RESULTS: Severe obesity (body mass index [BMI] >3 standard deviation score [SDS]) was prevalent in 45.4% at last visit. A lower EFS but better QoL was observed in children with age at diagnosis <6 years compared with ≥6 years. Reduced functional capacity percentiles were associated with increased BMI-SDS at last visit (rho = -0.125, 95% confidence interval [CI; -0.21; -0.04]) and age at diagnosis <2 years. Posterior hypothalamic involvement and hypothalamic lesion (HL) were independent risk factors for reduced EFS (hazard ratio = 1.59, 95% CI [1.12-2.26]) and obesity at last visit (odds ratio = 2.94, 95% CI [1.73-5.08]). Age at diagnosis did not contribute to severe obesity and reduced QoL.CONCLUSIONS: Diagnosis of CP at age <6 years may help patients to adapt early to disabilities but may lead to a higher probability of CP relapse. Not age at diagnosis but posterior HL may be the contributing factor to severe obesity and a reduced QoL.CLINICAL TRIAL REGISTRATION NUMBERS: NCT00258453; NCT01272622; NCT04158284.",
keywords = "Adolescent, Child, Child, Preschool, Humans, Infant, Cohort Studies, Craniopharyngioma/complications, Neoplasm Recurrence, Local, Obesity/complications, Obesity, Morbid, Pituitary Neoplasms/diagnosis, Quality of Life",
author = "Julia Beckhaus and Carsten Friedrich and Svenja Boekhoff and Gabriele Calaminus and Brigitte Bison and Maria Eveslage and Beate Timmermann and J{\"o}rg Flitsch and M{\"u}ller, {Hermann L}",
note = "{\textcopyright} The Author(s) 2023. Published by Oxford University Press on behalf of (ESE) European Society of Endocrinology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.",
year = "2023",
month = mar,
day = "2",
doi = "10.1093/ejendo/lvad027",
language = "English",
volume = "188",
pages = "300–309",
journal = "EUR J ENDOCRINOL",
issn = "0804-4643",
publisher = "BioScientifica Ltd.",
number = "3",

}

RIS

TY - JOUR

T1 - Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage

AU - Beckhaus, Julia

AU - Friedrich, Carsten

AU - Boekhoff, Svenja

AU - Calaminus, Gabriele

AU - Bison, Brigitte

AU - Eveslage, Maria

AU - Timmermann, Beate

AU - Flitsch, Jörg

AU - Müller, Hermann L

N1 - © The Author(s) 2023. Published by Oxford University Press on behalf of (ESE) European Society of Endocrinology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.

PY - 2023/3/2

Y1 - 2023/3/2

N2 - OBJECTIVE: Craniopharyngiomas (CP) are rare malformational tumors. Clinical presentation and outcome of pediatric patients with CP with specific regard to age at diagnosis is not clear. The aim of this cohort study was to determine clinical presentation and outcome in these patients diagnosed at different ages at diagnosis.DESIGN: Seven hundred and nine patients diagnosed with CP were recruited from 1999 to 2021 in HIT-Endo and KRANIOPHARYNGEOM 2000/2007/Registry 2019 and prospectively observed.METHODS: Age at diagnosis was categorized as infants and toddlers (<2 years), early childhood (2-6 years), middle childhood (6-12 years), and early adolescence (12-18 years). Overall and event-free survival (EFS), functional capacity (FMH), and quality of life (QoL) (PEDQOL) were assessed.RESULTS: Severe obesity (body mass index [BMI] >3 standard deviation score [SDS]) was prevalent in 45.4% at last visit. A lower EFS but better QoL was observed in children with age at diagnosis <6 years compared with ≥6 years. Reduced functional capacity percentiles were associated with increased BMI-SDS at last visit (rho = -0.125, 95% confidence interval [CI; -0.21; -0.04]) and age at diagnosis <2 years. Posterior hypothalamic involvement and hypothalamic lesion (HL) were independent risk factors for reduced EFS (hazard ratio = 1.59, 95% CI [1.12-2.26]) and obesity at last visit (odds ratio = 2.94, 95% CI [1.73-5.08]). Age at diagnosis did not contribute to severe obesity and reduced QoL.CONCLUSIONS: Diagnosis of CP at age <6 years may help patients to adapt early to disabilities but may lead to a higher probability of CP relapse. Not age at diagnosis but posterior HL may be the contributing factor to severe obesity and a reduced QoL.CLINICAL TRIAL REGISTRATION NUMBERS: NCT00258453; NCT01272622; NCT04158284.

AB - OBJECTIVE: Craniopharyngiomas (CP) are rare malformational tumors. Clinical presentation and outcome of pediatric patients with CP with specific regard to age at diagnosis is not clear. The aim of this cohort study was to determine clinical presentation and outcome in these patients diagnosed at different ages at diagnosis.DESIGN: Seven hundred and nine patients diagnosed with CP were recruited from 1999 to 2021 in HIT-Endo and KRANIOPHARYNGEOM 2000/2007/Registry 2019 and prospectively observed.METHODS: Age at diagnosis was categorized as infants and toddlers (<2 years), early childhood (2-6 years), middle childhood (6-12 years), and early adolescence (12-18 years). Overall and event-free survival (EFS), functional capacity (FMH), and quality of life (QoL) (PEDQOL) were assessed.RESULTS: Severe obesity (body mass index [BMI] >3 standard deviation score [SDS]) was prevalent in 45.4% at last visit. A lower EFS but better QoL was observed in children with age at diagnosis <6 years compared with ≥6 years. Reduced functional capacity percentiles were associated with increased BMI-SDS at last visit (rho = -0.125, 95% confidence interval [CI; -0.21; -0.04]) and age at diagnosis <2 years. Posterior hypothalamic involvement and hypothalamic lesion (HL) were independent risk factors for reduced EFS (hazard ratio = 1.59, 95% CI [1.12-2.26]) and obesity at last visit (odds ratio = 2.94, 95% CI [1.73-5.08]). Age at diagnosis did not contribute to severe obesity and reduced QoL.CONCLUSIONS: Diagnosis of CP at age <6 years may help patients to adapt early to disabilities but may lead to a higher probability of CP relapse. Not age at diagnosis but posterior HL may be the contributing factor to severe obesity and a reduced QoL.CLINICAL TRIAL REGISTRATION NUMBERS: NCT00258453; NCT01272622; NCT04158284.

KW - Adolescent

KW - Child

KW - Child, Preschool

KW - Humans

KW - Infant

KW - Cohort Studies

KW - Craniopharyngioma/complications

KW - Neoplasm Recurrence, Local

KW - Obesity/complications

KW - Obesity, Morbid

KW - Pituitary Neoplasms/diagnosis

KW - Quality of Life

U2 - 10.1093/ejendo/lvad027

DO - 10.1093/ejendo/lvad027

M3 - SCORING: Journal article

C2 - 36857103

VL - 188

SP - 300

EP - 309

JO - EUR J ENDOCRINOL

JF - EUR J ENDOCRINOL

SN - 0804-4643

IS - 3

ER -