Abstract
Purpose
All treatments for childhood craniopharyngioma are associated with complications that potentially affect quality of life. This study was designed to investigate the impact of gross total resection on long-term quality of life and sexual functioning in adulthood.
Methods
Adults treated with primary gross total resection for childhood craniopharyngioma and ≥ 10 years of follow-up were included in this retrospective cohort study. The Short Form 36 Health Survey Questionnaire Version 2 (SF-36v2), Medical Outcomes Study (MOS) sexual functioning survey, and a sociodemographic/health questionnaire were administered.
Results
Twenty-two subjects with a median length of follow-up of 19 years (range 12–30) completed the questionnaires. Fifty-five percent reported excellent or very good general health. There was no significant difference of the mean SF-36v2 score between the patient cohort and the normal population. Twenty-two percent of females and 54% of males reported at least “a little of a problem” in one or more areas of sexual functioning, similar to the normal population. The proportion of sexually active individuals was decreased in this cohort. The median BMI of the participants was 29.5 (range 22.1–50.0 kg/m2). Preoperative hypothalamic involvement correlated with a significantly higher BMI, although the proportion of participants with class 3 obesity (BMI ≥ 40) did not differ significantly from that of the general population (9% and 7%, respectively).
Conclusions
Young adults with gross total resection of childhood craniopharyngioma report similar quality of life and sexual functioning compared to the general population, but appear to be less sexually active. Hypothalamic involvement on preoperative imaging was associated with a higher BMI in long-term follow-up.
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References
Centers for Disease Control and Prevention (2016). Health, United States, 2015: With Special Feature on Racial and Ethnic Health Disparities. National Center for Health Statistics (US). Report number: 2016-1232
Bober SL, Zhou ES, Chen B, Manley PE, Kenney LB, Recklitis CJ (2013) Sexual function in childhood cancer survivors: a report from project REACH. J Sex Med 10:2084–2093. https://doi.org/10.1111/jsm.12193
Doll HA, Petersen SE, Stewart-Brown SL (2000) Obesity and physical and emotional well-being: associations between body mass index, chronic illness, and the physical and mental components of the SF-36 questionnaire. Obes Res 8:160–170. https://doi.org/10.1038/oby.2000.17
Elliott RE, Hsieh K, Hochm T, Belitskaya-Levy I, Wisoff J, Wisoff JH (2010) Efficacy and safety of radical resection of primary and recurrent craniopharyngiomas in 86 children. J Neurosurg Pediatr 5:30–48. https://doi.org/10.3171/2009.7.PEDS09215
Elowe-Gruau E, Beltrand J, Brauner R, Pinto G, Samara-Boustani D, Thalassinos C, Busiah K, Laborde K, Boddaert N, Zerah M, Alapetite C, Grill J, Touraine P, Sainte-Rose C, Polak M, Puget S (2013) Childhood craniopharyngioma: hypothalamus-sparing surgery decreases the risk of obesity. J Clin Endocrinol Metab 98:2376–2382. https://doi.org/10.1210/jc.2012-3928
Fjalldal S, Holmer H, Rylander L, Elfving M, Ekman B, Osterberg K, Erfurth EM (2013) Hypothalamic involvement predicts cognitive performance and psychosocial health in long-term survivors of childhood craniopharyngioma. J Clin Endocrinol Metab 98:3253–3262. https://doi.org/10.1210/jc.2013-2000
Hoffmann A, Warmth-Metz M, Gebhardt U, Pietsch T, Pohl F, Kortmann RD, Calaminus G, Muller HL (2014) Childhood craniopharyngioma - changes of treatment strategies in the trials KRANIOPHARYNGEOM 2000/2007. Klin Padiatr 226:161–168. https://doi.org/10.1055/s-0034-1368785
Langeveld NE, Ubbink MC, Last BF, Grootenhuis MA, Voute PA, De Haan RJ (2003) Educational achievement, employment and living situation in long-term young adult survivors of childhood cancer in the Netherlands. Psycho-oncology 12:213–225. https://doi.org/10.1002/pon.628
Laumann EO, Paik A, Rosen RC (1999) Sexual dysfunction in the United States: prevalence and predictors. JAMA 281:537–544
Lustig RH (2011) Hypothalamic obesity after craniopharyngioma: mechanisms, diagnosis, and treatment. Front Endocrinol (Lausanne) 2:60. https://doi.org/10.3389/fendo.2011.00060
Macartney G, Harrison MB, VanDenKerkhof E, Stacey D, McCarthy P (2014) Quality of life and symptoms in pediatric brain tumor survivors: a systematic review. J Pediatr Oncol Nurs 31:65–77. https://doi.org/10.1177/1043454213520191
Meadows AT, Gordon J, Massari DJ, Littman P, Fergusson J, Moss K (1981) Declines in IQ scores and cognitive dysfunctions in children with acute lymphocytic leukaemia treated with cranial irradiation. Lancet 2:1015–1018
Memmesheimer RM, Lange K, Dolle M, Heger S, Mueller I (2017) Psychological well-being and independent living of young adults with childhood-onset craniopharyngioma. Dev Med Child Neurol 59:829–836. https://doi.org/10.1111/dmcn.13444
Moons P, Van Deyk K, De Bleser L, Marquet K, Raes E, De Geest S, Budts W (2006) Quality of life and health status in adults with congenital heart disease: a direct comparison with healthy counterparts. Eur J Cardiovasc Prev Rehabil 13:407–413
Muller HL (2016) Craniopharyngioma and hypothalamic injury: latest insights into consequent eating disorders and obesity. Curr Opin Endocrinol Diabetes Obes 23:81–89. https://doi.org/10.1097/MED.0000000000000214
Muller HL, Bruhnken G, Emser A, Faldum A, Etavard-Gorris N, Gebhardt U, Kolb R, Sorensen N (2005) Longitudinal study on quality of life in 102 survivors of childhood craniopharyngioma. Childs Nerv Syst 21:975–980. https://doi.org/10.1007/s00381-004-1124-y
Packer RJ, Meadows AT, Rorke LB, Goldwein JL, D'Angio G (1987) Long-term sequelae of cancer treatment on the central nervous system in childhood. Med Pediatr Oncol 15:241–253
Pierre-Kahn A, Recassens C, Pinto G, Thalassinos C, Chokron S, Soubervielle JC, Brauner R, Zerah M, Sainte Rose C (2005) Social and psycho-intellectual outcome following radical removal of craniopharyngiomas in childhood. A prospective series. Childs Nerv Syst 21:817–824. https://doi.org/10.1007/s00381-005-1205-6
Puget S (2012) Treatment strategies in childhood craniopharyngioma. Front Endocrinol (Lausanne) 3:64. https://doi.org/10.3389/fendo.2012.00064
Puget S, Garnett M, Wray A, Grill J, Habrand JL, Bodaert N, Zerah M, Bezerra M, Renier D, Pierre-Kahn A, Sainte-Rose C (2007) Pediatric craniopharyngiomas: classification and treatment according to the degree of hypothalamic involvement. J Neurosurg 106:3–12. https://doi.org/10.3171/ped.2007.106.1.3
Sanford RA (1994) Craniopharyngioma: results of survey of the American society of pediatric neurosurgery. Pediatr Neurosurg 21(Suppl 1):39–43
Speechley KN, Barrera M, Shaw AK, Morrison HI, Maunsell E (2006) Health-related quality of life among child and adolescent survivors of childhood cancer. J Clin Oncol 24:2536–2543. https://doi.org/10.1200/JCO.2005.03.9628
Sterkenburg AS, Hoffmann A, Gebhardt U, Warmuth-Metz M, Daubenbuchel AM, Muller HL (2015) Survival, hypothalamic obesity, and neuropsychological/psychosocial status after childhood-onset craniopharyngioma: newly reported long-term outcomes. Neuro-oncology 17:1029–1038. https://doi.org/10.1093/neuonc/nov044
Stewart AL, Ware JE (1992) Measuring functioning and well-being: the medical outcomes study approach. Duke University Press, Durham
Stripp DC, Maity A, Janss AJ, Belasco JB, Tochner ZA, Goldwein JW, Moshang T, Rorke LB, Phillips PC, Sutton LN, Shu HK (2004) Surgery with or without radiation therapy in the management of craniopharyngiomas in children and young adults. Int J Radiat Oncol Biol Phys 58:714–720. https://doi.org/10.1016/S0360-3016(03)01570-0
van Dijk EM, van Dulmen-den Broeder E, Kaspers GJ, van Dam EW, Braam KI, Huisman J (2008) Psychosexual functioning of childhood cancer survivors. Psycho-oncology 17:506–511. https://doi.org/10.1002/pon.1274
Vespa J (2017) The changing economics and demographics of young adulthood: 1975–2016. United States Census Bureau. Report number: P20–579
Vinchon M, Dhellemmes P (2008) Craniopharyngiomas in children: recurrence, reoperation and outcome. Childs Nerv Syst 24:211–217. https://doi.org/10.1007/s00381-007-0456-9
Vinchon M, Weill J, Delestret I, Dhellemmes P (2009) Craniopharyngioma and hypothalamic obesity in children. Childs Nerv Syst 25:347–352. https://doi.org/10.1007/s00381-008-0754-x
Wang KW, Fleming A, Johnston DL, Zelcer SM, Rassekh SR, Ladhani S, Socha A, Shinuda J, Jaber S, Burrow S, Singh SK, Banfield L, de Souza RJ, Thabane L, Samaan MC (2018) Overweight, obesity and adiposity in survivors of childhood brain tumours: a systematic review and meta-analysis. Clin Obes 8:55–67. https://doi.org/10.1111/cob.12224
Ware JE Jr, Sherbourne CD (1992) The MOS 36-item short-form health survey (SF-36). I. Conceptual framework and item selection. Med Care 30:473–483
Wijnen M, van den Heuvel-Eibrink MM, Janssen J, Catsman-Berrevoets CE, Michiels EMC, van Veelen-Vincent MC, AHG D, van den Berge JH, van Rij CM, van der Lely AJ, Neggers S (2017) Very long-term sequelae of craniopharyngioma. Eur J Endocrinol 176:755–767. https://doi.org/10.1530/EJE-17-0044
Yano S, Kudo M, Hide T, Shinojima N, Makino K, Nakamura H, Kuratsu J (2016) Quality of life and clinical features of long-term survivors surgically treated for pediatric Craniopharyngioma. World Neurosurg 85:153–162. https://doi.org/10.1016/j.wneu.2015.08.059
Zebrack BJ, Foley S, Wittmann D, Leonard M (2010) Sexual functioning in young adult survivors of childhood cancer. Psycho-oncology 19:814–822. https://doi.org/10.1002/pon.1641
Funding
This publication is supported in part by the NYU CTSA grant UL1TR001445 from the National Center for Advancing Translational Sciences, National Institutes of Health and the Making Headway Foundation to Eveline Teresa Hidalgo.
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Supplemental Figure 1
Bar graph showing the distribution of weight classification among participants in the years 2006 and 2015. (PNG 1143 kb)
Supplemental Figure 2
Pie charts describing the responses to individual questions of sexual functioning for the participants of the study (PNG 948 kb)
Supplemental Figure 3
The Physical Component Score (PCS) and Mental Component Score (MCS) by Puget classification (PNG 76 kb)
Supplemental Figure 4
The body mass index of participants by location of their tumor in relation to the optic chiasm. (PNG 98 kb)
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Hidalgo, E.T., Orillac, C., Kvint, S. et al. Quality of life, hypothalamic obesity, and sexual function in adulthood two decades after primary gross-total resection for childhood craniopharyngioma. Childs Nerv Syst 36, 281–289 (2020). https://doi.org/10.1007/s00381-019-04161-9
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DOI: https://doi.org/10.1007/s00381-019-04161-9