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Predictors of narcolepsy and hypersomnia due to medical disorder in pediatric craniopharyngioma

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Several studies describe sleep–wake disturbances in pediatric craniopharyngioma, but none have determined the prevalence or associated predictors of excessive sleepiness in this group after diagnosis and prior to post-operative observation or adjuvant radiotherapy. In this study, we report sleep–wake disturbances in children and adolescents with craniopharyngioma and associated clinical and treatment variables.


After surgery and prior to radiotherapy or observation, pediatric patients (n = 110) with craniopharyngioma ≥ 3 years old completed a baseline sleep clinic evaluation by a pediatric sleep specialist, polysomnography (PSG) and next-day multiple sleep latency test (MSLT). MSLT was limited to those ≥ 6 years old. Logistic regression models were used to determine the relationship between patient characteristics and the presence and type of hypersomnia.


Amongst patients completing PSG and MSLT, 80% had polygraphic evidence of excessive daytime sleepiness. Hypersomnia due to medical condition was diagnosed in 45% and narcolepsy in 35%. Overweight or obese patients were more likely to be diagnosed with hypersomnia (P = 0.012) or narcolepsy (P = 0.009). Grade 2 hypothalamic involvement (HI) at diagnosis was associated with the diagnosis of narcolepsy (P = 0.0008).


This study describes the prevalence and associated predictors of hypersomnia for patients with craniopharyngioma after surgical resection. HI was predictive of narcolepsy diagnosis, and a higher body mass index z-score was associated with hypersomnia due to medical condition and narcolepsy. We recommend that sleep assessment and intervention begin after surgical resection, especially in overweight or obese patients and those with extensive tumors.

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  1. The MSLT is considered the “gold standard” for objective characterization of daytime sleepiness, but normative data from typically developing children and uniformly accepted cut-off values for pathological sleepiness in children are lacking. We chose a cut-off value for mean sleep latency of < 15 min in prepubescent children and < 10 min in pubescent youth. This is a deviation from the ICSD-3 recommendation (cut-off value of < 8 min), which refers to adults.

  2. Refer Footnote 1.


  1. Muller HL, Gebhardt U, Teske C, Faldum A, Zwiener I, Warmuth-Metz M, Pietsch T, Pohl F, Sorensen N, Calaminus G, Study Committee of K (2011) Post-operative hypothalamic lesions and obesity in childhood craniopharyngioma: results of the multinational prospective trial KRANIOPHARYNGEOM 2000 after 3-year follow-up. Eur J Endocrinol 165(1):17–24.

    Article  CAS  PubMed  Google Scholar 

  2. Ozyurt J, Thiel CM, Lorenzen A, Gebhardt U, Calaminus G, Warmuth-Metz M, Muller HL (2014) Neuropsychological outcome in patients with childhood craniopharyngioma and hypothalamic involvement. J Pediatr 164(4):876–881.

    Article  PubMed  Google Scholar 

  3. Muller HL (2016) Preoperative staging in childhood craniopharyngioma: standardization as a first step towards improved outcome. Endocrine 51(1):1–3.

    Article  CAS  PubMed  Google Scholar 

  4. Fournier-Goodnight AS, Ashford JM, Merchant TE, Boop FA, Indelicato DJ, Wang L, Zhang H, Conklin HM (2017) Neurocognitive functioning in pediatric craniopharyngioma: performance before treatment with proton therapy. J Neurooncol 134(1):97–105.

    Article  PubMed  PubMed Central  Google Scholar 

  5. Brzezinski A (1997) Melatonin in humans. N Engl J Med 336(3):186–195.

    Article  CAS  PubMed  Google Scholar 

  6. Jacola LM, Conklin HM, Scoggins MA et al (2016) Investigating the role of hypothalamic tumor involvement in sleep and cognitive outcomes among children treated for craniopharyngioma. J Pediatr Psychol 41:610

    Article  Google Scholar 

  7. Khan RB, Merchant TE, Sadighi ZS, Bello MS, Lu Z, Sykes A, Wise MS, Crabtree VM, Zabrowski J, Simmons A, Clark ME, Mandrell BN (2018) Prevalence, risk factors, and response to treatment for hypersomnia of central origin in survivors of childhood brain tumors. J Neurooncol 136(2):379–384.

    Article  PubMed  Google Scholar 

  8. Mandrell BN, Wise M, Schoumacher RA, Pritchard M, West N, Ness KK, Crabtree VM, Merchant TE, Morris B (2012) Excessive daytime sleepiness and sleep-disordered breathing disturbances in survivors of childhood central nervous system tumors. Pediatr Blood Cancer 58(5):746–751.

    Article  PubMed  Google Scholar 

  9. Muller HL (2010) Increased daytime sleepiness in patients with childhood craniopharyngioma and hypothalamic tumor involvement: review of the literature and perspectives. Int J Endocrinol 2010:519607.

    Article  PubMed  PubMed Central  Google Scholar 

  10. Cohen M, Guger S, Hamilton J (2011) Long term sequelae of pediatric craniopharyngioma—literature review and 20 years of experience. Front Endocrinol (Lausanne) 2:81.

    Article  Google Scholar 

  11. Berry RB, Gamaldo CE, Harding SM, Brooks R, Lloyd RM, Vaughn BV, Marcus CL (2015) AASM scoring manual version 2.2 updates: new chapters for scoring infant sleep staging and home sleep apnea testing. J Clin Sleep Med 11(11):1253–1254.

    Article  PubMed  PubMed Central  Google Scholar 

  12. Littner MR, Kushida C, Wise M, Davila DG, Morgenthaler T, Lee-Chiong T, Hirshkowitz M, Daniel LL, Bailey D, Berry RB, Kapen S, Kramer M, Standards of Practice Committee of the American Academy of Sleep M (2005) Practice parameters for clinical use of the multiple sleep latency test and the maintenance of wakefulness test. Sleep 28(1):113–121.

    Article  PubMed  Google Scholar 

  13. Gozal D, Wang M, Pope DW Jr (2001) Objective sleepiness measures in pediatric obstructive sleep apnea. Pediatrics 108(3):693–697.

    Article  CAS  PubMed  Google Scholar 

  14. Varni JW, Burwinkle TM, Katz ER, Meeske K, Dickinson P (2002) The PedsQL in pediatric cancer: reliability and validity of the pediatric quality of life inventory generic core scales, multidimensional fatigue scale, and cancer module. Cancer 94(7):2090–2106.

    Article  PubMed  Google Scholar 

  15. Palmer SN, Meeske KA, Katz ER, Burwinkle TM, Varni JW (2007) The PedsQL brain tumor module: initial reliability and validity. Pediatr Blood Cancer 49(3):287–293.

    Article  PubMed  Google Scholar 

  16. Muller HL, Muller-Stover S, Gebhardt U, Kolb R, Sorensen N, Handwerker G (2006) Secondary narcolepsy may be a causative factor of increased daytime sleepiness in obese childhood craniopharyngioma patients. J Pediatr Endocrinol Metab 19(Suppl 1):423–429.

    Article  PubMed  Google Scholar 

  17. Postiglione E, Antelmi E, Pizza F, Lecendreux M, Dauvilliers Y, Plazzi G (2018) The clinical spectrum of childhood narcolepsy. Sleep Med Rev 38:70–85.

    Article  PubMed  Google Scholar 

  18. Weil AG, Muir K, Hukin J, Desautels A, Martel V, Perreault S (2018) Narcolepsy and hypothalamic region tumors: presentation and evolution. Pediatr Neurol 84:27–31.

    Article  PubMed  Google Scholar 

  19. 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(8):3253–3262.

    Article  CAS  PubMed  Google Scholar 

  20. Crabtree VM, Klages KL, Sykes A, Wise MS, Lu Z, Indelicato D, Merchant TE, Avent Y, Mandrell BN (2019) Sensitivity and specificity of the modified epworth sleepiness scale in children with craniopharyngioma. J Clin Sleep Med 15(10):1487–1493.

    Article  PubMed  Google Scholar 

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The authors thank the patients and their families who participated in this study. This work was supported by Cancer Center Support Grant (CA21765) from the National Cancer Institute and the ALSAC.

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Correspondence to Belinda N. Mandrell.

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Mandrell, B.N., LaRosa, K., Hancock, D. et al. Predictors of narcolepsy and hypersomnia due to medical disorder in pediatric craniopharyngioma. J Neurooncol 148, 307–316 (2020).

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