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Cited by 15Pituitary Gland Disorders and TreatmentsSleep and Wakefulness ResearchCircadian rhythm and melatonin

Sleep Disturbances in Pediatric Craniopharyngioma: A Systematic Review

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Cordani, Ramona, Veneruso, Marco, Napoli, Flavia, Di Iorgi, Natascia, Milanaccio, Claudia, Consales, Alessandro, Disma, Nicola, De Grandis, Elisa +2 more·2022·Frontiers in Neurology
DOI 10.3389/fneur.2022.876011PMC PMC9150678
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Abstract

Craniopharyngiomas are rare brain tumors of the sellar region and are the most common non-neuroepithelial intracerebral neoplasm in children. Despite a low-grade histologic classification, craniopharyngiomas can have a severe clinical course due to hypothalamic involvement. The hypothalamus plays a crucial role in regulating vital functions, and it is a critical component of the sleep-wake regulatory system. This systematic review aims to provide an overview of the current knowledge on sleep disorders in patients with craniopharyngioma to unravel their underlying mechanisms and identify possible therapeutic strategies. A comprehensive electronic literature search of the PubMed/MEDLINE and Scopus databases was conducted in accordance with the PRISMA ® statement. Extensively published, peer-reviewed articles involving patients with childhood craniopharyngioma and focused on this specific topic were considered eligible for inclusion. Thirty-two articles were included; a high prevalence of excessive daytime sleepiness was reported in CP patients, with wide variability (25–100%) depending on the diagnostic method of detection (25–43% by subjective measures, 50–100% by objective investigations). In particular, secondary narcolepsy was reported in 14–35%, sleep-disordered breathing in 4–46%. Moreover, sleep-wake rhythm dysregulation has been notified, although no prevalence data are available. Possible mechanisms underlying these disorders are discussed, including hypothalamic injury, damage to the suprachiasmatic nucleus, low melatonin levels, hypocretin deficiency, and hypothalamic obesity. The diagnosis and management of sleep disorders and associated comorbidities are challenging. This review summarizes the pathophysiology of sleep disorders in childhood-onset CP and the main treatment options. Finally, a possible diagnostic algorithm in order to accurately identify and treat sleep disorders in these patients is proposed.

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  • Continuous positive airway pressure treatment has not consistently been shown to improve excess daytime sleepiness in those with sleep-disordered breathing.

    “Although continuous positive airway pressure treatment has not consistently been shown to improve excess daytime sleepiness in those with sleep-disordered breathing (67), its use remains strongly recommended because it avoids frequent apneic episodes and potential sequelae of untreated obstructive sleep apnea, including pulmonary hypertension.”

    From Approach to the patient with acquired hypothalamic syndrome (2026) · cites this paper as [67]

    The Discussion says noninvasive ventilation benefits sleep-related breathing disorders but rarely reduces daytime sleepiness. Table 3 reports mixed sleepiness outcomes with NIV/CPAP. Those findings concern patients with craniopharyngioma, so they do not establish the claim for people with sleep-disordered breathing generally; page numbers were not provided in our copy. (LLM, conf=0.85)

    Experimental — accuracy not yet measured

  • Excessive daytime sleepiness from narcolepsy, sleep-disordered breathing, and/or circadian rhythm dysregulation has been reported in 25% to 100% of individuals with craniopharyngioma, depending on cohort and tools.

    “Excessive daytime sleepiness from narcolepsy, sleep-disordered breathing, and/or circadian rhythm dysregulation has been reported in from 25% to 100% of individuals with craniopharyngioma, depending on the cohort and tools used to assess (67).”

    From Approach to the patient with acquired hypothalamic syndrome (2026) · cites this paper as [67]

  • Type 2 narcolepsy (sleepiness without cataplexy) is associated with hypothalamic injury.

    “The etiology of type 2 narcolepsy (sleepiness without cataplexy) is less clear but is also associated with hypothalamic injury (67).”

    From Approach to the patient with acquired hypothalamic syndrome (2026) · cites this paper as [67]

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Authors
Cordani, R., Veneruso, M., Napoli, F., Di Iorgi, N., Milanaccio, C., Consales, A., Disma, N., De Grandis, E., Maghnie, M., & Nobili, L.
Affiliations
  • Istituto Giannina Gaslini (IT)
  • University of Genoa (IT)
Corresponding author
  • Maghnie, Mohamad
  • Nobili, Lino
ORCID
Venue
Frontiers in Neurology
Publisher
Frontiers Media SA
Published
2022-05-13
Volume
13
Pages
876011-876011
ISSN
1664-2295 (ISSN-L)
DOI
10.3389/fneur.2022.876011
PMC
PMC9150678
OpenAlex
W4280646661
Cited by
15
References
3
Citations by year
2026: 4 · 2025: 8 · 2024: 1 · 2023: 2
FWCI
0.39
Type
journal article
Language
en
Open access
Licence
Locations
  • Frontiers in Neurology (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
  • CINECA IRIS Institutial Research Information System (University of Genoa) (submittedVersion)
Funders
  • Ministero della Salute (2018-, "5 x 1000".)
  • Ministero dell’Istruzione, dell’Università e della Ricerca (20182022, MIUR 2018-2022, 232/2016)
Topics
  • Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Pituitary Gland Disorders and Treatments
  • Life Sciences › Neuroscience › Cognitive Neuroscience › Sleep and Wakefulness Research
  • Life Sciences › Neuroscience › Endocrine and Autonomic Systems › Circadian rhythm and melatonin
Keywords
craniopharyngioma, pediatric brain tumors, sleep disorders, excessive daytime sleepiness, sleep-disordered breathing, hypothalamic dysfunction, suprachiasmatic nucleus, melatonin, hypocretin deficiency, hypothalamic obesity

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