Advertisement

Sleep and Breathing

, Volume 16, Issue 2, pp 427–434 | Cite as

Catathrenia under sodium oxybate in narcolepsy with cataplexy

  • Francesca Poli
  • Lara Ricotta
  • Stefano Vandi
  • Christian Franceschini
  • Fabio Pizza
  • Vincenzo Palaia
  • Keivan Kaveh Moghadam
  • Donatella Banal
  • Roberto Vetrugno
  • Michael J. Thorpy
  • Giuseppe PlazziEmail author
Original Article

Abstract

Purpose

This study aims to report on catathrenia occurring in narcolepsy with cataplexy (NC) patients under sodium oxybate (SO) treatment. Catathrenia is a parasomnia characterized by groaning and an abnormal respiratory pattern during sleep.

Methods

Fifty-one patients with NC and starting SO therapy underwent a baseline overnight polysomnography (PSG) to detect any sleep-related breathing disorders (SRBD). To avoid risks due to a possible central respiratory control depression by SO, all patients with concomitant obstructive sleep apnea (OSA) were treated with a nasal continuous positive airway pressure (nCPAP) device. After 2 months of treatment with SO, all patients underwent a follow-up overnight PSG to investigate possible newly occurring SRBD. They also underwent a semi-structured clinical interview to monitor other potential SO side effects.

Results

At baseline, four out of 51 patients showed simple snoring, and eight, mild to severe OSA. After a titration PSG night, patients with OSA received a nCPAP device. After 2 months of SO treatment, 28 patients (54.9%) showed SO-related side effects, including SRBD in 11 (21.6%). The follow-up PSG showed a respiratory pattern characteristic of catathrenia in seven patients (13.7%) as a newly observed and possibly benign SO side effect, and ruled out a worsening of OSA.

Conclusions

Catathrenia should be considered a possible side effect in NC patients under SO treatment and should be accurately identified to prevent unnecessary SO withdrawal.

Keywords

Groaning Parasomnia Sleep apnea Sleep-related breathing disorders Side effect 

Notes

Acknowledgments

We thank Ms. A. Collins for the English revision and Ms. A. Laffi for helping in manuscript editing (Department of Neurological Sciences, University of Bologna, Italy).

Conflict of interest

Michael Thorpy is a consultant for Cephalon and Jazz Pharmaceuticals. Giuseppe Plazzi is a consultant for Cephalon and UCB Pharma. The other authors declare that they have no conflict of interest.

Supplementary material

ESM 1

Catathrenia video clip showing catathrenia episodes in a NC patient after 2 months under SO treatment (MPG 13,479 kb).

References

  1. 1.
    Yoss RE, Daly DD (1960) Narcolepsy. Arch Intern Med 106:168–171PubMedCrossRefGoogle Scholar
  2. 2.
    Plazzi G, Serra L, Ferri R (2008) Nocturnal aspects of narcolepsy with cataplexy. Sleep Med Rev 12:109–128PubMedCrossRefGoogle Scholar
  3. 3.
    Broughton R, Mamelak M (1979) The treatment of narcolepsy-cataplexy with nocturnal gamma-hydroxybutyrate. Can J Neurol Sci 6:1–6PubMedGoogle Scholar
  4. 4.
    Mamelak M, Scharf MB, Woods M (1986) Treatment of narcolepsy with gamma-hydroxybutyrate. A review of clinical and sleep laboratory findings. Sleep 9:285–289PubMedGoogle Scholar
  5. 5.
    Thorpy MJ (2008) Sodium oxybate for the treatment of narcolepsy. Expert Opin Pharmacother 6:329–335CrossRefGoogle Scholar
  6. 6.
    Black J, Pardi D, Hornfeldt CS, Inhaber N (2009) The nightly administration of sodium oxybate results in significant reduction in the nocturnal sleep disruption of patients with narcolepsy. Sleep Med 10:829–835PubMedCrossRefGoogle Scholar
  7. 7.
    Huang YS, Guilleminault C (2009) Narcolepsy: action of two gamma-aminobutyric acid type B agonists, baclofen and sodium oxybate. Pediatr Neurol 41:9–16PubMedCrossRefGoogle Scholar
  8. 8.
    Vienne J, Bettler B, Franken P, Tafti M (2010) Differential effects of GABAB receptor subtypes, {gamma}-hydroxybutyric acid, and baclofen on EEG activity and sleep regulation. J Neurosci 30:194–204CrossRefGoogle Scholar
  9. 9.
    Akins BE, Miranda E, Lacy JM, Logan BK (2009) A multi-drug intoxication fatality involving Xyrem (GHB). J Forensic Sci 54:495–496PubMedCrossRefGoogle Scholar
  10. 10.
    Zvosec DL, Smith SW, Hall BJ (2009) Three deaths associated with use of Xyrem. Sleep Med 10:490–493PubMedCrossRefGoogle Scholar
  11. 11.
    Feldman NT (2009) Xyrem safety: the debate continues. Sleep Med 10:405–406PubMedCrossRefGoogle Scholar
  12. 12.
    Feldman NT (2010) Clinical perspective: monitoring sodium oxybate-treated narcolepsy patients for the development of sleep-disordered breathing. Sleep Breath 14:77–79PubMedCrossRefGoogle Scholar
  13. 13.
    George CF, Feldman N, Inhaber N, Steininger TL, Grzeschik SM, Lai C, Zheng Y (2010) A safety trial of sodium oxybate in patients with obstructive sleep apnea: acute effects on sleep-disordered breathing. Sleep Med 11:38–42PubMedCrossRefGoogle Scholar
  14. 14.
    Kovacević-Ristanović R, Kuźniar TJ (2010) Use of sodium oxybate (Xyrem) in patients with dual diagnosis of narcolepsy and sleep apnea. Sleep Med 11:5–6PubMedCrossRefGoogle Scholar
  15. 15.
    Wang YG, Swick TJ, Carter LP, Thorpy MJ, Benowitz NL (2009) Safety overview of postmarketing and clinical experience of sodium oxybate (Xyrem): abuse, misuse, dependence, and diversion. J Clin Sleep Med 5:365–371PubMedGoogle Scholar
  16. 16.
    Seeck-Hirschner M, Baier PC, von Freier A, Aldenhoff J, Göder R (2009) Increase in sleep-related breathing disturbances after treatment with sodium oxybate in patients with narcolepsy and mild obstructive sleep apnea syndrome: two case reports. Sleep Med 10:154–155PubMedCrossRefGoogle Scholar
  17. 17.
    Mamelak M, Webster P (1981) Treatment of narcolepsy and sleep apnea with gammahydroxybutyrate: a clinical and polysomnographic case study. Sleep 4:105–111PubMedGoogle Scholar
  18. 18.
    Sériès F, Sériès I, Cormier Y (1992) Effects of enhancing slow-wave sleep by gamma-hydroxybutyrate on obstructive sleep apnea. Am Rev Respir Dis 145:1378–1383PubMedGoogle Scholar
  19. 19.
    Ortega-Albas JJ, López-Bernabé R, Diaz JR, Serrano AL (2010) Sodium oxybate and breathing. Sleep Breath 14:259PubMedCrossRefGoogle Scholar
  20. 20.
    Chokroverty S (1986) Sleep apnea in narcolepsy. Sleep 9:250–253PubMedGoogle Scholar
  21. 21.
    Sansa G, Iranzo A, Santamaria J (2010) Obstructive sleep apnea in narcolepsy. Sleep Med 11:93–95PubMedCrossRefGoogle Scholar
  22. 22.
    American Academy of Sleep Medicine (2005) International classification of sleep disorders, 2nd ed.: diagnostic and coding manual. American Academy of Sleep Medicine, WestchesterGoogle Scholar
  23. 23.
    Murali H, Kotagal S (2006) Off-label treatment of severe childhood narcolepsy-cataplexy with sodium oxybate. Sleep 29:1025–1029PubMedGoogle Scholar
  24. 24.
    Vetrugno R, Provini F, Plazzi G, Vignatelli L, Lugaresi E, Montagna P (2001) Catathrenia (nocturnal groaning): a new type of parasomnia. Neurology 56:681–683PubMedGoogle Scholar
  25. 25.
    Pevernagie DA, Boon PA, Mariman AN, Verhaeghen DB, Pauwels RA (2001) Vocalization during episodes of prolonged expiration: a parasomnia related to REM sleep. Sleep Med 2:19–30PubMedCrossRefGoogle Scholar
  26. 26.
    Oldani A, Manconi M, Zucconi M, Castronovo V, Ferini-Strambi L (2005) ‘Nocturnal groaning’: just a sound or parasomnia? J Sleep Res 14:305–310PubMedCrossRefGoogle Scholar
  27. 27.
    Vetrugno R, Lugaresi E, Plazzi G, Provini F, D'Angelo R, Montagna P (2007) Catathrenia (nocturnal groaning): an abnormal respiratory pattern during sleep. Eur J Neurol 14:1236–1243PubMedCrossRefGoogle Scholar
  28. 28.
    Guilleminault C, Hagen CC, Khaja AM (2008) Catathrenia: parasomnia or uncommon feature of sleep disordered breathing? Sleep 31:132–139PubMedGoogle Scholar
  29. 29.
    Ramar K, Olson EJ, Morgenthaler TI (2008) Catathrenia. Sleep Med 9:457–459PubMedCrossRefGoogle Scholar
  30. 30.
    Prihodova I, Sonka K, Kemlink D, Volna J, Nevsimalova S (2009) Arousals in nocturnal groaning. Sleep Med 10:1051–1055PubMedCrossRefGoogle Scholar
  31. 31.
    Laborit H (1964) Sodium 4-hydroxybutyrate. Int J Neuropharmacol 13:433–451Google Scholar
  32. 32.
    Alshaikh MK, Gacuan D, George S, Sharif M, Bahammam AS (2011) Long-term follow-up of patients with narcolepsy-cataplexy treated with sodium oxybate (Xyrem). Clin Neuropharmacol 34:1–4PubMedCrossRefGoogle Scholar
  33. 33.
    Lammers GJ, Bassetti C, Billiard M, Black J, Broughton R, Dauvilliers Y, Ferini Strambi L, Garcia-Borreguero D, Goswami M, Högl B, Iranzo A, Jennum P, Khatami R, Lecendreux M, Mayer G, Mignot E, Montplaisir J, Nevsimalova S, Peraita-Adrados R, Plazzi G, Scammell T, Silber M, Sonka K, Tafti M, Thorpy M (2010) Sodium oxybate is an effective and safe treatment for narcolepsy. Sleep Med 11:105–106PubMedCrossRefGoogle Scholar

Copyright information

© Springer-Verlag 2011

Authors and Affiliations

  • Francesca Poli
    • 1
  • Lara Ricotta
    • 1
  • Stefano Vandi
    • 1
  • Christian Franceschini
    • 1
  • Fabio Pizza
    • 1
  • Vincenzo Palaia
    • 1
  • Keivan Kaveh Moghadam
    • 1
  • Donatella Banal
    • 2
  • Roberto Vetrugno
    • 1
  • Michael J. Thorpy
    • 3
  • Giuseppe Plazzi
    • 1
    Email author
  1. 1.Department of Neurological SciencesUniversity of BolognaBolognaItaly
  2. 2.Sleep Disorders CenterSan Lorenzino HospitalCesenaItaly
  3. 3.Sleep–Wake Disorders CenterMontefiore Medical CenterNew York CityUSA

Personalised recommendations