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Review
. 2022 Sep 1;18(9):2143-2154.
doi: 10.5664/jcsm.10062.

Sleep-disordered breathing in patients with Chiari malformation type II: a case-control study and review of the literature

Ilaria Lazzareschi  1 , Antonietta Curatola  1 , Luca Massimi  2 , Claudia Rendeli  1 , Eleonora Rollo  3 , Irene Scala  3 , Giacomo Della Marca  4 , Valerio Brunetti  4
Affiliations
Review

Sleep-disordered breathing in patients with Chiari malformation type II: a case-control study and review of the literature

Ilaria Lazzareschi et al. J Clin Sleep Med. .

Abstract

Study objectives: The main aim was to evaluate the prevalence of sleep-disordered breathing (SDB) in patients with Chiari II malformation (CM-II). The secondary objectives were to evaluate the association between SDB, morphological abnormalities, and neurological symptoms and to review the literature on patients with SDB and CM-II.

Methods: The study has a cross-sectional, case-control design. Patients with CM-II (patients) were compared to control patients referred for clinical polysomnography in the Sleep Medicine Unit, matched for age and sex. All patients underwent brain and spinal cord magnetic resonance imaging, and polysomnography was conducted for all participants. A review of the literature about SDB in patients with CM-II was performed.

Results: Forty patients were included (20 patients vs 20 control patients). SDB was identified in 45% of patients, a significantly higher prevalence compared to control patients. Three patients presented with purely obstructive SDB, 3 patients with purely central SDB, and 3 patients with both obstructive and central SDB. Compared with control patients, patients with CM-II showed a higher oxygen desaturation index (median: CM-II, 3.7; interquartile range, 1.6-19.5; control patients: 1.1; interquartile range, 0.3-3.2) and obstructive apnea-hypopnea index (median: CM-II, 1.5; interquartile range, 0.5-5.1; control patients, 0.1; interquartile range, 0.0-0.7). A logistic regression showed that the risk of developing SDB in patients affected by CM-II was 14.7 times higher than in the control population.

Conclusions: Our study and literature review showed a high prevalence of SDB in patients with CM-II. These patients are often asymptomatic at diagnosis, suggesting that PSG should be routinely provided in this population.

Citation: Lazzareschi I, Curatola A, Massimi L, et al. Sleep-disordered breathing in patients with Chiari malformation type II: a case-control study and review of the literature. J Clin Sleep Med. 2022;18(9):2143-2154.

Keywords: Chiari II malformation; central sleep apnea; myelomeningocele; obstructive sleep apnea; personalized medicine; polysomnography; sleep; sleep-disordered breathing.

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Conflict of interest statement

All authors participated in a meaningful way in the preparation of the manuscript and agreed upon the manuscript content. The authors report no conflicts of interest.

Figures

Figure 1
Figure 1. Flow diagram of this study selection.
Flow diagram template from Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. doi: 10.1136/bmj.n71. CM-II = Chiari type II malformation.
Figure 2
Figure 2. Bar graphs comparing the prevalence of SDB and snoring in patients with CM-II and control patients.
CM-II = Chiari type II malformation, SDB = sleep-disordered breathing.
Figure 3
Figure 3. Comparisons of PSG parameters between patients with CM-II vs control patients.
Boxplots show O-AHI and ODI indices greater in patients with CM-II compared to control patients, whereas REM sleep SPT is higher in control patients. No difference emerges regarding C-AHI in the 2 groups. C-AHI = central apnea-hypopnea index, CM-II = Chiari type II malformation, O-AHI = obstructive apnea-hypopnea index, ODI = oxygen desaturation index, PSG = polysomnography, REM = rapid eye movement, SPT = sleep period time.
See this image and copyright information in PMC

References

    1. Charney EB , Rorke LB , Sutton LN , Schut L . Management of Chiari II complications in infants with myelomeningocele . J Pediatr. 1987. ; 111 ( 3 ): 364 – 371 . - PubMed
    1. Gilbert JN , Jones KL , Rorke LB , Chernoff GF , James HE . Central nervous system anomalies associated with meningomyelocele, hydrocephalus, and the Arnold-Chiari malformation: reappraisal of theories regarding the pathogenesis of posterior neural tube closure defects . Neurosurgery. 1986. ; 18 ( 5 ): 559 – 564 . - PubMed
    1. Kuhn J , Emmady PD . Chiari II Malformation. Treasure Island, FL: : StatPearls; ; 2021. .
    1. Sandler AD . Children with spina bifida: key clinical issues . Pediatr Clin North Am. 2010. ; 57 ( 4 ): 879 – 892 . - PubMed
    1. Krimsky WR , Leiter JC . Physiology of breathing and respiratory control during sleep . Semin Respir Crit Care Med. 2005. ; 26 ( 1 ): 5 – 12 . - PubMed

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