- Email: [email protected]
Maxillary expansion and maxillomandibular expansion for adult OSA: A systematic review and meta-analysis
Accepted Manuscript Maxillary Expansion and Maxillomandibular Expansion for Adult OSA: A Systematic Review and Meta-Analysis Jose Abdullatif, MD, Victor Certal, MD, PhD, Soroush Zaghi, MD, Sungjin A. Song, MD, Edward T. Chang, MD, MS, M. Boyd Gillespie, MD, MSc, Macario Camacho, MD PII:
S1010-5182(16)00043-3
DOI:
10.1016/j.jcms.2016.02.001
Reference:
YJCMS 2301
To appear in:
Journal of Cranio-Maxillo-Facial Surgery
Received Date: 2 November 2015 Revised Date:
15 January 2016
Accepted Date: 1 February 2016
Please cite this article as: Abdullatif J, Certal V, Zaghi S, Song SA, Chang ET, Gillespie MB, Camacho M, Maxillary Expansion and Maxillomandibular Expansion for Adult OSA: A Systematic Review and Meta-Analysis, Journal of Cranio-Maxillofacial Surgery (2016), doi: 10.1016/j.jcms.2016.02.001. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Jose Abdullatif, MD, Victor Certal, MD, PhD, Soroush Zaghi, MD, Sungjin A. Song MD, Edward T. Chang MD, MS, M. Boyd Gillespie MD, MSc, Macario Camacho, MD
SC
Jose Abdullatif, MD Department of Otorhinolaryngology Hospital Bernardino Rivadavia Buenos Aires, Argentina
RI PT
Maxillary Expansion and Maxillomandibular Expansion for Adult OSA: A Systematic Review and Meta-Analysis
M AN U
Victor Certal, MD, PhD a Department of Otorhinolaryngology/Sleep Medicine Centre – Hospital CUF Porto, Portugal b
CINTESIS – Centre for Research in Health Technologies and Information Systems University of Porto Porto, Portugal
TE D
Soroush Zaghi, MD Department of Otolaryngology-Head and Neck Surgery Division of Sleep Surgery Stanford Hospital and Clinics Stanford, CA, USA 95304
EP
Sungjin A. Song, MD Otolaryngology-Head and Neck Surgery Tripler Army Medical Center, Honolulu, HI, USA
AC C
Edward T. Chang, MD, MS Otolaryngology-Head and Neck Surgery Tripler Army Medical Center, Honolulu, HI, USA M. Boyd Gillespie, MD, MSc Department of Otolaryngology - Head and Neck Surgery Medical University of South Carolina, Charleston, SC, USA *Macario Camacho, MD *corresponding author and author to whom correspondence, reprint requests, and proofs will be sent a Otolaryngology-Head and Neck Surgery Division of Sleep Surgery and Medicine Tripler Army Medical Center 1 Jarrett White Road
ACCEPTED MANUSCRIPT
Honolulu, HI, USA 96859 Phone: 808-433-4883 Fax: 808-433-9033 Email: [email protected]
AC C
EP
TE D
M AN U
SC
Department of Psychiatry and Behavioral Sciences Sleep Medicine Division Stanford Hospital and Clinics Stanford, CA, USA 95304
RI PT
b
ACCEPTED MANUSCRIPT
Abstract Objective
RI PT
This study sought to systematically review the international literature for articles
evaluating maxillary expansion and maxillomandibular expansion as treatments for
Data Sources
M AN U
Nine databases (including MEDLINE/PubMed).
SC
obstructive sleep apnea (OSA) in adults and to perform a meta-analysis.
Review Methods
Searches were performed through January 8, 2016. The PRISMA statement was
Results
TE D
followed.
EP
Eight adult studies (39 patients) reported polysomnography and/or sleepiness outcomes. Six studies reported outcomes for maxillary expansion (36 patients), and the apnea-
AC C
hypopnea index (AHI) decreased from a mean (M) ± standard deviation (SD) of 24.3±27.5 [95% CI 15.3, 33.3] to 9.9±13.7 [95% CI 5.4, 14.4] events/hr (relative reduction: 59.3%). Maxillary expansion improved lowest oxygen saturation (LSAT) from a M±SD of 84.3±8.1% [95% CI 81.7, 87.0] to 86.9±5.6% [95% CI 85.1, 88.7]. Maxillomandibular expansion was reported in two studies (3 patients) and AHI decreased from a M±SD of 47.53±29.81 [95% CI -26.5 to 121.5] to 10.7±3.2 [95% CI 2.8, 18.6] events/hr (relative reduction: 77.5%). Maxillomandibular expansion improved LSAT
ACCEPTED MANUSCRIPT
from a M±SD of 76.7±14.5% [95% CI 40.7, 112.7] to 89.3±3.1 [95% CI 81.6, 97]. Conclusion
RI PT
The current literature demonstrates that maxillary expansion can improve and
maxillomandibular expansion can possibly improve AHI and LSAT in adults; however, given the paucity of studies, these remain open for additional research efforts.
SC
Key Words
Systematic Review; Meta-Analysis
TE D
Introduction
M AN U
Maxillary Expansion, Maxillomandibular Expansion, Sleep Apnea Syndromes;
Obstructive sleep apnea (OSA) is a common disorder in adults treated with either medical management (myofunctional therapy (Camacho et al 2015a), positional therapy,
EP
nasopharyngeal airway devices (Kumar et al 2014), continuous positive airway pressure devices, etc.) or with sleep surgeries. Sleep surgery for OSA initially consisted of
AC C
tracheostomies (Camacho et al 2014), however, over the years several additional surgeries have been performed such as tonsillectomy (Senchak et al 2015), uvulopalatopharyngoplasty, hypoglossal nerve stimulation (Certal et al 2015), maxillomandibular advancement (Camacho et al 2015b), etc.). Maxillary expansion has been demonstrated to improve OSA in children (Guilleminault et al 2008), however the effect of maxillary expansion in adults has not been studied as extensively.
ACCEPTED MANUSCRIPT
Transverse maxillary deficiency is a maxillofacial deformity that is characterized as a discrepant width of the maxilla relative to the mandible often due to asymmetric growth of the maxilla or mandible. Variations in the development and the size of the
RI PT
maxilla can lead to increased nasal airflow resistance and displacement of the tongue posteriorly (Vinha et al 2015). Because the floor of the nose is higher, patients with
transverse maxillary deficiency have a greater propensity to developing nasal airway
SC
obstruction, especially in the setting of a deviated nasal septum or large inferior
turbinates (Camacho et al 2015c). Transverse maxillary deficiency may be in fact be an
M AN U
under recognized finding among adult patients with nasal obstruction or OSA. For such patients, maxillary expansion increases the size of the nasal cavity thereby decreasing airway resistance (10% improvement in the size of the nasal cavity corresponds to a 21% improvement in airflow (Powell et al 2001)). By widening the maxilla in the transverse
TE D
dimension, it has been hypothesized that the increased oral cavity space allows for a better position of the tongue and a greater posterior pharyngeal airway space, with a subsequent reduction in airway obstruction (Vinha et al 2015). In children, the maxilla
EP
can be expanded daily between 0.4 to 0.8 mm without surgery by placing an intraoral appliance that is anchored by the teeth (Vinha et al 2015). In adults, the midpalatal
AC C
sutures of the maxilla are fused, therefore, in most cases surgery is used to assist maxillary expansion. Articles have also been published describing the additional expansion of the mandible in the transverse dimension (Guilleminault and Li 2004, Bonetti et al 2009). Given that there has been a limited amount of data with regard to the use of maxillary expansion and maxillomandibular expansion for OSA in adults, our objective was to systematically review the international literature for articles on these two
ACCEPTED MANUSCRIPT
treatments in adult OSA patients and to perform a meta-analysis on the available data.
RI PT
Materials and Methods
Three authors (J.A., E.T.C., and M.C.) independently searched the international
literature without regard to language from inception of each database through January 8,
SC
2016. Databases searched included The Cochrane Library, PubMed/MEDLINE, Scopus,
M AN U
Embase, Google Scholar, Web of Science, Book Citation Index – Science, Cumulative Index to Nursing and Allied Health (CINAHL) and Conference Proceedings Citation Index – Science from the inception of each database. An example of a search strategy for PubMed/MEDLINE is: ((((“sleep”) OR (“snoring”)) AND ((("Biobloc") OR (“Palatal Expansion Technique"[Mesh]) OR (“Maxilla”) OR ("Maxillary”) OR ("Palatal”) OR
TE D
("Palate”) OR (“Orthodontic”)) AND ((“Distraction”) OR (“Widening”) OR (“Expansion”)))) OR ((“Sleep”) AND (“Enuresis”) AND ((“Orthodontic”) OR
EP
(“Distraction”) OR (“Widening”) OR (“Expansion”)))). The Preferred Reporting Items for Systematic Reviews and Meta-Analysis
AC C
(PRISMA) statement was adhered to as much as possible in the performance of this review (Moher et al 2009). Because this is a systematic review of publically available articles, this study is exempted from review by any of the authors’ Institutional Review Boards.
Study selection
ACCEPTED MANUSCRIPT
Inclusion criteria for this review: (1) Patients: adults (≥18 years (y)) with OSA, (2) Intervention: isolated maxillary expansion or maxillomandibular expansion, (3) comparison: quantitative data pre- and post-maxillary expansion or maxillomandibular
RI PT
expansion, (4) Outcome: general outcomes, polysomnography data and sleepiness; and (5) Study design: all designs, published and unpublished studies, and all languages.
Exclusion criteria: (1) studies in which additional surgeries were also performed and
SC
there was no sub-stratification for maxillary expansion or maxillomandibular expansion; (2) studies with qualitative outcomes only; (3) patients with central sleep apnea; and (4)
weight (this can confound outcomes).
M AN U
studies in which individual patient data is reported and patients lost ≥10% of their body
TE D
Quality assessment of included studies
To evaluate the quality of studies, the National Institute for Health and Clinical Excellence (NICE) quality assessment tool (National Institute for Health and Clinical
EP
Excellence 2009) was utilized. This tool was selected because it permits for assessment
AC C
between studies, despite the studies being categorized as having the same level of evidence.
Statistics For this study, the null hypothesis was that there is no difference in polysomnography or sleepiness outcomes pre- and post-maxillary expansion or
ACCEPTED MANUSCRIPT
maxillomandibular expansion. For overall analyses, the IBM Statistical Package for Social Sciences software (SPSS) version 20.0 (Aramonk, New York) was utilized. When evaluating two-tailed, paired t-tests, a p-value <0.05 was considered statistically
RI PT
significant. For the meta-analysis, the Cochrane Collaboration’s Review Manager
Software (REVMAN) version 5.3 was used. The calculations for the mean differences (MD), standardized mean differences (SMD) and 95% confidence intervals [95% CI]
SC
were performed with REVMAN. For interpreting the magnitudes of effect for
standardized mean differences, Cohen’s guidelines (Cohen 1988) were followed and the
M AN U
assigned effects were: small = 0.2, medium = 0.5, and large = 0.8.
Heterogeneity was evaluated between studies by using REVMAN to calculate the Cochran Q statistic (Q statistic), and as recommended, a p-value ≤0.10 was used as the cutoff for statistically significant heterogeneity. (Lau et al 1997) REVMAN was utilized
TE D
to calculate the Inconsistency (I2 statistic), and levels of inconsistency guidelines were used to assign levels: low inconsistency = 25%, moderate inconsistency = 50% and high inconsistency = 75%.( Higgins et al 2003) In evaluating the heterogeneity and
EP
inconsistency, if they were present, then REVMAN’s sensitivity analysis function was
AC C
used by removing one study at a time until heterogeneity and/or inconsistency were not present, therefore, identifying which study or studies were responsible. For evaluation of polysomnography data, when the AHI and respiratory disturbance index (RDI) were reported within a single study, then the AHI was used for combining data and metaanalysis. If however, a study reported only the RDI then the plan was to contact the authors at least twice in order to obtain the AHI. The plan for a funnel plot assessment for bias if at least ten studies were identified for any given variable (as recommended by the
ACCEPTED MANUSCRIPT
Cochrane Collaboration). If any additional data were needed for meta-analysis, then the
RI PT
plan was also to contact the corresponding author at least twice to try to obtain the data.
Results
The systematic review searches provided a total of 207 potentially relevant
SC
studies, of which 98 were downloaded. Eight adult studies (39 patients) reported
M AN U
polysomnography and/or sleepiness outcomes. The case series studies met between four to seven out of the eight possible NICE quality assessment criteria, corresponding to
Maxillary Expansion
TE D
moderate to high quality studies.
Six studies (Palmisano et al 1996, Cistulli et al 1998, Belfor et al 2010, Bach et al
EP
2013, Singh et al 2013, Vinha et al 2015) reported outcomes for maxillary expansion (36 patients) and AHI decreased from a mean (M) ± standard deviation (SD) of 24.3±27.5
AC C
[95% CI 15.3, 33.3] to 9.9±13.7 [95% CI 5.4, 14.4] events/hr (relative reduction: 59.3%). A sub-analysis using random effects modelling demonstrated a mean difference of -9.71 [95% CI -15.41, -4.01] events/hr, overall effect Z score = 3.34 (p = 0.0008), Q statistic p = 0.10 (statistically significant heterogeneity), I2 = 57% (moderate inconsistency). After the removal of the study by Bach et al., there was no heterogeneity (Q statistic = 0.55) and there was no inconsistency (I2 = 0%). The standardized mean difference (SMD) for maxillary expansion demonstrated a large magnitude of effect for AHI (-1.40 [95% CI -
ACCEPTED MANUSCRIPT
2.67, -0.13], overall all effect Z score = 2.17 (p = 0.03), Q statistic p = 0.01 (statistically significant heterogeneity) and the I2 = 78% (high inconsistency). Removal of the study by Cistulli et al. (Cistulli et al 1998) resulted in no heterogeneity (p = 0.52) and no
RI PT
inconsistency (I2 = 0%).
Maxillary expansion improved LSAT from a M±SD of 84.3±8.1% [95% CI 81.7,
SC
87.0] to 86.9±5.6% [95% CI 85.1, 88.7]. The mean LSAT difference was 2.03 [95% CI 1.16, 2.89], overall effect Z score 4.59 (p <0.00001), Q statistic 0.72 (no heterogeneity)
M AN U
and I2 = 0% (no inconsistency). The LSAT SMD was 1.09 [95% CI -0.41, 2.59] (large magnitude of effect), overall effect Z score = 1.42 (p = 0.15), Q statistic p = 0.02 (statistically significant heterogeneity), I2 = 81% (high inconsistency). Sleepiness was reported in the study by Vinha et al. (Vinha et al 2015) with an
TE D
Epworth sleepiness scale (Johns et al 1991) (ESS) pre and post-maxillary expansion
EP
M±SD value of 12.5±5.3 and 7.2±3.5, respectively.
Maxillomandibular Expansion
AC C
Maxillomandibular expansion was reported in two studies (Guilleminault and Li
2004, Bonetti et al 2009) (3 patients) and AHI decreased from a M±SD of 47.53±29.81 [95% CI -26.5 to 121.5] to 10.7±3.2 [95% CI 2.8, 18.6] events/hr (relative reduction: 77.5%). Maxillomandibular expansion improved LSAT from a M±SD of 76.7±14.5% [95% CI 40.7, 112.7] to 89.3±3.1 [95% CI 81.6, 97]. A sub-analysis with random effects modelling could not be performed given there was only one patient in the study by
ACCEPTED MANUSCRIPT
Bonetti et al (Bonetti et al 2009). Sleepiness was reported for 2 patients in the study by Guilleminault and Li (Guilleminault and Li 2004), with improvement from an pre and
RI PT
post-maxillomandibular expansion M±SD of 10.5±0.71 and 7.5±0.71, respectively.
Discussion
SC
There are four main findings from this systematic review and meta-analysis. First,
M AN U
maxillary expansion can improve AHI, and maxillomandibular expansion potentially can improve AHI. There were thirty-six patients in the maxillary expansion studies and three patients in the maxillomandibular expansion studies. Overall, maxillary expansion reduced AHI by 59.3% and maxillomandibular expansion reduced AHI by 77.5%. Six studies have reported outcomes for maxillary expansion and AHI, while only two studies
TE D
have been published for maxillomandibular expansion and AHI. It has been hypothesized that maxillary expansion improves OSA variables by widening the floor of the nose and improving the position of the tongue. We additionally hypothesize that by widening the
EP
maxilla, there is tension on the muscles that attach to the palate such as the palatoglossus
AC C
and palatopharyngeus, with associated anterior displacement of these muscles, which can therefore reduce the collapsibility of the upper airway during sleep. It is logical that the addition of transverse expansion of the mandible would improve AHI even more; however, additional studies are needed to confirm this hypothesis as there have only been three patients in the literature. Second, LSAT has improved following both maxillary expansion and maxillomandibular expansion. However, there are fewer studies reporting outcomes for
ACCEPTED MANUSCRIPT
LSAT than AHI. There were only two studies reporting outcomes in twenty-six patients for LSAT outcomes in maxillary expansion patients and only two studies reporting LSAT outcomes in maxillomandibular expansion (three patients). The improvement was
RI PT
actually not that significant clinically for maxillary expansion, with a 2.6 point
improvement; however, there was a more clinically significant improvement (12.6 points)
given that there were only three patients.
SC
for maxillomandibular expansion. We caution against drawing conclusions for LSAT
M AN U
Third, sleepiness seems to decrease for both patients who have undergone either maxillary expansion or maxillomandibular expansion. For maxillary expansion, Vinha et al. (Vinha et al 2015) reported an improvement in ESS from a pre and post-maxillary expansion M±SD value of 12.5±5.3 and 7.2±3.5, respectively. For maxillomandibular expansion, sleepiness was reported for 2 patients in one study (Guilleminault and Li)
TE D
(Guilleminault and Li 2004), with improvement from a pre and post-maxillomandibular expansion M±SD of 10.5±0.71 and 7.5±0.71, respectively. Therefore, in the patients who were treated with either maxillary expansion or maxillomandibular expansion the patients
EP
were hypersomnolent and afterwards they were not.
AC C
Fourth, additional research is needed for both maxillary expansion and especially
for maxillomandibular expansion. With thirty-six patients in the literature for maxillary expansion and only three patients undergoing maxillomandibular expansion, the need for more publications is recommended. The relative reduction in the MME was higher (77.5%) compared with the AHI relative reduction of the ME (59.3%); however, the number of cases that met the criteria for inclusion in the MME review was extremely low and no conclusions can be made. Because of this, more studies evaluating the effect of
ACCEPTED MANUSCRIPT
maxillomandibular expansion in OSA are needed. Questions that have not been addressed which could be studied include the effect of combining ME or MME with maxillomandibular advancements in selected patients. Additionally, in order to further
RI PT
evaluate the outcomes for OSA patients, the publication of individual patient data with variables such as age, BMI, prior surgeries, AHI, LSAT, mean time spent under 90%, oxygen desaturation index, hypopnea scoring criteria, and sleepiness outcomes
SC
(specifically the Epworth sleepiness scale (Johns et al 1991)) would allow for individual patient data meta-analyses. The reporting of complications with associated percentage
M AN U
rates for each complication would also facilitate determination of complication rates (such as the rates of wound infections, the need for revisions and bleeding rates).
TE D
Limitations
There are limitations to this study, first the number of studies reporting outcomes as treatment for OSA are limited (six maxillary expansion and two maxillomandibular
EP
expansion studies); however, the data for the published studies have consistently
AC C
demonstrated an improvement in OSA variables. Second, like any systematic review, it is possible that we missed identifying articles; to minimize this possibility, we had three authors independently search the international literature without regard to language. Third, given the limited number of studies that are currently published, we would like to encourage surgeons to publish their outcomes with pre and post-treatment means and standard deviations for variables such as apnea-hypopnea index, sleepiness and oxygen saturation data. Lastly, because there can be a publication bias against negative studies, it
ACCEPTED MANUSCRIPT
is possible that studies finding minimal or no differences in polysomnography variables were either not submitted for publication or were selected against; because there were not at least 10 studies, as recommended by the Cochrane Collaboration, we did not perform
RI PT
an analysis for publication bias as the funnel plot analysis would not be as accurate.
SC
Conclusion
Current literature demonstrates that maxillary expansion can improve and
M AN U
maxillomandibular expansion possibly can improve AHI and LSAT in adults. Additional
AC C
EP
TE D
research is needed, especially for maxillomandibular expansion.
ACCEPTED MANUSCRIPT
References "Methods for development of NICE public health guidance." London: National
RI PT
Institute for Health and Clinical Excellence 2009. Bach N, Tuomilehto H, Gauthier C, Papadakis A, Remise C, Lavigne F, Lavigne GJ and Huynh N. "The effect of surgically assisted rapid maxillary expansion on
M AN U
Rehabil 40(11): 818-825, 2013.
SC
sleep architecture: an exploratory risk study in healthy young adults." J Oral
Belfor TR. "Epigenetic orthodontics: facial and airway development." N Y State Dent J 76(6): 18-21, 2010.
Bonetti GA, Piccin O, Lancellotti L, Bianchi A and Marchetti C. "A case report
TE D
on the efficacy of transverse expansion in severe obstructive sleep apnea syndrome." Sleep Breath 13(1): 93-96, 2009. Camacho M, Certal V, Brietzke SE, Holty JE, Guilleminault C and Capasso R.
EP
"Tracheostomy as treatment for adult obstructive sleep apnea: a systematic review
AC C
and meta-analysis." Laryngoscope 124(3): 803-811, 2014. Camacho M, Certal V, Abdullatif J; Zaghi S; Ruoff C; Capasso R; Kushida C. Myofunctional Therapy to Treat Obstructive Sleep Apnea: A Systematic Review and Meta-analysis. Sleep; 38:669-675, 2015a. Camacho M, Liu SY, Certal V, Capasso R, Powell NB and Riley RW. "Large Maxillomandibular Advancements for Obstructive Sleep Apnea: An Operative
ACCEPTED MANUSCRIPT
Technique Evolved Over 30 Years." Journal of Cranio-Maxillofacial Surgery, 2015b.
RI PT
Camacho M, Zaghi S, Certal V, Abdullatif J, Means C, Acevedo J, Liu S, Brietzke SE, Kushida CA and Capasso R. "Inferior Turbinate classification
system, grades 1 to 4: Development and validation study." Laryngoscope 125(2):
SC
296-302, 2015c.
Certal VF, Zaghi S, Riaz M, Vieira A S, Pinheiro CT, Kushida C, Capasso R and
M AN U
Camacho M. "Hypoglossal nerve stimulation in the treatment of obstructive sleep apnea: A systematic review and meta-analysis." Laryngoscope 125(5): 12541264, 2015.
Cistulli PA, Palmisano RG and Poole MD. "Treatment of obstructive sleep apnea
TE D
syndrome by rapid maxillary expansion." Sleep 21(8): 831-835, 1998. Cohen J . Statistical Power Analysis for the Behavioral Sciences. Hillsdale, NJ,
EP
USA, Erlbaum, 1988.
Guilleminault C and Li K. "Maxillomandibular expansion for the treatment of
AC C
sleep-disordered breathing: preliminary result." Laryngoscope 114(5): 893-896, 2004.
Guilleminault C, Quo S, Huynh NT and Li K. "Orthodontic expansion treatment and adenotonsillectomy in the treatment of obstructive sleep apnea in prepubertal children." Sleep 31(7): 953-957, 2008.
ACCEPTED MANUSCRIPT
Higgins JP, Thompson SG, Deeks JJ and Altman DG . "Measuring inconsistency in meta-analyses." BMJ 327(7414): 557-560, 2003.
RI PT
Johns, MW . "A new method for measuring daytime sleepiness: the Epworth sleepiness scale." Sleep 14(6): 540-545, 1991.
Kumar AR, Guilleminault C, Certal V, Li D, Capasso R, Camacho M.
SC
Nasopharyngeal airway stenting devices for obstructive sleep apnoea: A
systematic review and meta-analysis. The Journal of laryngology and otology :1-
M AN U
9, 2014.
Lau J, Ioannidis JP and Schmid CH. "Quantitative synthesis in systematic reviews." Ann Intern Med 127(9): 820-826, 1997.
TE D
Moher D, Liberati A, Tetzlaff J and Altman DG. "Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement." PLoS Med 6(7): e1000097, 2009.
EP
Palmisano RG, Wilcox I, Sullivan CE and Cistulli PA. "Treatment of snoring and obstructive sleep apnoea by rapid maxillary expansion." Aust N Z J Med 26(3):
AC C
428-429, 1996.
Powell NB, Zonato AI, Weaver EM, Li K, Troell R, Riley RW and Guilleminault C. "Radiofrequency treatment of turbinate hypertrophy in subjects using continuous positive airway pressure: a randomized, double-blind, placebocontrolled clinical pilot trial." Laryngoscope 111(10): 1783-1790, 2001.
ACCEPTED MANUSCRIPT
Senchak AJ, McKinlay AJ, Acevedo J, Swain B, Tiu MC, Chen BS, Robitschek J, Ruhl DS, Williams LL, Camacho M, Frey WC and O'Connor PD. "The effect of tonsillectomy alone in adult obstructive sleep apnea." Otolaryngol Head Neck
RI PT
Surg 152(5): 969-973, 2015.
Singh GD and Callister JD. "Effect of a maxillary appliance in an adult with
SC
obstructive sleep apnea: a case report." Cranio 31(3): 171-175, 2013.
Vinha PP, Eckeli AL, Faria AC, Xavier SP and de Mello-Filho FV. "Effects of
M AN U
surgically assisted rapid maxillary expansion on obstructive sleep apnea and
AC C
EP
TE D
daytime sleepiness." Sleep Breath 2015.
Additional studies identified from reference lists N=5
Articles excluded N = 90 Pediatric studies = 77 No quantitative data = 6 Other treatment = 7
M AN U
Potentially relevant articles retrieved for detailed evaluation N = 93
Excluded articles: N = 113 Not OSA = 105 Review articles = 8
SC
Articles screened for relevance (after removal of duplicates) N = 207
RI PT
ACCEPTED MANUSCRIPT
Included Studies N=8
AC C
EP
TE D
Figure 1: Flow chart for study selection. Abbreviations: N = number of studies, and pts = patients.
SC
RI PT
ACCEPTED MANUSCRIPT
Figure 2: Mean difference in AHI pre and post-maxillary expansion. 95% confidence
AC C
EP
TE D
M AN U
intervals are provided.
ACCEPTED MANUSCRIPT
4
Canada
4
USA
5
NR Australia Australia
Italy
Quality assessment of included studies* 2 3 4 5
6
7
8
AHI, ESS, LSAT, RDI AHI, MSAT, SAT<90% AHI, ODI, snoring AHI, RDI
N
Y
Y
Y
Y
Y
Y
Y
N
Y
Y
N
Y
N
Y
N
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
4
AHI, LSAT
N
Y
Y
N
Y
N
Y
N
5
AHI
NA
NA
NA
NA
NA
NA
NA
NA
5
AHI, LSAT,
NA
NA
NA
NA
NA
NA
NA
NA
TE D
Brazil
EP
Maxillary Expansion Vinha et al. 2015, PCS Bach et al. 2013 PCS Singh et al. 2013 RCR Belfor et al. 2010 RCR Cistulli et al. 1998 PCS Palmisano et al. 1996 RCR Maxillomandibular Expansion Bonetti et al.
Study site
Study’s General Characteristics and number of included patients Evidence Outcomes 1 level analysed
5
AC C
Year, Study, Design
M AN U
SC
RI PT
Table 1. General characteristics and quality criteria of included studies. *Quality Assessment of cases series studies checklist from National Institute for Health and Clinical Excellence (NICE): 1) Was the case series collected in more than one centre, i.e. multicentre study? 2) Is the hypothesis/aim/objective of the study clearly described? 3) Are the inclusion and exclusion criteria (case definition) clearly reported? 4) Is there a clear definition of the outcomes reported? 5) Were data collected prospectively? 6) Is there an explicit statement that patients were recruited consecutively? 7) Are the main findings of the study clearly described? 8) Are outcomes stratified? (e.g., by abnormal results, disease stage, patient characteristics)? Abbreviations: AHI = Apnea-Hypopnea index; ESS = Epworth Sleepiness Scale; LSAT = lowest oxygen saturation; MSAT = mean oxygen saturation; N.A. = Not applicable; N.R. = Not reported; RCR = retrospective case report; RCS = retrospective case series; RDI = respiratory disturbance index; SAT<90% = time spent under oxygen saturation of 90%.
ACCEPTED MANUSCRIPT
M AN U TE D
Y
Y
RI PT
N
SC
4
EP
USA
SAT<90% AHI, ESS, LSAT
AC C
2009 RCR Guilleminault and Li 2004 PCS
Y
Y
Y
Y
Y
ACCEPTED MANUSCRIPT
Table 2: Demographic and polysomnographic data pre and post-rapid maxillary expansion or maxillomandibular expansion. Abbreviations: [ ] denotes lower and upper 95% confidence intervals; AHI = apnea-hypopnea index; low O2 = lowest oxygen saturation; mo = months; N = number; NR = not reported. *Total is based on means and standard deviations. **AHI change is based on the relative reduction in AHI N
16
40.2±10. 29.8±4. 2 4
Bach et al. 2013
7
NR
NR
Singh et al. 2013 Belfor et al. 2010 Cistulli et al. 1998
1
64
26
1
NR
NR
10
27±2
29.8±2. 6
Total*
3
PostOp AHI
AHI change
Pre-Op low O2
Post-Op low O2
14.5±19. -56.3 4 [5.0, 24.0]
-50.9%
NR
NR
-82.4%
NR
NR
28
5
-82.1
NR
NR
19±4 [16.5, 21.5] 20
7±4 [4.5, 9.5] 4
-63.2%
35.7±11. 29.9±3. 7 [31.9, 9 39.5] [28.6, 31.2]
24.3±27. 5 [15.3, 33.3]
37
M AN U
SC
Pre-Op AHI
5.7±5.4 [1.7, 9.7 ] 4.5
81.3±9.2 [76.8, 85.8]
84.3±5.7 [81.5, 87.1]
91±1 [90.4, 91.6]
9.9±13.7 -59.3** [5.4, 14.4]
84.3±8.1 [ 81.7, 87.0]
86.9±5.6 [85.1, 88.7]
80
9
60
90
32±15.6 27.2±0. [-108.2, 1 [26.3, 172.2] 28.1]
31.3±14. 0 [-94.5, 157.1]
11.5±4.4 -63.3% [-28.0, 51.0]
85±1.4 [72.4, 97.6]
89±4.2 [51.3, 126.7]
33.7±11. 26.8±0. 4 [5.4, 69 62.0] [25.1, 28.5]
47.5±29. 8 [ -26.5 to 121.5]
10.7±3.2 [2.8, 77.5%* 18.6] *
76.7±14.5 % [40.7, 112.7]]
89.3±3.1 [81.6, 97].
TE D
89±1 [ 88.4, 89.6] NR
22
36
26
AC C
Maxillomandib ular Expansion Bonetti et al. 1 2009 Guilleminault 2 and Li 2004
BMI
33.2 ±39.5 [13.9, 52.6] 11.2±4.6 [7.7, 14.7] 25.6
EP
Palmisano et al. 1 1996 Total* 36
Age
RI PT
Study Authors Year Maxillary Expansion Vinha et al. 2015
-80.0%
-88.8%
NR
S1010-5182(16)00043-3
DOI:
10.1016/j.jcms.2016.02.001
Reference:
YJCMS 2301
To appear in:
Journal of Cranio-Maxillo-Facial Surgery
Received Date: 2 November 2015 Revised Date:
15 January 2016
Accepted Date: 1 February 2016
Please cite this article as: Abdullatif J, Certal V, Zaghi S, Song SA, Chang ET, Gillespie MB, Camacho M, Maxillary Expansion and Maxillomandibular Expansion for Adult OSA: A Systematic Review and Meta-Analysis, Journal of Cranio-Maxillofacial Surgery (2016), doi: 10.1016/j.jcms.2016.02.001. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Jose Abdullatif, MD, Victor Certal, MD, PhD, Soroush Zaghi, MD, Sungjin A. Song MD, Edward T. Chang MD, MS, M. Boyd Gillespie MD, MSc, Macario Camacho, MD
SC
Jose Abdullatif, MD Department of Otorhinolaryngology Hospital Bernardino Rivadavia Buenos Aires, Argentina
RI PT
Maxillary Expansion and Maxillomandibular Expansion for Adult OSA: A Systematic Review and Meta-Analysis
M AN U
Victor Certal, MD, PhD a Department of Otorhinolaryngology/Sleep Medicine Centre – Hospital CUF Porto, Portugal b
CINTESIS – Centre for Research in Health Technologies and Information Systems University of Porto Porto, Portugal
TE D
Soroush Zaghi, MD Department of Otolaryngology-Head and Neck Surgery Division of Sleep Surgery Stanford Hospital and Clinics Stanford, CA, USA 95304
EP
Sungjin A. Song, MD Otolaryngology-Head and Neck Surgery Tripler Army Medical Center, Honolulu, HI, USA
AC C
Edward T. Chang, MD, MS Otolaryngology-Head and Neck Surgery Tripler Army Medical Center, Honolulu, HI, USA M. Boyd Gillespie, MD, MSc Department of Otolaryngology - Head and Neck Surgery Medical University of South Carolina, Charleston, SC, USA *Macario Camacho, MD *corresponding author and author to whom correspondence, reprint requests, and proofs will be sent a Otolaryngology-Head and Neck Surgery Division of Sleep Surgery and Medicine Tripler Army Medical Center 1 Jarrett White Road
ACCEPTED MANUSCRIPT
Honolulu, HI, USA 96859 Phone: 808-433-4883 Fax: 808-433-9033 Email: [email protected]
AC C
EP
TE D
M AN U
SC
Department of Psychiatry and Behavioral Sciences Sleep Medicine Division Stanford Hospital and Clinics Stanford, CA, USA 95304
RI PT
b
ACCEPTED MANUSCRIPT
Abstract Objective
RI PT
This study sought to systematically review the international literature for articles
evaluating maxillary expansion and maxillomandibular expansion as treatments for
Data Sources
M AN U
Nine databases (including MEDLINE/PubMed).
SC
obstructive sleep apnea (OSA) in adults and to perform a meta-analysis.
Review Methods
Searches were performed through January 8, 2016. The PRISMA statement was
Results
TE D
followed.
EP
Eight adult studies (39 patients) reported polysomnography and/or sleepiness outcomes. Six studies reported outcomes for maxillary expansion (36 patients), and the apnea-
AC C
hypopnea index (AHI) decreased from a mean (M) ± standard deviation (SD) of 24.3±27.5 [95% CI 15.3, 33.3] to 9.9±13.7 [95% CI 5.4, 14.4] events/hr (relative reduction: 59.3%). Maxillary expansion improved lowest oxygen saturation (LSAT) from a M±SD of 84.3±8.1% [95% CI 81.7, 87.0] to 86.9±5.6% [95% CI 85.1, 88.7]. Maxillomandibular expansion was reported in two studies (3 patients) and AHI decreased from a M±SD of 47.53±29.81 [95% CI -26.5 to 121.5] to 10.7±3.2 [95% CI 2.8, 18.6] events/hr (relative reduction: 77.5%). Maxillomandibular expansion improved LSAT
ACCEPTED MANUSCRIPT
from a M±SD of 76.7±14.5% [95% CI 40.7, 112.7] to 89.3±3.1 [95% CI 81.6, 97]. Conclusion
RI PT
The current literature demonstrates that maxillary expansion can improve and
maxillomandibular expansion can possibly improve AHI and LSAT in adults; however, given the paucity of studies, these remain open for additional research efforts.
SC
Key Words
Systematic Review; Meta-Analysis
TE D
Introduction
M AN U
Maxillary Expansion, Maxillomandibular Expansion, Sleep Apnea Syndromes;
Obstructive sleep apnea (OSA) is a common disorder in adults treated with either medical management (myofunctional therapy (Camacho et al 2015a), positional therapy,
EP
nasopharyngeal airway devices (Kumar et al 2014), continuous positive airway pressure devices, etc.) or with sleep surgeries. Sleep surgery for OSA initially consisted of
AC C
tracheostomies (Camacho et al 2014), however, over the years several additional surgeries have been performed such as tonsillectomy (Senchak et al 2015), uvulopalatopharyngoplasty, hypoglossal nerve stimulation (Certal et al 2015), maxillomandibular advancement (Camacho et al 2015b), etc.). Maxillary expansion has been demonstrated to improve OSA in children (Guilleminault et al 2008), however the effect of maxillary expansion in adults has not been studied as extensively.
ACCEPTED MANUSCRIPT
Transverse maxillary deficiency is a maxillofacial deformity that is characterized as a discrepant width of the maxilla relative to the mandible often due to asymmetric growth of the maxilla or mandible. Variations in the development and the size of the
RI PT
maxilla can lead to increased nasal airflow resistance and displacement of the tongue posteriorly (Vinha et al 2015). Because the floor of the nose is higher, patients with
transverse maxillary deficiency have a greater propensity to developing nasal airway
SC
obstruction, especially in the setting of a deviated nasal septum or large inferior
turbinates (Camacho et al 2015c). Transverse maxillary deficiency may be in fact be an
M AN U
under recognized finding among adult patients with nasal obstruction or OSA. For such patients, maxillary expansion increases the size of the nasal cavity thereby decreasing airway resistance (10% improvement in the size of the nasal cavity corresponds to a 21% improvement in airflow (Powell et al 2001)). By widening the maxilla in the transverse
TE D
dimension, it has been hypothesized that the increased oral cavity space allows for a better position of the tongue and a greater posterior pharyngeal airway space, with a subsequent reduction in airway obstruction (Vinha et al 2015). In children, the maxilla
EP
can be expanded daily between 0.4 to 0.8 mm without surgery by placing an intraoral appliance that is anchored by the teeth (Vinha et al 2015). In adults, the midpalatal
AC C
sutures of the maxilla are fused, therefore, in most cases surgery is used to assist maxillary expansion. Articles have also been published describing the additional expansion of the mandible in the transverse dimension (Guilleminault and Li 2004, Bonetti et al 2009). Given that there has been a limited amount of data with regard to the use of maxillary expansion and maxillomandibular expansion for OSA in adults, our objective was to systematically review the international literature for articles on these two
ACCEPTED MANUSCRIPT
treatments in adult OSA patients and to perform a meta-analysis on the available data.
RI PT
Materials and Methods
Three authors (J.A., E.T.C., and M.C.) independently searched the international
literature without regard to language from inception of each database through January 8,
SC
2016. Databases searched included The Cochrane Library, PubMed/MEDLINE, Scopus,
M AN U
Embase, Google Scholar, Web of Science, Book Citation Index – Science, Cumulative Index to Nursing and Allied Health (CINAHL) and Conference Proceedings Citation Index – Science from the inception of each database. An example of a search strategy for PubMed/MEDLINE is: ((((“sleep”) OR (“snoring”)) AND ((("Biobloc") OR (“Palatal Expansion Technique"[Mesh]) OR (“Maxilla”) OR ("Maxillary”) OR ("Palatal”) OR
TE D
("Palate”) OR (“Orthodontic”)) AND ((“Distraction”) OR (“Widening”) OR (“Expansion”)))) OR ((“Sleep”) AND (“Enuresis”) AND ((“Orthodontic”) OR
EP
(“Distraction”) OR (“Widening”) OR (“Expansion”)))). The Preferred Reporting Items for Systematic Reviews and Meta-Analysis
AC C
(PRISMA) statement was adhered to as much as possible in the performance of this review (Moher et al 2009). Because this is a systematic review of publically available articles, this study is exempted from review by any of the authors’ Institutional Review Boards.
Study selection
ACCEPTED MANUSCRIPT
Inclusion criteria for this review: (1) Patients: adults (≥18 years (y)) with OSA, (2) Intervention: isolated maxillary expansion or maxillomandibular expansion, (3) comparison: quantitative data pre- and post-maxillary expansion or maxillomandibular
RI PT
expansion, (4) Outcome: general outcomes, polysomnography data and sleepiness; and (5) Study design: all designs, published and unpublished studies, and all languages.
Exclusion criteria: (1) studies in which additional surgeries were also performed and
SC
there was no sub-stratification for maxillary expansion or maxillomandibular expansion; (2) studies with qualitative outcomes only; (3) patients with central sleep apnea; and (4)
weight (this can confound outcomes).
M AN U
studies in which individual patient data is reported and patients lost ≥10% of their body
TE D
Quality assessment of included studies
To evaluate the quality of studies, the National Institute for Health and Clinical Excellence (NICE) quality assessment tool (National Institute for Health and Clinical
EP
Excellence 2009) was utilized. This tool was selected because it permits for assessment
AC C
between studies, despite the studies being categorized as having the same level of evidence.
Statistics For this study, the null hypothesis was that there is no difference in polysomnography or sleepiness outcomes pre- and post-maxillary expansion or
ACCEPTED MANUSCRIPT
maxillomandibular expansion. For overall analyses, the IBM Statistical Package for Social Sciences software (SPSS) version 20.0 (Aramonk, New York) was utilized. When evaluating two-tailed, paired t-tests, a p-value <0.05 was considered statistically
RI PT
significant. For the meta-analysis, the Cochrane Collaboration’s Review Manager
Software (REVMAN) version 5.3 was used. The calculations for the mean differences (MD), standardized mean differences (SMD) and 95% confidence intervals [95% CI]
SC
were performed with REVMAN. For interpreting the magnitudes of effect for
standardized mean differences, Cohen’s guidelines (Cohen 1988) were followed and the
M AN U
assigned effects were: small = 0.2, medium = 0.5, and large = 0.8.
Heterogeneity was evaluated between studies by using REVMAN to calculate the Cochran Q statistic (Q statistic), and as recommended, a p-value ≤0.10 was used as the cutoff for statistically significant heterogeneity. (Lau et al 1997) REVMAN was utilized
TE D
to calculate the Inconsistency (I2 statistic), and levels of inconsistency guidelines were used to assign levels: low inconsistency = 25%, moderate inconsistency = 50% and high inconsistency = 75%.( Higgins et al 2003) In evaluating the heterogeneity and
EP
inconsistency, if they were present, then REVMAN’s sensitivity analysis function was
AC C
used by removing one study at a time until heterogeneity and/or inconsistency were not present, therefore, identifying which study or studies were responsible. For evaluation of polysomnography data, when the AHI and respiratory disturbance index (RDI) were reported within a single study, then the AHI was used for combining data and metaanalysis. If however, a study reported only the RDI then the plan was to contact the authors at least twice in order to obtain the AHI. The plan for a funnel plot assessment for bias if at least ten studies were identified for any given variable (as recommended by the
ACCEPTED MANUSCRIPT
Cochrane Collaboration). If any additional data were needed for meta-analysis, then the
RI PT
plan was also to contact the corresponding author at least twice to try to obtain the data.
Results
The systematic review searches provided a total of 207 potentially relevant
SC
studies, of which 98 were downloaded. Eight adult studies (39 patients) reported
M AN U
polysomnography and/or sleepiness outcomes. The case series studies met between four to seven out of the eight possible NICE quality assessment criteria, corresponding to
Maxillary Expansion
TE D
moderate to high quality studies.
Six studies (Palmisano et al 1996, Cistulli et al 1998, Belfor et al 2010, Bach et al
EP
2013, Singh et al 2013, Vinha et al 2015) reported outcomes for maxillary expansion (36 patients) and AHI decreased from a mean (M) ± standard deviation (SD) of 24.3±27.5
AC C
[95% CI 15.3, 33.3] to 9.9±13.7 [95% CI 5.4, 14.4] events/hr (relative reduction: 59.3%). A sub-analysis using random effects modelling demonstrated a mean difference of -9.71 [95% CI -15.41, -4.01] events/hr, overall effect Z score = 3.34 (p = 0.0008), Q statistic p = 0.10 (statistically significant heterogeneity), I2 = 57% (moderate inconsistency). After the removal of the study by Bach et al., there was no heterogeneity (Q statistic = 0.55) and there was no inconsistency (I2 = 0%). The standardized mean difference (SMD) for maxillary expansion demonstrated a large magnitude of effect for AHI (-1.40 [95% CI -
ACCEPTED MANUSCRIPT
2.67, -0.13], overall all effect Z score = 2.17 (p = 0.03), Q statistic p = 0.01 (statistically significant heterogeneity) and the I2 = 78% (high inconsistency). Removal of the study by Cistulli et al. (Cistulli et al 1998) resulted in no heterogeneity (p = 0.52) and no
RI PT
inconsistency (I2 = 0%).
Maxillary expansion improved LSAT from a M±SD of 84.3±8.1% [95% CI 81.7,
SC
87.0] to 86.9±5.6% [95% CI 85.1, 88.7]. The mean LSAT difference was 2.03 [95% CI 1.16, 2.89], overall effect Z score 4.59 (p <0.00001), Q statistic 0.72 (no heterogeneity)
M AN U
and I2 = 0% (no inconsistency). The LSAT SMD was 1.09 [95% CI -0.41, 2.59] (large magnitude of effect), overall effect Z score = 1.42 (p = 0.15), Q statistic p = 0.02 (statistically significant heterogeneity), I2 = 81% (high inconsistency). Sleepiness was reported in the study by Vinha et al. (Vinha et al 2015) with an
TE D
Epworth sleepiness scale (Johns et al 1991) (ESS) pre and post-maxillary expansion
EP
M±SD value of 12.5±5.3 and 7.2±3.5, respectively.
Maxillomandibular Expansion
AC C
Maxillomandibular expansion was reported in two studies (Guilleminault and Li
2004, Bonetti et al 2009) (3 patients) and AHI decreased from a M±SD of 47.53±29.81 [95% CI -26.5 to 121.5] to 10.7±3.2 [95% CI 2.8, 18.6] events/hr (relative reduction: 77.5%). Maxillomandibular expansion improved LSAT from a M±SD of 76.7±14.5% [95% CI 40.7, 112.7] to 89.3±3.1 [95% CI 81.6, 97]. A sub-analysis with random effects modelling could not be performed given there was only one patient in the study by
ACCEPTED MANUSCRIPT
Bonetti et al (Bonetti et al 2009). Sleepiness was reported for 2 patients in the study by Guilleminault and Li (Guilleminault and Li 2004), with improvement from an pre and
RI PT
post-maxillomandibular expansion M±SD of 10.5±0.71 and 7.5±0.71, respectively.
Discussion
SC
There are four main findings from this systematic review and meta-analysis. First,
M AN U
maxillary expansion can improve AHI, and maxillomandibular expansion potentially can improve AHI. There were thirty-six patients in the maxillary expansion studies and three patients in the maxillomandibular expansion studies. Overall, maxillary expansion reduced AHI by 59.3% and maxillomandibular expansion reduced AHI by 77.5%. Six studies have reported outcomes for maxillary expansion and AHI, while only two studies
TE D
have been published for maxillomandibular expansion and AHI. It has been hypothesized that maxillary expansion improves OSA variables by widening the floor of the nose and improving the position of the tongue. We additionally hypothesize that by widening the
EP
maxilla, there is tension on the muscles that attach to the palate such as the palatoglossus
AC C
and palatopharyngeus, with associated anterior displacement of these muscles, which can therefore reduce the collapsibility of the upper airway during sleep. It is logical that the addition of transverse expansion of the mandible would improve AHI even more; however, additional studies are needed to confirm this hypothesis as there have only been three patients in the literature. Second, LSAT has improved following both maxillary expansion and maxillomandibular expansion. However, there are fewer studies reporting outcomes for
ACCEPTED MANUSCRIPT
LSAT than AHI. There were only two studies reporting outcomes in twenty-six patients for LSAT outcomes in maxillary expansion patients and only two studies reporting LSAT outcomes in maxillomandibular expansion (three patients). The improvement was
RI PT
actually not that significant clinically for maxillary expansion, with a 2.6 point
improvement; however, there was a more clinically significant improvement (12.6 points)
given that there were only three patients.
SC
for maxillomandibular expansion. We caution against drawing conclusions for LSAT
M AN U
Third, sleepiness seems to decrease for both patients who have undergone either maxillary expansion or maxillomandibular expansion. For maxillary expansion, Vinha et al. (Vinha et al 2015) reported an improvement in ESS from a pre and post-maxillary expansion M±SD value of 12.5±5.3 and 7.2±3.5, respectively. For maxillomandibular expansion, sleepiness was reported for 2 patients in one study (Guilleminault and Li)
TE D
(Guilleminault and Li 2004), with improvement from a pre and post-maxillomandibular expansion M±SD of 10.5±0.71 and 7.5±0.71, respectively. Therefore, in the patients who were treated with either maxillary expansion or maxillomandibular expansion the patients
EP
were hypersomnolent and afterwards they were not.
AC C
Fourth, additional research is needed for both maxillary expansion and especially
for maxillomandibular expansion. With thirty-six patients in the literature for maxillary expansion and only three patients undergoing maxillomandibular expansion, the need for more publications is recommended. The relative reduction in the MME was higher (77.5%) compared with the AHI relative reduction of the ME (59.3%); however, the number of cases that met the criteria for inclusion in the MME review was extremely low and no conclusions can be made. Because of this, more studies evaluating the effect of
ACCEPTED MANUSCRIPT
maxillomandibular expansion in OSA are needed. Questions that have not been addressed which could be studied include the effect of combining ME or MME with maxillomandibular advancements in selected patients. Additionally, in order to further
RI PT
evaluate the outcomes for OSA patients, the publication of individual patient data with variables such as age, BMI, prior surgeries, AHI, LSAT, mean time spent under 90%, oxygen desaturation index, hypopnea scoring criteria, and sleepiness outcomes
SC
(specifically the Epworth sleepiness scale (Johns et al 1991)) would allow for individual patient data meta-analyses. The reporting of complications with associated percentage
M AN U
rates for each complication would also facilitate determination of complication rates (such as the rates of wound infections, the need for revisions and bleeding rates).
TE D
Limitations
There are limitations to this study, first the number of studies reporting outcomes as treatment for OSA are limited (six maxillary expansion and two maxillomandibular
EP
expansion studies); however, the data for the published studies have consistently
AC C
demonstrated an improvement in OSA variables. Second, like any systematic review, it is possible that we missed identifying articles; to minimize this possibility, we had three authors independently search the international literature without regard to language. Third, given the limited number of studies that are currently published, we would like to encourage surgeons to publish their outcomes with pre and post-treatment means and standard deviations for variables such as apnea-hypopnea index, sleepiness and oxygen saturation data. Lastly, because there can be a publication bias against negative studies, it
ACCEPTED MANUSCRIPT
is possible that studies finding minimal or no differences in polysomnography variables were either not submitted for publication or were selected against; because there were not at least 10 studies, as recommended by the Cochrane Collaboration, we did not perform
RI PT
an analysis for publication bias as the funnel plot analysis would not be as accurate.
SC
Conclusion
Current literature demonstrates that maxillary expansion can improve and
M AN U
maxillomandibular expansion possibly can improve AHI and LSAT in adults. Additional
AC C
EP
TE D
research is needed, especially for maxillomandibular expansion.
ACCEPTED MANUSCRIPT
References "Methods for development of NICE public health guidance." London: National
RI PT
Institute for Health and Clinical Excellence 2009. Bach N, Tuomilehto H, Gauthier C, Papadakis A, Remise C, Lavigne F, Lavigne GJ and Huynh N. "The effect of surgically assisted rapid maxillary expansion on
M AN U
Rehabil 40(11): 818-825, 2013.
SC
sleep architecture: an exploratory risk study in healthy young adults." J Oral
Belfor TR. "Epigenetic orthodontics: facial and airway development." N Y State Dent J 76(6): 18-21, 2010.
Bonetti GA, Piccin O, Lancellotti L, Bianchi A and Marchetti C. "A case report
TE D
on the efficacy of transverse expansion in severe obstructive sleep apnea syndrome." Sleep Breath 13(1): 93-96, 2009. Camacho M, Certal V, Brietzke SE, Holty JE, Guilleminault C and Capasso R.
EP
"Tracheostomy as treatment for adult obstructive sleep apnea: a systematic review
AC C
and meta-analysis." Laryngoscope 124(3): 803-811, 2014. Camacho M, Certal V, Abdullatif J; Zaghi S; Ruoff C; Capasso R; Kushida C. Myofunctional Therapy to Treat Obstructive Sleep Apnea: A Systematic Review and Meta-analysis. Sleep; 38:669-675, 2015a. Camacho M, Liu SY, Certal V, Capasso R, Powell NB and Riley RW. "Large Maxillomandibular Advancements for Obstructive Sleep Apnea: An Operative
ACCEPTED MANUSCRIPT
Technique Evolved Over 30 Years." Journal of Cranio-Maxillofacial Surgery, 2015b.
RI PT
Camacho M, Zaghi S, Certal V, Abdullatif J, Means C, Acevedo J, Liu S, Brietzke SE, Kushida CA and Capasso R. "Inferior Turbinate classification
system, grades 1 to 4: Development and validation study." Laryngoscope 125(2):
SC
296-302, 2015c.
Certal VF, Zaghi S, Riaz M, Vieira A S, Pinheiro CT, Kushida C, Capasso R and
M AN U
Camacho M. "Hypoglossal nerve stimulation in the treatment of obstructive sleep apnea: A systematic review and meta-analysis." Laryngoscope 125(5): 12541264, 2015.
Cistulli PA, Palmisano RG and Poole MD. "Treatment of obstructive sleep apnea
TE D
syndrome by rapid maxillary expansion." Sleep 21(8): 831-835, 1998. Cohen J . Statistical Power Analysis for the Behavioral Sciences. Hillsdale, NJ,
EP
USA, Erlbaum, 1988.
Guilleminault C and Li K. "Maxillomandibular expansion for the treatment of
AC C
sleep-disordered breathing: preliminary result." Laryngoscope 114(5): 893-896, 2004.
Guilleminault C, Quo S, Huynh NT and Li K. "Orthodontic expansion treatment and adenotonsillectomy in the treatment of obstructive sleep apnea in prepubertal children." Sleep 31(7): 953-957, 2008.
ACCEPTED MANUSCRIPT
Higgins JP, Thompson SG, Deeks JJ and Altman DG . "Measuring inconsistency in meta-analyses." BMJ 327(7414): 557-560, 2003.
RI PT
Johns, MW . "A new method for measuring daytime sleepiness: the Epworth sleepiness scale." Sleep 14(6): 540-545, 1991.
Kumar AR, Guilleminault C, Certal V, Li D, Capasso R, Camacho M.
SC
Nasopharyngeal airway stenting devices for obstructive sleep apnoea: A
systematic review and meta-analysis. The Journal of laryngology and otology :1-
M AN U
9, 2014.
Lau J, Ioannidis JP and Schmid CH. "Quantitative synthesis in systematic reviews." Ann Intern Med 127(9): 820-826, 1997.
TE D
Moher D, Liberati A, Tetzlaff J and Altman DG. "Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement." PLoS Med 6(7): e1000097, 2009.
EP
Palmisano RG, Wilcox I, Sullivan CE and Cistulli PA. "Treatment of snoring and obstructive sleep apnoea by rapid maxillary expansion." Aust N Z J Med 26(3):
AC C
428-429, 1996.
Powell NB, Zonato AI, Weaver EM, Li K, Troell R, Riley RW and Guilleminault C. "Radiofrequency treatment of turbinate hypertrophy in subjects using continuous positive airway pressure: a randomized, double-blind, placebocontrolled clinical pilot trial." Laryngoscope 111(10): 1783-1790, 2001.
ACCEPTED MANUSCRIPT
Senchak AJ, McKinlay AJ, Acevedo J, Swain B, Tiu MC, Chen BS, Robitschek J, Ruhl DS, Williams LL, Camacho M, Frey WC and O'Connor PD. "The effect of tonsillectomy alone in adult obstructive sleep apnea." Otolaryngol Head Neck
RI PT
Surg 152(5): 969-973, 2015.
Singh GD and Callister JD. "Effect of a maxillary appliance in an adult with
SC
obstructive sleep apnea: a case report." Cranio 31(3): 171-175, 2013.
Vinha PP, Eckeli AL, Faria AC, Xavier SP and de Mello-Filho FV. "Effects of
M AN U
surgically assisted rapid maxillary expansion on obstructive sleep apnea and
AC C
EP
TE D
daytime sleepiness." Sleep Breath 2015.
Additional studies identified from reference lists N=5
Articles excluded N = 90 Pediatric studies = 77 No quantitative data = 6 Other treatment = 7
M AN U
Potentially relevant articles retrieved for detailed evaluation N = 93
Excluded articles: N = 113 Not OSA = 105 Review articles = 8
SC
Articles screened for relevance (after removal of duplicates) N = 207
RI PT
ACCEPTED MANUSCRIPT
Included Studies N=8
AC C
EP
TE D
Figure 1: Flow chart for study selection. Abbreviations: N = number of studies, and pts = patients.
SC
RI PT
ACCEPTED MANUSCRIPT
Figure 2: Mean difference in AHI pre and post-maxillary expansion. 95% confidence
AC C
EP
TE D
M AN U
intervals are provided.
ACCEPTED MANUSCRIPT
4
Canada
4
USA
5
NR Australia Australia
Italy
Quality assessment of included studies* 2 3 4 5
6
7
8
AHI, ESS, LSAT, RDI AHI, MSAT, SAT<90% AHI, ODI, snoring AHI, RDI
N
Y
Y
Y
Y
Y
Y
Y
N
Y
Y
N
Y
N
Y
N
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
NA
4
AHI, LSAT
N
Y
Y
N
Y
N
Y
N
5
AHI
NA
NA
NA
NA
NA
NA
NA
NA
5
AHI, LSAT,
NA
NA
NA
NA
NA
NA
NA
NA
TE D
Brazil
EP
Maxillary Expansion Vinha et al. 2015, PCS Bach et al. 2013 PCS Singh et al. 2013 RCR Belfor et al. 2010 RCR Cistulli et al. 1998 PCS Palmisano et al. 1996 RCR Maxillomandibular Expansion Bonetti et al.
Study site
Study’s General Characteristics and number of included patients Evidence Outcomes 1 level analysed
5
AC C
Year, Study, Design
M AN U
SC
RI PT
Table 1. General characteristics and quality criteria of included studies. *Quality Assessment of cases series studies checklist from National Institute for Health and Clinical Excellence (NICE): 1) Was the case series collected in more than one centre, i.e. multicentre study? 2) Is the hypothesis/aim/objective of the study clearly described? 3) Are the inclusion and exclusion criteria (case definition) clearly reported? 4) Is there a clear definition of the outcomes reported? 5) Were data collected prospectively? 6) Is there an explicit statement that patients were recruited consecutively? 7) Are the main findings of the study clearly described? 8) Are outcomes stratified? (e.g., by abnormal results, disease stage, patient characteristics)? Abbreviations: AHI = Apnea-Hypopnea index; ESS = Epworth Sleepiness Scale; LSAT = lowest oxygen saturation; MSAT = mean oxygen saturation; N.A. = Not applicable; N.R. = Not reported; RCR = retrospective case report; RCS = retrospective case series; RDI = respiratory disturbance index; SAT<90% = time spent under oxygen saturation of 90%.
ACCEPTED MANUSCRIPT
M AN U TE D
Y
Y
RI PT
N
SC
4
EP
USA
SAT<90% AHI, ESS, LSAT
AC C
2009 RCR Guilleminault and Li 2004 PCS
Y
Y
Y
Y
Y
ACCEPTED MANUSCRIPT
Table 2: Demographic and polysomnographic data pre and post-rapid maxillary expansion or maxillomandibular expansion. Abbreviations: [ ] denotes lower and upper 95% confidence intervals; AHI = apnea-hypopnea index; low O2 = lowest oxygen saturation; mo = months; N = number; NR = not reported. *Total is based on means and standard deviations. **AHI change is based on the relative reduction in AHI N
16
40.2±10. 29.8±4. 2 4
Bach et al. 2013
7
NR
NR
Singh et al. 2013 Belfor et al. 2010 Cistulli et al. 1998
1
64
26
1
NR
NR
10
27±2
29.8±2. 6
Total*
3
PostOp AHI
AHI change
Pre-Op low O2
Post-Op low O2
14.5±19. -56.3 4 [5.0, 24.0]
-50.9%
NR
NR
-82.4%
NR
NR
28
5
-82.1
NR
NR
19±4 [16.5, 21.5] 20
7±4 [4.5, 9.5] 4
-63.2%
35.7±11. 29.9±3. 7 [31.9, 9 39.5] [28.6, 31.2]
24.3±27. 5 [15.3, 33.3]
37
M AN U
SC
Pre-Op AHI
5.7±5.4 [1.7, 9.7 ] 4.5
81.3±9.2 [76.8, 85.8]
84.3±5.7 [81.5, 87.1]
91±1 [90.4, 91.6]
9.9±13.7 -59.3** [5.4, 14.4]
84.3±8.1 [ 81.7, 87.0]
86.9±5.6 [85.1, 88.7]
80
9
60
90
32±15.6 27.2±0. [-108.2, 1 [26.3, 172.2] 28.1]
31.3±14. 0 [-94.5, 157.1]
11.5±4.4 -63.3% [-28.0, 51.0]
85±1.4 [72.4, 97.6]
89±4.2 [51.3, 126.7]
33.7±11. 26.8±0. 4 [5.4, 69 62.0] [25.1, 28.5]
47.5±29. 8 [ -26.5 to 121.5]
10.7±3.2 [2.8, 77.5%* 18.6] *
76.7±14.5 % [40.7, 112.7]]
89.3±3.1 [81.6, 97].
TE D
89±1 [ 88.4, 89.6] NR
22
36
26
AC C
Maxillomandib ular Expansion Bonetti et al. 1 2009 Guilleminault 2 and Li 2004
BMI
33.2 ±39.5 [13.9, 52.6] 11.2±4.6 [7.7, 14.7] 25.6
EP
Palmisano et al. 1 1996 Total* 36
Age
RI PT
Study Authors Year Maxillary Expansion Vinha et al. 2015
-80.0%
-88.8%
NR