Tongue Reduction for Pediatric Obstructive Sleep Apnea: A Systematic Review

Adrian Williamson, IV; Rohit Nallani; Jason R Brown

doi:10.1002/oto2.70215

. 2026 Mar 3;10(1):e70215. doi: 10.1002/oto2.70215

Tongue Reduction for Pediatric Obstructive Sleep Apnea: A Systematic Review

Adrian Williamson IV ^1,^2,^✉, Rohit Nallani ^3,⁴, Jason R Brown ^3,⁵

PMCID: PMC12955833 PMID: 41783520

Abstract

Objective

Obstructive sleep apnea (OSA) is a prevalent pediatric disorder that, if untreated, can result in substantial developmental and health consequences. Tongue reduction procedures, such as midline posterior glossectomy (MPG), are increasingly used to address tongue base obstruction. This systematic review evaluates the effectiveness and safety of tongue reduction procedures for pediatric OSA.

Data Sources

Embase, PubMed, and Web of Science databases were queried for studies from inception to September 20, 2023.

Review Methods

Studies reporting on outcomes of pediatric patients (≤21 years) undergoing tongue reduction surgery for OSA were included.

Results

From 477 abstracts, 9 studies met the inclusion criteria, representing 146 pediatric patients. Surgical techniques included MPG, transoral robotic‐assisted tongue base resection, and keyhole partial glossectomy. Primary outcomes assessed were apnea‐hypopnea index (AHI), minimum oxygen saturation, and adverse events. All studies reported a reduction in AHI and increased minimum oxygen saturation following tongue reduction despite the method. Adverse events appear to be rare but included postoperative bleeding, need for reintubation, and wound dehiscence.

Conclusion

Tongue reduction procedures appear to be safe and effective for managing pediatric OSA, improving key clinical outcomes. However, long‐term results, comparative studies of surgical techniques, and refined patient selection criteria are needed. Future research should also identify risk factors for persistent OSA following tongue base reduction. These findings suggest that tongue reduction is a valuable addition to the treatment arsenal for pediatric OSA.

Keywords: obstructive sleep apnea, OSA, macroglossia, midline posterior glossectomy, tongue reduction, pediatric

Obstructive sleep apnea (OSA) is caused by intermittent upper airway obstruction leading to apneas and/or hypopneas during sleep. Approximately 1% to 10% of the pediatric population is impacted by OSA with some studies suggesting a prevalence of up to 25%. ¹ , ² , ³ , ⁴ , ⁵ Children with OSA can experience a range of symptoms and complications including disturbed sleep, daytime somnolence, nocturnal enuresis, cardiovascular complications, metabolic dysfunction, neurocognitive dysfunction resulting in academic and behavioral issues, and significantly decreased disease‐specific quality of life. ⁶ , ⁷ , ⁸ , ⁹ , ¹⁰ , ¹¹ , ¹² , ¹³ , ¹⁴

The treatment of pediatric OSA is guided by the underlying anatomical and functional contributors to airway obstruction. The primary treatment for pediatric OSA, recommended by the American Academy of Otolaryngology–Head and Neck Surgery and the American Academy of Pediatrics, is adenotonsillectomy (AT) as this is the most common site of upper airway obstruction. ⁵ , ¹⁵ However, this standardized treatment approach may not be appropriate for all patients. Additionally, despite AT, 20% to 75% of children may have persistent OSA. ¹⁶ , ¹⁷ , ¹⁸ , ¹⁹ Tongue reduction surgery alone for patients with primary macroglossia, such as those with Beckwith‐Wiedemann Syndrome, or as part of multilevel airway surgery, has been described as an initial treatment for some children with OSA. ²⁰ , ²¹ , ²² , ²³ , ²⁴

For patients with ongoing symptoms of OSA after AT, the American Academy of Pediatrics recommends obtaining a polysomnogram (PSG). ⁵ PSG is also indicated in patients who are at risk for severe OSA. ²⁵ Additional management of patients with OSA may include observation, medications such as fluticasone or montelukast, weight loss, positive airway pressure (PAP) therapy, or additional surgical intervention beyond AT guided by the site of airway obstruction. ²⁶ , ²⁷ Surgical intervention for persistent OSA after AT is guided by the level and severity of specific anatomic obstruction which can be multileveled.

Drug Induced Sleep Endoscopy (DISE), Cinematic Magnetic Resonance Imaging (CINE MRI), awake flexible laryngoscopy and radiographic studies have been used to ascertain the location of obstruction in patients with persistent OSA despite AT or in patients that are at high risk for OSA despite AT. ²⁶ , ²⁸ The most common site of obstruction identified in children with persistent OSA after AT is the retrolingual airway. ²⁹ , ³⁰ , ³¹ Tongue base obstruction can be secondary to hypotonia, lingual tonsil hypertrophy, macroglossia, glossoptosis, or a combination of these factors.

For patients with macroglossia or glossoptosis, tongue reduction surgeries have been employed to alleviate this obstruction. Additional methods to address tongue base obstruction include hypoglossal nerve stimulation and tongue base suspension suture. ³² , ³³ , ³⁴ Surgical tongue reduction has been described as a primary treatment for pediatric OSA, as a part of multilevel airway surgery, or as a treatment for persistent OSA despite prior AT. Methods of tongue reduction include from intramural tongue ablation to various forms of partial glossectomy include base of tongue wedge resection or midline posterior glossectomy (MPG). The purpose of this review is to investigate the safety and efficacy of surgical tongue reduction in pediatric patients with OSA, either as a primary or secondary treatment after AT, or as a part of multilevel surgical treatment. By synthesizing the current evidence, we seek to highlight the strengths and limitations of the existing data and identify areas for future research to optimize the management of tongue base obstruction in pediatric OSA.

Methods

Study Design

An exemption was provided by the Children's Mercy Hospital Institutional Review Board. Preferred Reporting Items for Systematic Reviews and Meta‐analyses (PRISMA) guidelines were utilized for this study. ³⁵ This systematic review was registered with PROSPERO (Registration ID: CRD420251006809). We performed a systematic search of the Embase, PubMed, and Web of Science databases from inception to September 20th, 2023. Supplemental Tables SA1‐SA3, available online, outline the search strategy with specific criteria used for each database.

Studies examining sleep and surgical outcomes following tongue reduction surgery for pediatric OSA were the focus of the searches. All studies were uploaded to Covidence© software. ³⁶ Two authors (AW and RN) independently reviewed all titles and abstracts for eligibility and inclusion. Any disagreements were reconciled by a third author (JB). Studies were then included for final data extraction following full‐text review by two authors (AW and RN), with disagreements again reconciled by a third author (JB). Supplemental Table SA4, available online summarizes the inter‐rater author agreement and reliability for the screening process. The quality of included studies was evaluated using the modified Downs and Black checklist for assessment of methodological quality. ³⁷ This assessment was completed independently by 2 authors (AW and RN) and disagreements were reconciled by the third author (JB). Consistent with prior publications, studies were categorized based on quality as follows: excellent (26‐28), good (20‐25), fair (15‐19), and poor (≤14). ³⁸ , ³⁹

Inclusion and Exclusion Criteria

Studies were included for review if they focused on pediatric patients with OSA ≤ 21 years old who underwent tongue reduction by surgical excision. Tongue reduction procedures included MPG, tongue base wedge resection using coblation or transoral robotic‐assisted surgery (TORS), and other forms of partial glossectomy. Intramural tongue ablation by radiofrequency ablation or electrocautery was not included unless partial glossectomy was explicitly performed. Additionally, lingual tonsillectomy (LT) was considered distinct from the tongue reduction surgeries that we intended to study. Studies that included only patients undergoing LT without additional resection of the intrinsic or base of tongue did not meet inclusion criteria. Studies were not excluded if other sleep surgeries (ie, tonsillectomy, adenoidectomy, LT, supraglottoplasty, etc.) were performed on patients in the sample in addition to tongue reduction procedures. Exclusion criteria included studies on adult patients (age > 21), tongue reduction surgery or glossectomy performed for non‐OSA indication, and absence of sleep, surgical, or other clinical outcomes data. If data specific to patients who had tongue reduction surgeries could not be extracted, the study was also excluded. Studies were also excluded if they were conference abstracts, nonaccessible abstracts or manuscripts, single‐patient case reports, systematic reviews/meta‐analyses, or commentaries.

Data Extraction

Study authors, publication information, study design, population of study, specific type of tongue reduction surgery assessed, and patient outcomes were extracted from the included studies. We extracted total sample size in addition to the pertinent sample size who underwent tongue reduction with available outcomes data, age of the sample, reported body‐mass‐index (preoperative and postoperative), additional sleep surgeries performed, and if surgeries were directed by DISE or CINE MRI. Outcomes data were sleep‐specific or surgical‐specific. Sleep outcomes information included preoperative and postoperative apnea‐hypopnea index (AHI) or obstructive apnea‐hypopnea index (oAHI) and minimum oxygen concentration with relevant measures of effect size or P‐values. Other measures of surgical success and hospital length of stay were also extracted. Postoperative complications captured included bleeding, re‐intubation, pneumonia, wound dehiscence, readmission for poor oral intake or pain control, or need for additional tongue reduction surgery.

Results

Literature Search and Study Screening

Figure 1 displays the PRISMA diagram for study selection. An initial search was performed of the Embase, PubMed, and Web of Science databases, and 477 studies were identified. Following removal of duplicates, 216 studies remained. These were screened using title and abstract review to assess for relevance, and 34 studies were identified. The full text of these studies was reviewed, and 25 studies were excluded. Thirteen studies were abstracts only or had no complete text available. Data specific to MPG could not be extracted from 6 studies. Another 4 studies included adult patients over age 21. Two other studies did not report any outcomes or safety data. Nine studies were eventually included based on inclusion criteria.

PRISMA flow diagram of study selection. This PRISMA (Preferred Reporting Items for Systematic Reviews and Meta‐Analyses) flow diagram illustrates the progression through the stages of screening, eligibility assessment, and final inclusion.

Study Characteristics and Patients

Studies were published between the years 1997 and 2023. ²⁰ , ²¹ , ²² , ²³ , ⁴⁰ , ⁴¹ , ⁴² , ⁴³ , ⁴⁴ Table 1 includes a summary of the included studies. The majority of studies were retrospective cohort (n = 5), 2 were prospective cohorts, and two were case series. The quality of studies ranged from fair to poor based on the modified Downs Black checklist for assessment of methodological quality (Table 1). Four studies specifically assessed outcomes in syndromic patients with Beckwith‐Wiedemann (BW) syndrome or Down Syndrome, while the other five had a mix of syndromic and non‐syndromic patients. Sample sizes of the included studies ranged from 2 to 67, with a total of 149 patients captured for all studies together. Patient age ranged from less than 3 months to 18.7 years.

Table 1.

Summary of Included Studies

Author, year	Study type	n	Method of tongue reduction	Other airway procedures performed?^a	Included patients that did not undergo prior AT?	Included syndromic or non‐syndromic patients?	Summary of findings	Study quality (modified Downs and Black checklist score)
Lefaivre et al. 1997	Prospective cohort study	6^b	CO₂ laser central tongue reduction	Yes	Yes	Syndromic only (DS)	Aggressive multi‐level soft tissue and skeletal surgery in children with DS and OSA results in significant AHI improvement.	Poor (14)
Maturo and Mair 2006	Case series	2^c	SMILE	No	Not explicitly specified	Syndromic only (DS and BWS)	SMILE operation was safe and effective at reducing obstructive macroglossia and improving objective and subjective sleep concerns.	Poor (7)
Thottam et al. 2015	Prospective cohort study	9	TORS tongue base reduction + LT	No	No	Mixed syndromic (DS and Noonan Syndrome) and non‐syndromic	There were statistically significant improvements in AHI, hypopnea events, and O2 nadir in all patients.	Fair (17)
Montevecchi et al. 2017	Case series	3	TORS tongue base reduction	Yes	Yes	Non‐syndromic only	There was improvement in AHI following TORS tongue base reduction with or without other procedures.	Poor (12)
Propst et al. 2017	Prospective cohort study	13	SMILE + LT	No	No	Syndromic only (DS)	SMILE + LT is most beneficial in normal weight and overweight children with DS with refractory OSA while preoperative or postoperative obesity is associated with worse outcomes.	Fair (18)
Ulualp 2019	Retrospective cohort	10	SMILE + LT	No	No	Mixed syndromic (DS) and non‐syndromic	DISE‐directed SMILE + LT significantly improves oAHI and subjective OSA symptoms.	Fair (17)
Dmowska et al. 2020	Case series	3	Tongue base reduction with coblation + LT	Yes	Yes	Mixed syndromic (DS) and non‐syndromic	For 2 of the 3 patients, DISE‐directed tongue base reduction with adenotonsillectomy resulted in improved AHI.	Poor (14)
Cohen et al. 2020	Retrospective cohort	36^d	Modified W excision	Yes	Yes	Syndromic only (BWS)	Tongue base reduction is a safe and effective option for young children with BWS and OSA.	Fair (18)
Williamson et al. 2023	Retrospective cohort	67	SMILE ± LT	Yes	Yes	Mixed syndromic (DS) and nonsyndromic	DISE‐directed tongue base reduction significantly improves oAHI with few complications and high surgical success.	Fair (17)

Open in a new tab

Abbreviations: AHI, Apnea‐Hypopnea Index; BWS, Beckwith‐Wiedemann Syndrome; DISE, Drug‐induced sleep endoscopy; DS, Down Syndrome; LT, Lingual tonsillectomy; OSA, Obstructive Sleep Apnea; SMILE, Submucosal minimally invasive lingual excision.

^{^a}

Excludes LT as an additional airway procedure. Reference column entitled “Method of tongue reduction” for reference of which studies included LT.

^{^b}

Only reported PSG data for 5 of the 6 patients.

^{^c}

There were two other patients in this case series which underwent tongue reduction for macroglossia without OSA. These two patients were thus not included in our review.

^{^d}

Only reported PSG data for 12 of the 36 patients.

Nearly all patients in the included studies had refractory OSA following AT or other sleep surgery. However, one or more patients in studies by Cohen et al (36 patients), Lefaivre et al (6 patients), Dmowska et al (3 patients), and Montevecchi et al (1 of 3 patients) did not have prior surgery to address OSA before tongue base reduction. ²⁰ , ²¹ , ²² , ²³ Patients in the study by Cohen et al underwent tongue reduction for macroglossia due to BW syndrome, but only 12 of the 36 patients had preoperative and postoperative PSG data available. However, all patients were included in our results as other outcomes data was reported for these patients. Patients underwent procedures in addition to tongue reduction in 5 studies (Table 1). Other procedures included adenoidectomy, tonsillectomy, pharyngoplasty, supraglottoplasty, epiglottoplasty, hyoid suspension, septoplasty, turbinate reduction, and/or mandibular distraction or other skeletal expansion surgery. To identify areas of obstruction, patients were evaluated preoperatively with DISE, CINE MRI, physical exam, and awake flexible laryngoscopy. Five of the nine studies in this review (Lefaivre 1997, Thottam et al 2015, Uluap 2019, Dmowska et al 2020, and Williamson et al 2023) utilized DISE to identify the sites of obstruction prior to surgical intervention. ²¹ , ²² , ⁴¹ , ⁴³ , ⁴⁴ Propst et al used CINE MRI and awake flexible laryngoscopy in sitting and supine position to identify tongue base obstruction prior to surgical tongue reduction. ⁴² Montevecchi et al used a combination of awake flexible endoscopy and MRI to determine the sites of obstruction in their case series prior to tongue base resection with TORS. ²³

Surgical Techniques

Several methods of partial glossectomy have been described to relieve retrolingual obstruction in children with OSA. ⁴⁵ For the studies reported here (Table 1), the most common method was MPG using the submucosal minimally invasive lingual excision (SMILE) technique with or without LT (Maturo and Mair 20006, Propst et al 2017, Ulualp 2019, and Williamson et al 2023). ⁴⁰ , ⁴² , ⁴³ , ⁴⁴ Maturo and Mair demonstrated safety of the SMILE technique using 5 cadaver models along with a case series, ensuring up to 20 cm³ of tongue base could be removed without injury to the lingual artery given its lateral position at the tongue base. ⁴⁰ LT is often performed before MPG to ensure the tongue base is cleared of lingual tonsillar tissue and easily accessible. Thottam et al and Montevecchi et al utilized the Da Vinci™ robot to perform transoral robotic tongue base reduction. ²³ , ⁴¹ Lefaivre et al performed a central tongue reduction using a CO₂ laser and Dmowska et al utilized coblation for tongue base reduction but did not specify if it was the SMILE technique. ²¹ , ²² Cohen et al described partial glossectomy by a Modified W excision with keyhole for patients with OSA and BW syndrome. ²⁰

Clinical Efficacy and Safety

All studies reported clinical success of tongue base reduction procedures. All studies reported preoperative AHI (total or obstructive component), and most studies reported oxygenation nadirs on preoperative and postoperative PSG (Table 2). All studies reported improvements in AHI or oAHI following surgical management of tongue base obstruction. These changes were statistically significant in 5 studies (Thottam et al 2015, Propst et al 2017, Ulualp 2019, Cohen et al 2020, and Williamson et al 2023). ²⁰ , ⁴¹ , ⁴² , ⁴³ , ⁴⁴ In 4 of the studies (Lefaivre et al 1997, Maturo and Mair 2006, Montevecchi et al 2017, and Williamson et al 2023), postoperative AHI was less than 5 in the collective patient sample, reflective of mild OSA. ²¹ , ²³ , ⁴⁰ , ⁴⁴ In another 4 studies (Thottam et al 2015, Propst et al 2017, Dwomska et al 2020, and Cohen et al 2020), postoperative AHI was still 10 or greater despite decrease from baseline. ²⁰ , ²² , ⁴¹ , ⁴² Oxygen saturation nadir improved in 6 of the studies but worsened on average for the 13 patients in the Propst et al. ⁴² Two studies did not report PSG oxygenation information. Of the nine studies included, only 3 studies reported (Lefaivre et al 1997, Montevecchi et al 2017, and Cohen et al 2020) long‐term follow‐up data after surgery (Table 2). ²⁰ , ²¹ , ²³

Table 2.

Summary of PSG Data

			AHI (events/h)			Oxygen saturation nadir (%)
Author, year	Timing of postoperative sleep study	Follow up period (mean unless otherwise noted)	Preoperative (mean)	Postoperative (mean)	Change in AHI (mean)	Preoperative (mean)	Postoperative (mean)
Lefaivre et al. 1997^a,^b	Prior to discharge	31.3 months	30.82	1.41	29.41	65.8	68
Maturo and Mair 2006^a	3‐4 months	Not reported	10.0	0.55	9.45	80.5	89.5
Thottam et al. 2015^a	4 months	Not reported	27.1	10.9	16.2	74.2	83
Montevecchi et al. 2017	3‐6 months	5 months	17.2	4.1	13.1	N/A^c	N/A^c
Propst et al. 2017^a	Not reported	Not reported	44.3^d	34.1^d	10.2^d	74.8	73.7
Ulualp 2019^a	2‐5 months	Not reported	26.3^e	5.3^e	21.0^e	91^e	92.5^e
Dmowska et al. 2020^a	3‐6 months	Not reported	31.5	15.4	16.1	Not reported	Not reported
Cohen et al. 2020^a	Not reported	7 months^d	30.9^d	10.0^d	20.9^d	72^f	83^f
Williamson et al. 2023^a,^c	>2 months	Not reported	5.23^d,^g	2.68^d,^g	2.55^d,^g	88.2^g	89.3^g
Williamson et al. 2023^a,^c	>2 months	Not reported	7.22^d,^h	3.53^d,^h	3.70^d,^h	88.9^h	90.1^h

Open in a new tab

Abbreviation: AHI, Apnea‐Hypopnea Index.

^{^a}

Included syndromic patients.

^{^b}

Only had data available for 5 of 6 patients.

^{^c}

Did not have O₂ nadir data available for all patients.

^{^d}

Reported obstructive AHI.

^{^e}

Median reported.

^{^f}

Only had data available for 10 of 12 patients.

^{^g}

Patients underwent tongue base reduction alone.

^{^h}

Patients underwent tongue base reduction + lingual tonsillectomy.

Overall, tongue reduction procedures were demonstrated to be safe across all studies (Table 3). Hospital length of stay was reported in 6 studies, ranging from less than 24 hours to 19.3 days. Few complications were reported, with an overall rate of 12.1% (18 complications in 149 patients). The most common complication was postoperative bleeding, seen in seven patients (4.7%). Interestingly, all patients with postoperative bleeding had LT performed in addition to MPG.

Table 3.

Summary of Safety Outcomes

Author, year	Mean hospital length of stay (days)	Postoperative bleeding (n)	Intubation postoperatively (n)	Pneumonia or other respiratory infection (n)	Wound dehiscence (n)
Lefaivre et al. 1997^a	19.3	0	0	0	1
Maturo and Mair 2006^a	Not reported	0	0	0	0
Thottam et al. 2015^a	4.3	1	1	1	0
Montevecchi et al. 2017	4.3	0	0	0	0
Propst et al. 2017^a	6.62	2	1	2	0
Ulualp 2019^a	<1	0	0	0	0
Dmowska et al. 2020^a	Not reported	0	0	0	0
Cohen et al. 2020^a,^b	7.8	0	0	0	1
Williamson et al. 2023^a	1.08^c	0^b	0^c	2^c	0^c
Williamson et al. 2023^a	1.62^d	4^d	0^d	0^d	0^d

Open in a new tab

^{^a}

Included syndromic patients.

^{^b}

Only reported for 27 of 36 patients.

^{^c}

Patients underwent tongue base reduction alone.

^{^d}

Patients underwent tongue base reduction + lingual tonsillectomy.

Discussion

While AT can be highly effective in treating children with OSA, many patients will have persistent OSA that may continue to negatively impact general health and quality of life. Known risk factors for persistent OSA despite AT include obesity and those older than 7 years old, craniofacial abnormalities, trisomy 21, and neurologic disorders. ⁴⁶ , ⁴⁷ , ⁴⁸ , ⁴⁹ For many patients with refractory OSA, the tongue base is identified as a common source of obstruction. ²⁹ , ³⁰ , ³¹ The studies included in this systematic review suggest that tongue reduction surgery is a safe and effective treatment option for pediatric patients with OSA and obstruction secondary to glossoptosis or macroglossia. The studies included patients who underwent tongue reduction surgery for primary treatment of OSA with tongue reduction as well as patients with refractory OSA despite AT.

Safety and Efficacy Profile

The reported complication rate in the studies included in this review was relatively low with the most common complication being postoperative bleeding, seen in seven patients out of the 149 total patients (4.7%) which is comparable to the postoperative bleeding rate of palatine tonsillectomy. ⁵⁰ Postoperative hospital length of stay varied greatly among the studies ranging from less than 24 hours to 19.3 days (Table 3). However, 4 of the 9 studies (Thottam et al 2015, Montevecchi et al 2017, Propst et al 2017, Ulualp 2019, and Willamson et al 2023) reported mean hospital stay of less than 1 week. Lefaivre et al reported the longest mean hospital stay postoperatively but this is likely because patients in this study underwent postoperative sleep study prior to discharge.

While all studies in this review reported clinical success of surgical tongue reduction, only 5 studies (Thottam et al 2015, Propst et al 2017, Ulualp 2019, Cohen et al 2020, and Williamson et al 2023) reported statistically significant improvement in AHI or oAHI. ²⁰ , ⁴¹ , ⁴² , ⁴³ , ⁴⁴ The remaining studies did report improvements in AHI. However, the sample sizes were insufficient to demonstrate statistical difference. While postoperative AHI improvement demonstrated by the studies in this review (Table 2) is compelling, 5 studies reported postoperative AHI ≥ 5 indicating persistent moderate OSA and 4 studies reported postoperative AHI ≥ 10 indicating persistent severe OSA. ²⁰ , ²² , ⁴¹ , ⁴² , ⁴³ It is important to note that the preoperative AHI for the patients in these studies was considerably higher compared to the other studies. The largest study (Williamson et al 2023) uniquely included many patients with mild to moderate OSA (preoperative mean oAHI ranged from 1.28 to 88.8) but also reported stricter criteria for surgical success. ⁴⁴ In Williamson et al, surgical failure was defined as either oAHI ≥1 with concurrent symptoms of OSA or oAHI ≥5 regardless of symptoms. Surgical failure was identified in 5 patients (20%) in the MPG group and 11 patients (26.2%) in the MPG + LT group. ⁴⁴ In the Propst et al study, BMI was analyzed to assess impact on outcomes. The only significant decrease in oAHI was seen in normal‐overweight children who remained normal/overweight after surgery (47.0‐5.6, P = .031). ⁴² Interestingly, oAHI increased, but not significantly, in obese patients who remained obese postoperatively (23.7‐30.2, P > .05). ⁴²

A recent systematic review and meta‐analysis by Camacho et al 2017 evaluated the efficacy of tongue surgeries for pediatric appropriate sleep apnea. ³² This systematic review included studies on patients <18 years old with OSA who underwent tongue surgery alone. The tongue surgeries included in that study were base of tongue reduction (n = 114), tongue base suspension suture (n = 1), and hypoglossal nerve stimulation (n = 1). ³² While there is overlap between this systematic review and the review presented here there are important differences. The tongue base reduction surgeries in the systematic review by Camacho et al included LT, MPG, and SMILE. In contrast, we included studies that underwent multilevel sleep surgery. Additionally, we did not include studies that only reported LT to focus on tongue reduction surgery by excision of a portion of the intrinsic tongue. This systematic review provides an important review of different surgical techniques that have been used to relieve tongue obstruction due to congenital or acquired macroglossia rather than lingual tonsil hypertrophy or glossoptosis.

DISE and CINE MRI

For children with persistent OSA despite AT, there is considerable variation in the clinical workup and management. DISE and CINE MRI have allowed otolaryngologists to identify the specific sites of obstruction, allowing for targeted surgery. ²⁸ , ³⁰ , ⁵¹ , ⁵² DISE and CINE MRI have been used alone or to complement each other. The studies identified during this systematic review varied greatly in the clinical workup prior to partial glossectomy which included identification of macroglossia on physical exam, DISE and CINE MRI. While DISE was commonly used to determine presence of tongue base obstruction, the findings on DISE were not reported quantitatively and different DISE scoring systems were used in each of the studies which limits comparison of indications between studies. Future research would benefit from reporting DISE findings and using a standardized and validated scoring system such as the International Pediatric Sleep Endoscopy Scale (IPSES). ⁵³ , ⁵⁴ CINE MRI can complement DISE by allowing for soft tissue evaluation of the tongue base and thickness of lingual tonsil tissue if present. CINE MRI can also provide quantitative measurements of airway diameter.

Study Limitations

While surgical tongue reduction appears to be a safe and effective treatment option for children with OSA, there are several limitations of this systematic review to consider. First, the majority of included studies were case series or small cohorts, which are inherently prone to bias and may not provide high‐quality evidence. It is important to note that the quality of studies included in this systematic review ranged from fair to poor based on the modified Downs and Black checklist for assessment of methodological quality. This highlights a need for more robust prospective and controlled studies on this topic.

The heterogeneity of surgical techniques and patient populations across studies makes it challenging to compare outcomes directly. For example, patients in five studies underwent additional airway surgeries. ²⁰ , ²¹ , ²² , ²³ , ⁴⁴ These included adenoidectomy, tonsillectomy, uvulopalatopharyngoplasty, septoplasty, turbinate reduction, supraglottoplasty, hyoid suspension, LT, mandibular distraction, and maxillomandibular advancement. Inclusion of these other procedures makes it challenging to draw definitive conclusions on efficacy of tongue base reduction alone. Additionally, some studies in this review included only patients with Trisomy 21 or Beckwith‐Wiedemann Syndrome while others included both syndromic and nonsyndromic patients further adding to the heterogeneity.

Beyond these differences in patient populations and surgical interventions, there also may exist inconsistency in outcome reporting across studies. The exact timing of PSG postoperatively was often not reported, and the quality of PSG may vary between institutions. Patient‐reported outcomes of postoperative OSA symptoms and long‐term recurrence data were often not available or inconsistently reported. Furthermore, the decision to proceed with tongue reduction surgery was guided by a variety of methods ranging from physical exam to DISE or CINE MRI. The precise findings on DISE and CINE MRI were inconsistently reported or absent. For these reasons, a meta‐analysis was not felt to be appropriate which also limits the impact of the study.

Finally, publication bias may also be present limiting this systematic review. Tongue reduction for pediatric OSA is an uncommon procedure and the included studies were relatively small. Thus, publications may be disproportionately driven by centers with relatively higher volume and more successful outcomes.

Conclusion

Tongue reduction surgery appears to be a valuable treatment option for pediatric patients with OSA who do not respond to conventional treatments like AT or CPAP and for those with primary macroglossia contributing to OSA. While these procedures can effectively reduce AHI and improve oxygen saturation, leading to symptomatic improvement in patients, there remains room for further improvement as there are still patients with persistent OSA despite surgical reduction of the tongue base. It is not clear which of the surgical methods identified in this review are superior or which patient population would benefit the most from surgical tongue reduction. While further research is needed to address the limitations of the current evidence, tongue reduction surgery can be considered a safe and effective site‐directed intervention option for pediatric OSA.

Meeting Information

This study was presented at the American Society of Pediatric Otolaryngology Annual Meeting at Combined Otolaryngology Spring Meetings (COSM) on May 19, 2024, in Chicago, IL.

Author Contributions

Adrian Williamson, MD, conception of study, design of study, data collection and interpretation, selection of studies to be included and excluded, drafting and editing of manuscript; Rohit Nallani, MD, design of study, data collection and interpretation, selection of studies to be included and excluded, drafting and editing of manuscript; Jason R. Brown, DO, design of study, selection of studies to be included and excluded, drafting and editing of manuscript.

Disclosures

Competing interests

None.

Funding source

None.

Supporting information

Table A1. Web of Science search strategy. This table demonstrates the search strategy used for Web of Science using keywords related to sleep apnea and base of tongue surgery. Filters were applied to limit the results to pediatric populations. Table A2. PubMed search strategy. This table outlines the PubMed search strategy using a combination of MeSH terms, keywords, and Boolean operators. Filters were applied to limit the results to pediatric populations and exclude case reports. Table A3. Embase search strategy. This table outlines the Embase search strategy including a combination of MeSH terms, keywords, and Boolean operators. Filters were applied to limit results to pediatric populations while excluding case reports. Table A4. Summary of author agreement. This table presents the agreement levels among authors during abstract and title screening, as well as during full‐text review.

OTO2-10-e70215-s001.docx^{(19.6KB, docx)}

Acknowledgments

We would like to thank Children's Mercy Library Services and Keri Swaggart, MLIS, AHIP who assisted with the electronic database search.

References

1. Bixler EO, Vgontzas AN, Lin HM, et al. Sleep disordered breathing in children in a general population sample: prevalence and risk factors. Sleep. 2009;32(6):731‐736. 10.1093/sleep/32.6.731 [DOI] [PMC free article] [PubMed] [Google Scholar]
2. Ishman SL, Tawfik KO, Smith DF, et al. Screening for pediatric obstructive sleep apnea before ambulatory surgery. J Clin Sleep Med. 2015;11(07):751‐755. 10.5664/jcsm.4852 [DOI] [PMC free article] [PubMed] [Google Scholar]
3. Gulotta G, Iannella G, Vicini C, et al. Risk factors for obstructive sleep apnea syndrome in children: state of the art. Int J Environ Res Public Health. 2019;16(18):3235. 10.3390/ijerph16183235 [DOI] [PMC free article] [PubMed] [Google Scholar]
4. Alkhalifah K, Alsalhi S, Alrashed A, et al. Worldwide prevalence of obstructive sleep apnea among pediatrics: a systematic review and meta‐analysis. Int J Med Dev Countries. 2024; 8(1):455‐459. 10.24911/IJMDC.51-1701581085 [DOI] [Google Scholar]
5. Marcus CL, Brooks LJ, Draper KA, et al. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2012;130(3):576‐584. 10.1542/peds.2012-1671 [DOI] [PubMed] [Google Scholar]
6. Capdevila OS, Kheirandish‐Gozal L, Dayyat E, Gozal D. Pediatric obstructive sleep apnea: complications, management, and long‐term outcomes. Proc Am Thorac Soc. 2008;5(2):274‐282. 10.1513/pats.200708-138MG [DOI] [PMC free article] [PubMed] [Google Scholar]
7. Gozal D. Sleep, sleep disorders and inflammation in children. Sleep Med. 2009;10:S12‐S16. 10.1016/j.sleep.2009.07.003 [DOI] [PubMed] [Google Scholar]
8. O'Brien LM, Holbrook CR, Mervis CB, et al. Sleep and neurobehavioral characteristics of 5‐ to 7‐year‐old children with parentally reported symptoms of attention‐deficit/hyperactivity disorder. Pediatrics. 2003;111(3):554‐563. 10.1542/peds.111.3.554 [DOI] [PubMed] [Google Scholar]
9. Suratt PM, Barth JT, Diamond R, et al. Reduced time in bed and obstructive sleep‐disordered breathing in children are associated with cognitive impairment. Pediatrics. 2007;119(2):320‐329. 10.1542/peds.2006-1969 [DOI] [PubMed] [Google Scholar]
10. Mitchell RB, Kelly J. Behavior, neurocognition and quality‐of‐life in children with sleep‐disordered breathing. Int J Pediatr Otorhinolaryngol. 2006;70(3):395‐406. 10.1016/j.ijporl.2005.10.020 [DOI] [PubMed] [Google Scholar]
11. Bhattacharjee R, Kheirandish‐Gozal L, Pillar G, Gozal D. Cardiovascular complications of obstructive sleep apnea syndrome: evidence from children. Prog Cardiovasc Dis. 2009;51(5):416‐433. 10.1016/j.pcad.2008.03.002 [DOI] [PubMed] [Google Scholar]
12. Li AM, Au CT, Sung RYT, et al. Ambulatory blood pressure in children with obstructive sleep apnoea: a community based study. Thorax. 2008;63(9):803‐809. 10.1136/thx.2007.091132. [DOI] [PubMed] [Google Scholar]
13. Dutt N, Janmeja A, Mohapatra P, Singh A. Quality of life impairment in patients of obstructive sleep apnea and its relation with the severity of disease. Lung India. 2013;30(4):289. 10.4103/0970-2113.120603. [DOI] [PMC free article] [PubMed] [Google Scholar]
14. Baldassari CM, Mitchell RB, Schubert C, Rudnick EF. Pediatric obstructive sleep apnea and quality of life: a meta‐analysis. Otolaryngol Head Neck Surg. 2008;138(3):265‐273. 10.1016/j.otohns.2007.11.003 [DOI] [PubMed] [Google Scholar]
15. Mitchell RB, Archer SM, Ishman SL, et al. Clinical practice guideline: tonsillectomy in children (update)‐executive summary. Otolaryngol Head Neck Surg. 2019;160(2):187‐205. 10.1177/0194599818807917 [DOI] [PubMed] [Google Scholar]
16. Brietzke SE, Gallagher D. The effectiveness of tonsillectomy and adenoidectomy in the treatment of pediatric obstructive sleep apnea/hypopnea syndrome: a meta‐analysis. Otolaryngol Head Neck Surg. 2006;134(6):979‐984. 10.1016/j.otohns.2006.02.033 [DOI] [PubMed] [Google Scholar]
17. Friedman M, Wilson M, Lin HC, Chang HW. Updated systematic review of tonsillectomy and adenoidectomy for treatment of pediatric obstructive sleep apnea/hypopnea syndrome. Otolaryngol Head Neck Surg. 2009;140(6):800‐808. 10.1016/j.otohns.2009.01.043 [DOI] [PubMed] [Google Scholar]
18. Tauman R, Gulliver TE, Krishna J, et al. Persistence of obstructive sleep apnea syndrome in children after adenotonsillectomy. J Pediatr. 2006;149(6):803‐808. 10.1016/j.jpeds.2006.08.067 [DOI] [PubMed] [Google Scholar]
19. O'Brien LM, Sitha S, Baur LA, Waters KA. Obesity increases the risk for persisting obstructive sleep apnea after treatment in children. Int J Pediatr Otorhinolaryngol. 2006;70(9):1555‐1560. 10.1016/j.ijporl.2006.04.003 [DOI] [PubMed] [Google Scholar]
20. Cohen JL, Cielo CM, Kupa J, et al. The utility of early tongue reduction surgery for macroglossia in Beckwith‐Wiedemann syndrome. Plast Reconstruct Surg. 2020;145(4):803e‐813e. 10.1097/PRS.0000000000006673 [DOI] [PMC free article] [PubMed] [Google Scholar]
21. Lefaivre JF, Cohen SR, Burstein FD, et al. Down syndrome: identification and surgical management of obstructive sleep apnea. Plast Reconstr Surg. 1997;99(3):629‐637. 10.1097/00006534-199703000-00004 [DOI] [PubMed] [Google Scholar]
22. Dmowska J, Larson SR, Gillespie MB, Sheyn A. Effect of drug induced sleep endoscopy on intraoperative decision making in pediatric sleep surgery. Int J Pediatr Otorhinolaryngol. 2020;130:109810. 10.1016/j.ijporl.2019.109810 [DOI] [PubMed] [Google Scholar]
23. Montevecchi F, Bellini C, Meccariello G, et al. Transoral robotic‐assisted tongue base resection in pediatric obstructive sleep apnea syndrome: case presentation, clinical and technical consideration. Eur Arch Otrhinolaryngol. 2017;274(2):1161‐1166. 10.1007/s00405-016-4269-x [DOI] [PubMed] [Google Scholar]
24. Williamson A, McArdle EH, Morrow VR, Zalzal HG, Carr MM, Coutras SW. Base of Tongue Surgery and Pediatric Obstructive Sleep Apnea. Otolaryngol Head Neck Surg. 2023;168(4):839‐847. 10.1177/01945998221094211 [DOI] [PubMed] [Google Scholar]
25. Roland PS, Rosenfeld RM, Brooks LJ, et al. Clinical practice guideline: polysomnography for sleep‐disordered breathing prior to tonsillectomy in children. Otolaryngol Head Neck Surg. 2011;145(1 Suppl):S1‐S15. 10.1177/0194599811409837 [DOI] [PubMed] [Google Scholar]
26. Ishman SL, Maturo S, Schwartz S, et al. Expert consensus statement: management of pediatric persistent obstructive sleep apnea after adenotonsillectomy. Otolaryngol Head Neck Surg. 2023;168(2):115‐130. 10.1002/ohn.159 [DOI] [PMC free article] [PubMed] [Google Scholar]
27. Liming BJ, Ryan M, Mack D, Ahmad I, Camacho M. Montelukast and nasal corticosteroids to treat pediatric obstructive sleep apnea: a systematic review and meta‐analysis. Otolaryngol Head Neck Surg. 2019;160(4):594‐602. 10.1177/0194599818815683 [DOI] [PubMed] [Google Scholar]
28. Manickam PV, Shott SR, Boss EF, et al. Systematic review of site of obstruction identification and non‐CPAP treatment options for children with persistent pediatric obstructive sleep apnea. Laryngoscope. 2016;126(2):491‐500. 10.1002/lary.25459 [DOI] [PubMed] [Google Scholar]
29. Socarras MA, Landau BP, Durr ML. Diagnostic techniques and surgical outcomes for persistent pediatric obstructive sleep apnea after adenotonsillectomy: a systematic review and meta‐analysis. Int J Pediatr Otorhinolaryngol. 2019;121:179‐187. 10.1016/j.ijporl.2019.02.030 [DOI] [PubMed] [Google Scholar]
30. Durr ML, Meyer AK, Kezirian EJ, Rosbe KW. Drug‐induced sleep endoscopy in persistent pediatric sleep‐disordered breathing after adenotonsillectomy. Arch Otolaryngol Head Neck Surg. 2012;138(7):638. 10.1001/archoto.2012.1067 [DOI] [PubMed] [Google Scholar]
31. Clark C, Ulualp SO. Multimodality assessment of upper airway obstruction in children with persistent obstructive sleep apnea after adenotonsillectomy. Laryngoscope. 2017;127(5):1224‐1230. 10.1002/lary.26174 [DOI] [PubMed] [Google Scholar]
32. Camacho M, Noller MW, Zaghi S, et al. Tongue surgeries for pediatric obstructive sleep apnea: a systematic review and meta‐analysis. Eur Arch Otrhinolaryngol. 2017;274(8):2981‐2990. 10.1007/s00405-017-4545-4 [DOI] [PubMed] [Google Scholar]
33. Diercks GR, Wentland C, Keamy D, et al. Hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea. JAMA Otolaryngol Head Neck Surg. 2017;144(1):37‐42. 10.1001/jamaoto.2017.1871 [DOI] [PMC free article] [PubMed] [Google Scholar]
34. Stenerson ME, Yu PK, Kinane TB, Skotko BG, Hartnick CJ. Long‐term stability of hypoglossal nerve stimulation for the treatment of obstructive sleep apnea in children with Down syndrome. Int J Pediatr Otorhinolaryngol. 2021;149:110868. 10.1016/j.ijporl.2021.110868 [DOI] [PubMed] [Google Scholar]
35. Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. 10.1136/bmj.n71 [DOI] [PMC free article] [PubMed] [Google Scholar]
36.Covidence. www.covidence.org
37. Downs SH, Black N. The feasibility of creating a checklist for the assessment of the methodological quality both of randomised and non‐randomised studies of health care interventions. J Epidemiol Community Health. 1998;52(6):377‐384. [DOI] [PMC free article] [PubMed] [Google Scholar]
38. Almufarrij I, Dillon H, Munro KJ. Is the outcome of fitting hearing aids to adults affected by whether an audiogram‐based prescription formula is individually applied? A systematic review protocol. BMJ Open. 2021;11(8):e045899. 10.1136/bmjopen-2020-045899 [DOI] [PMC free article] [PubMed] [Google Scholar]
39. Scarpa A, Viola P, Ralli M, et al. Post‐operative radiotherapy in adenoid cystic carcinoma of salivary glands versus surgery alone: what is the evidence about survival and local control? A systematic review and meta‐analysis. Eur Arch Otrhinolaryngol. 2024;281(2):563‐571. 10.1007/s00405-023-08252-x [DOI] [PubMed] [Google Scholar]
40. Maturo SC, Mair EA. Submucosal minimally invasive lingual excision: an effective, novel surgery for pediatric tongue base reduction. Ann Otol, Rhinol, Laryngol. 2006;115(8):624‐630. 10.1177/000348940611500809 [DOI] [PubMed] [Google Scholar]
41. Thottam PJ, Govil N, Duvvuri U, Mehta D. Transoral robotic surgery for sleep apnea in children: is it effective? Int J Pediatr Otorhinolaryngol. 2015;79(12):2234‐2237. 10.1016/j.ijporl.2015.10.010 [DOI] [PubMed] [Google Scholar]
42. Propst EJ, Amin R, Talwar N, et al. Midline posterior glossectomy and lingual tonsillectomy in obese and nonobese children with down syndrome: biomarkers for success. Laryngoscope. 2017;127(3):757‐763. 10.1002/lary.26104 [DOI] [PubMed] [Google Scholar]
43. Ulualp S. Outcomes of tongue base reduction and lingual tonsillectomy for residual pediatric obstructive sleep apnea after adenotonsillectomy. Int Arch Otorhinolaryngol. 2019;23(4):e415‐e421. 10.1055/s-0039-1685156 [DOI] [PMC free article] [PubMed] [Google Scholar]
44. Williamson A, McArdle EH, Morrow VR, Zalzal HG, Carr MM, Coutras SW. Base of tongue surgery and pediatric obstructive sleep apnea. Otolaryngol Head Neck Surg. 2023;168(4):839‐847. 10.1177/01945998221094211 [DOI] [PubMed] [Google Scholar]
45. Ishman SL, Chang KW, Kennedy AA. Techniques for evaluation and management of tongue‐base obstruction in pediatric obstructive sleep apnea. Curr Opin Otolaryngol Head Neck Surg. 2018;26(6):409‐416. 10.1097/MOO.0000000000000489 [DOI] [PubMed] [Google Scholar]
46. Bhattacharjee R, Kheirandish‐Gozal L, Spruyt K, et al. Adenotonsillectomy outcomes in treatment of obstructive sleep apnea in children: a multicenter retrospective study. Am J Respir Crit Care Med. 2010;182(5):676‐683. 10.1164/rccm.200912-1930OC [DOI] [PubMed] [Google Scholar]
47. Hoeve LJH, Pijpers M, Joosten KFM. OSAS in craniofacial syndromes: an unsolved problem. Int J Pediatr Otorhinolaryngol. 2003;67:S111‐S113. 10.1016/j.ijporl.2003.08.007 [DOI] [PubMed] [Google Scholar]
48. Shete MM, Stocks RMS, Sebelik ME, Schoumacher RA. Effects of adeno‐tonsillectomy on polysomnography patterns in Down syndrome children with obstructive sleep apnea: a comparative study with children without Down syndrome. Int J Pediatr Otorhinolaryngol. 2010;74(3):241‐244. 10.1016/j.ijporl.2009.11.006 [DOI] [PubMed] [Google Scholar]
49. Ali NES, Alyono JC, Kumar AR, Cheng H, Koltai PJ. Sleep surgery in syndromic and neurologically impaired children. Am J Otolaryngol. 2020;41(4):102566. 10.1016/j.amjoto.2020.102566 [DOI] [PubMed] [Google Scholar]
50. Gutierrez JA, Shannon CM, Nguyen SA, Labadie RF, White DR. The impact of surgical indication on posttonsillectomy hemorrhage: a systematic review and meta‐analysis. Otolaryngol Head Neck Surg. 2023;169(4):780‐791. 10.1002/ohn.339 [DOI] [PubMed] [Google Scholar]
51. Baldassari CM, Lam DJ, Ishman SL, et al. Expert consensus statement: pediatric drug‐induced sleep endoscopy. Otolaryngol Head Neck Surg. 2021;165(4):578‐591. 10.1177/0194599820985000 [DOI] [PubMed] [Google Scholar]
52. Isaiah A, Kiss E, Olomu P, Koral K, Mitchell RB. Characterization of upper airway obstruction using cine MRI in children with residual obstructive sleep apnea after adenotonsillectomy. Sleep Med. 2018;50:79‐86. 10.1016/j.sleep.2017.10.006 [DOI] [PubMed] [Google Scholar]
53. Parikh SR, Boudewyns A, Friedman NR, et al. International Pediatric Otolaryngology Group (IPOG) consensus on scoring of pediatric Drug Induced Sleep Endoscopy (DISE. Int J Pediatr Otorhinolaryngol. 2023;171:111627. 10.1016/j.ijporl.2023.111627 [DOI] [PubMed] [Google Scholar]
54. Lam DJ, Friedman NR, Chan KC, et al. Development and validation of the international pediatric sleep endoscopy scale (IPSES). Int J Pediatr Otorhinolaryngol. 2023;174:111748. 10.1016/j.ijporl.2023.111748 [DOI] [PubMed] [Google Scholar]

Associated Data

This section collects any data citations, data availability statements, or supplementary materials included in this article.

Supplementary Materials

OTO2-10-e70215-s001.docx^{(19.6KB, docx)}

[oto270215-bib-0001] 1. Bixler EO, Vgontzas AN, Lin HM, et al. Sleep disordered breathing in children in a general population sample: prevalence and risk factors. Sleep. 2009;32(6):731‐736. 10.1093/sleep/32.6.731 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0002] 2. Ishman SL, Tawfik KO, Smith DF, et al. Screening for pediatric obstructive sleep apnea before ambulatory surgery. J Clin Sleep Med. 2015;11(07):751‐755. 10.5664/jcsm.4852 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0003] 3. Gulotta G, Iannella G, Vicini C, et al. Risk factors for obstructive sleep apnea syndrome in children: state of the art. Int J Environ Res Public Health. 2019;16(18):3235. 10.3390/ijerph16183235 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0004] 4. Alkhalifah K, Alsalhi S, Alrashed A, et al. Worldwide prevalence of obstructive sleep apnea among pediatrics: a systematic review and meta‐analysis. Int J Med Dev Countries. 2024; 8(1):455‐459. 10.24911/IJMDC.51-1701581085 [DOI] [Google Scholar]

[oto270215-bib-0005] 5. Marcus CL, Brooks LJ, Draper KA, et al. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2012;130(3):576‐584. 10.1542/peds.2012-1671 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0006] 6. Capdevila OS, Kheirandish‐Gozal L, Dayyat E, Gozal D. Pediatric obstructive sleep apnea: complications, management, and long‐term outcomes. Proc Am Thorac Soc. 2008;5(2):274‐282. 10.1513/pats.200708-138MG [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0007] 7. Gozal D. Sleep, sleep disorders and inflammation in children. Sleep Med. 2009;10:S12‐S16. 10.1016/j.sleep.2009.07.003 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0008] 8. O'Brien LM, Holbrook CR, Mervis CB, et al. Sleep and neurobehavioral characteristics of 5‐ to 7‐year‐old children with parentally reported symptoms of attention‐deficit/hyperactivity disorder. Pediatrics. 2003;111(3):554‐563. 10.1542/peds.111.3.554 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0009] 9. Suratt PM, Barth JT, Diamond R, et al. Reduced time in bed and obstructive sleep‐disordered breathing in children are associated with cognitive impairment. Pediatrics. 2007;119(2):320‐329. 10.1542/peds.2006-1969 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0010] 10. Mitchell RB, Kelly J. Behavior, neurocognition and quality‐of‐life in children with sleep‐disordered breathing. Int J Pediatr Otorhinolaryngol. 2006;70(3):395‐406. 10.1016/j.ijporl.2005.10.020 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0011] 11. Bhattacharjee R, Kheirandish‐Gozal L, Pillar G, Gozal D. Cardiovascular complications of obstructive sleep apnea syndrome: evidence from children. Prog Cardiovasc Dis. 2009;51(5):416‐433. 10.1016/j.pcad.2008.03.002 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0012] 12. Li AM, Au CT, Sung RYT, et al. Ambulatory blood pressure in children with obstructive sleep apnoea: a community based study. Thorax. 2008;63(9):803‐809. 10.1136/thx.2007.091132. [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0013] 13. Dutt N, Janmeja A, Mohapatra P, Singh A. Quality of life impairment in patients of obstructive sleep apnea and its relation with the severity of disease. Lung India. 2013;30(4):289. 10.4103/0970-2113.120603. [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0014] 14. Baldassari CM, Mitchell RB, Schubert C, Rudnick EF. Pediatric obstructive sleep apnea and quality of life: a meta‐analysis. Otolaryngol Head Neck Surg. 2008;138(3):265‐273. 10.1016/j.otohns.2007.11.003 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0015] 15. Mitchell RB, Archer SM, Ishman SL, et al. Clinical practice guideline: tonsillectomy in children (update)‐executive summary. Otolaryngol Head Neck Surg. 2019;160(2):187‐205. 10.1177/0194599818807917 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0016] 16. Brietzke SE, Gallagher D. The effectiveness of tonsillectomy and adenoidectomy in the treatment of pediatric obstructive sleep apnea/hypopnea syndrome: a meta‐analysis. Otolaryngol Head Neck Surg. 2006;134(6):979‐984. 10.1016/j.otohns.2006.02.033 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0017] 17. Friedman M, Wilson M, Lin HC, Chang HW. Updated systematic review of tonsillectomy and adenoidectomy for treatment of pediatric obstructive sleep apnea/hypopnea syndrome. Otolaryngol Head Neck Surg. 2009;140(6):800‐808. 10.1016/j.otohns.2009.01.043 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0018] 18. Tauman R, Gulliver TE, Krishna J, et al. Persistence of obstructive sleep apnea syndrome in children after adenotonsillectomy. J Pediatr. 2006;149(6):803‐808. 10.1016/j.jpeds.2006.08.067 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0019] 19. O'Brien LM, Sitha S, Baur LA, Waters KA. Obesity increases the risk for persisting obstructive sleep apnea after treatment in children. Int J Pediatr Otorhinolaryngol. 2006;70(9):1555‐1560. 10.1016/j.ijporl.2006.04.003 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0020] 20. Cohen JL, Cielo CM, Kupa J, et al. The utility of early tongue reduction surgery for macroglossia in Beckwith‐Wiedemann syndrome. Plast Reconstruct Surg. 2020;145(4):803e‐813e. 10.1097/PRS.0000000000006673 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0021] 21. Lefaivre JF, Cohen SR, Burstein FD, et al. Down syndrome: identification and surgical management of obstructive sleep apnea. Plast Reconstr Surg. 1997;99(3):629‐637. 10.1097/00006534-199703000-00004 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0022] 22. Dmowska J, Larson SR, Gillespie MB, Sheyn A. Effect of drug induced sleep endoscopy on intraoperative decision making in pediatric sleep surgery. Int J Pediatr Otorhinolaryngol. 2020;130:109810. 10.1016/j.ijporl.2019.109810 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0023] 23. Montevecchi F, Bellini C, Meccariello G, et al. Transoral robotic‐assisted tongue base resection in pediatric obstructive sleep apnea syndrome: case presentation, clinical and technical consideration. Eur Arch Otrhinolaryngol. 2017;274(2):1161‐1166. 10.1007/s00405-016-4269-x [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0024] 24. Williamson A, McArdle EH, Morrow VR, Zalzal HG, Carr MM, Coutras SW. Base of Tongue Surgery and Pediatric Obstructive Sleep Apnea. Otolaryngol Head Neck Surg. 2023;168(4):839‐847. 10.1177/01945998221094211 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0025] 25. Roland PS, Rosenfeld RM, Brooks LJ, et al. Clinical practice guideline: polysomnography for sleep‐disordered breathing prior to tonsillectomy in children. Otolaryngol Head Neck Surg. 2011;145(1 Suppl):S1‐S15. 10.1177/0194599811409837 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0026] 26. Ishman SL, Maturo S, Schwartz S, et al. Expert consensus statement: management of pediatric persistent obstructive sleep apnea after adenotonsillectomy. Otolaryngol Head Neck Surg. 2023;168(2):115‐130. 10.1002/ohn.159 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0027] 27. Liming BJ, Ryan M, Mack D, Ahmad I, Camacho M. Montelukast and nasal corticosteroids to treat pediatric obstructive sleep apnea: a systematic review and meta‐analysis. Otolaryngol Head Neck Surg. 2019;160(4):594‐602. 10.1177/0194599818815683 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0028] 28. Manickam PV, Shott SR, Boss EF, et al. Systematic review of site of obstruction identification and non‐CPAP treatment options for children with persistent pediatric obstructive sleep apnea. Laryngoscope. 2016;126(2):491‐500. 10.1002/lary.25459 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0029] 29. Socarras MA, Landau BP, Durr ML. Diagnostic techniques and surgical outcomes for persistent pediatric obstructive sleep apnea after adenotonsillectomy: a systematic review and meta‐analysis. Int J Pediatr Otorhinolaryngol. 2019;121:179‐187. 10.1016/j.ijporl.2019.02.030 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0030] 30. Durr ML, Meyer AK, Kezirian EJ, Rosbe KW. Drug‐induced sleep endoscopy in persistent pediatric sleep‐disordered breathing after adenotonsillectomy. Arch Otolaryngol Head Neck Surg. 2012;138(7):638. 10.1001/archoto.2012.1067 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0031] 31. Clark C, Ulualp SO. Multimodality assessment of upper airway obstruction in children with persistent obstructive sleep apnea after adenotonsillectomy. Laryngoscope. 2017;127(5):1224‐1230. 10.1002/lary.26174 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0032] 32. Camacho M, Noller MW, Zaghi S, et al. Tongue surgeries for pediatric obstructive sleep apnea: a systematic review and meta‐analysis. Eur Arch Otrhinolaryngol. 2017;274(8):2981‐2990. 10.1007/s00405-017-4545-4 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0033] 33. Diercks GR, Wentland C, Keamy D, et al. Hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea. JAMA Otolaryngol Head Neck Surg. 2017;144(1):37‐42. 10.1001/jamaoto.2017.1871 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0034] 34. Stenerson ME, Yu PK, Kinane TB, Skotko BG, Hartnick CJ. Long‐term stability of hypoglossal nerve stimulation for the treatment of obstructive sleep apnea in children with Down syndrome. Int J Pediatr Otorhinolaryngol. 2021;149:110868. 10.1016/j.ijporl.2021.110868 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0035] 35. Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. 10.1136/bmj.n71 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0036] 36.Covidence. www.covidence.org

[oto270215-bib-0037] 37. Downs SH, Black N. The feasibility of creating a checklist for the assessment of the methodological quality both of randomised and non‐randomised studies of health care interventions. J Epidemiol Community Health. 1998;52(6):377‐384. [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0038] 38. Almufarrij I, Dillon H, Munro KJ. Is the outcome of fitting hearing aids to adults affected by whether an audiogram‐based prescription formula is individually applied? A systematic review protocol. BMJ Open. 2021;11(8):e045899. 10.1136/bmjopen-2020-045899 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0039] 39. Scarpa A, Viola P, Ralli M, et al. Post‐operative radiotherapy in adenoid cystic carcinoma of salivary glands versus surgery alone: what is the evidence about survival and local control? A systematic review and meta‐analysis. Eur Arch Otrhinolaryngol. 2024;281(2):563‐571. 10.1007/s00405-023-08252-x [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0040] 40. Maturo SC, Mair EA. Submucosal minimally invasive lingual excision: an effective, novel surgery for pediatric tongue base reduction. Ann Otol, Rhinol, Laryngol. 2006;115(8):624‐630. 10.1177/000348940611500809 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0041] 41. Thottam PJ, Govil N, Duvvuri U, Mehta D. Transoral robotic surgery for sleep apnea in children: is it effective? Int J Pediatr Otorhinolaryngol. 2015;79(12):2234‐2237. 10.1016/j.ijporl.2015.10.010 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0042] 42. Propst EJ, Amin R, Talwar N, et al. Midline posterior glossectomy and lingual tonsillectomy in obese and nonobese children with down syndrome: biomarkers for success. Laryngoscope. 2017;127(3):757‐763. 10.1002/lary.26104 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0043] 43. Ulualp S. Outcomes of tongue base reduction and lingual tonsillectomy for residual pediatric obstructive sleep apnea after adenotonsillectomy. Int Arch Otorhinolaryngol. 2019;23(4):e415‐e421. 10.1055/s-0039-1685156 [DOI] [PMC free article] [PubMed] [Google Scholar]

[oto270215-bib-0044] 44. Williamson A, McArdle EH, Morrow VR, Zalzal HG, Carr MM, Coutras SW. Base of tongue surgery and pediatric obstructive sleep apnea. Otolaryngol Head Neck Surg. 2023;168(4):839‐847. 10.1177/01945998221094211 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0045] 45. Ishman SL, Chang KW, Kennedy AA. Techniques for evaluation and management of tongue‐base obstruction in pediatric obstructive sleep apnea. Curr Opin Otolaryngol Head Neck Surg. 2018;26(6):409‐416. 10.1097/MOO.0000000000000489 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0046] 46. Bhattacharjee R, Kheirandish‐Gozal L, Spruyt K, et al. Adenotonsillectomy outcomes in treatment of obstructive sleep apnea in children: a multicenter retrospective study. Am J Respir Crit Care Med. 2010;182(5):676‐683. 10.1164/rccm.200912-1930OC [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0047] 47. Hoeve LJH, Pijpers M, Joosten KFM. OSAS in craniofacial syndromes: an unsolved problem. Int J Pediatr Otorhinolaryngol. 2003;67:S111‐S113. 10.1016/j.ijporl.2003.08.007 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0048] 48. Shete MM, Stocks RMS, Sebelik ME, Schoumacher RA. Effects of adeno‐tonsillectomy on polysomnography patterns in Down syndrome children with obstructive sleep apnea: a comparative study with children without Down syndrome. Int J Pediatr Otorhinolaryngol. 2010;74(3):241‐244. 10.1016/j.ijporl.2009.11.006 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0049] 49. Ali NES, Alyono JC, Kumar AR, Cheng H, Koltai PJ. Sleep surgery in syndromic and neurologically impaired children. Am J Otolaryngol. 2020;41(4):102566. 10.1016/j.amjoto.2020.102566 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0050] 50. Gutierrez JA, Shannon CM, Nguyen SA, Labadie RF, White DR. The impact of surgical indication on posttonsillectomy hemorrhage: a systematic review and meta‐analysis. Otolaryngol Head Neck Surg. 2023;169(4):780‐791. 10.1002/ohn.339 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0051] 51. Baldassari CM, Lam DJ, Ishman SL, et al. Expert consensus statement: pediatric drug‐induced sleep endoscopy. Otolaryngol Head Neck Surg. 2021;165(4):578‐591. 10.1177/0194599820985000 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0052] 52. Isaiah A, Kiss E, Olomu P, Koral K, Mitchell RB. Characterization of upper airway obstruction using cine MRI in children with residual obstructive sleep apnea after adenotonsillectomy. Sleep Med. 2018;50:79‐86. 10.1016/j.sleep.2017.10.006 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0053] 53. Parikh SR, Boudewyns A, Friedman NR, et al. International Pediatric Otolaryngology Group (IPOG) consensus on scoring of pediatric Drug Induced Sleep Endoscopy (DISE. Int J Pediatr Otorhinolaryngol. 2023;171:111627. 10.1016/j.ijporl.2023.111627 [DOI] [PubMed] [Google Scholar]

[oto270215-bib-0054] 54. Lam DJ, Friedman NR, Chan KC, et al. Development and validation of the international pediatric sleep endoscopy scale (IPSES). Int J Pediatr Otorhinolaryngol. 2023;174:111748. 10.1016/j.ijporl.2023.111748 [DOI] [PubMed] [Google Scholar]

PERMALINK

Tongue Reduction for Pediatric Obstructive Sleep Apnea: A Systematic Review

Adrian Williamson IV, MD

Rohit Nallani, MD

Jason R Brown, DO

Abstract

Objective

Data Sources

Review Methods

Results

Conclusion

Methods

Study Design

Inclusion and Exclusion Criteria

Data Extraction

Results

Literature Search and Study Screening

Figure 1.

Study Characteristics and Patients

Table 1.

Surgical Techniques

Clinical Efficacy and Safety

Table 2.

Table 3.

Discussion

Safety and Efficacy Profile

DISE and CINE MRI

Study Limitations

Conclusion

Meeting Information

Author Contributions

Disclosures

Competing interests

Funding source

Supporting information

Acknowledgments

References

Associated Data

Supplementary Materials

ACTIONS

PERMALINK

RESOURCES

Similar articles

Cited by other articles

Links to NCBI Databases