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Treatment of for Reasons Other Than : A Systematic Review Sivakumar Chinnadurai, MD, MPHa, David O. Francis, MD, MSa,b, Richard A. Epstein, PhD, MPHc,d, Anna Morad, MDe, Sahar Kohanim, MDf, Melissa McPheeters, PhD, MPHc,g

BACKGROUND AND OBJECTIVE: Children with ankyloglossia, an abnormally short, thickened, or tight abstract lingual frenulum, may have restricted mobility and sequelae, such as speech and feeding difficulties and social concerns. We systematically reviewed literature on feeding, speech, and social outcomes of treatments for infants and children with ankyloglossia. METHODS: Medline, PsycINFO, Cumulative Index of Nursing and Allied Health Literature, and Embase were searched. Two reviewers independently assessed studies against predetermined inclusion/exclusion criteria. Two investigators independently extracted data on study populations, interventions, and outcomes and assessed study quality. RESULTS: Two randomized controlled trials, 2 cohort studies, and 11 case series assessed the effects of frenotomy on feeding, speech, and social outcomes. Bottle feeding and social concerns, such as ability to use the tongue to eat ice cream and clean the , improved more in treatment groups in comparative studies. Supplementary bottle feedings decreased over time in case series. Two cohort studies reported improvement in articulation and intelligibility with treatment. Other benefits were unclear. One randomized controlled trial reported improved articulation after Z-frenuloplasty compared with horizontal-to-vertical frenuloplasty. Numerous noncomparative studies reported speech benefits posttreatment; however, studies primarily discussed modalities, with outcomes including safety or feasibility, rather than speech. We included English-language studies, and few studies addressed longer- term speech, social, or feeding outcomes; nonsurgical approaches, such as complementary and alternative medicine; and outcomes beyond infancy, when speech or social concerns may arise. CONCLUSIONS: Data are currently insufficient for assessing the effects of frenotomy on nonbreastfeeding outcomes that may be associated with ankyloglossia.

aDepartment of Otolaryngology, Vanderbilt University Medical Center, Nashville, Tennessee; and bCenter for Surgical Quality and Outcomes Research, cInstitute for Medicine and Public Health, Evidence-based Practice Center, dDepartments of Psychiatry, eGeneral Pediatrics, fOphthalmology and Visual Sciences, and gHealth Policy, Vanderbilt University School of Medicine, Nashville, Tennessee

Dr McPheeters conceptualized and designed the study, and drafted the initial manuscript; Drs Francis, Epstein, Morad, Chinnadurai, and Kohanim carried out the initial analyses, and reviewed and revised the manuscript; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work. www.pediatrics.org/cgi/doi/10.1542/peds.2015-0660 DOI: 10.1542/peds.2015-0660 Accepted for publication Mar 9, 2015 Address correspondence to Melissa McPheeters, PhD, MPH, Vanderbilt Evidence-based Practice Center, Institute for Medicine and Public Health, Vanderbilt University Medical Center, Nashville, TN 37203-1738. E-mail: [email protected] PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275). Copyright © 2015 by the American Academy of Pediatrics

Downloaded from www.aappublications.org/news by guest on September 25, 2021 PEDIATRICS Volume 135, number 6, June 2015 REVIEW ARTICLE Ankyloglossia is a congenital This review describes outcomes prevent, mitigate, or remedy at- condition characterized by an reported in studies identified for tributable medium- and long-term abnormally short, thickened, or tight a broader Agency for Healthcare other sequelae, including articula- lingual frenulum that restricts Research and Quality (AHRQ)- tion disorders, poor , mobility of the tongue. Ankyloglossia commissioned systematic review of oral and oropharyngeal , may be associated with other interventions for infants and children sleep-disordered , ortho- craniofacial abnormalities, but is also with congenital ankyloglossia. The dontic issues including malocclu- often an isolated anomaly.1 It can aim of this article was to investigate sion, open bite due to reverse reduce tongue mobility and has been the benefits and harms of treatment , lingual tipping of the associated with functional of infants and children who are born lower central incisors, separation limitations in breastfeeding, with ankyloglossia and who present of upper central incisors, crowding, swallowing, and articulation; some time in childhood for treatment narrow palatal arch, and dental orthodontic problems, including because of nonbreastfeeding caries? fi , open bite, and dif culties. The full review and its • What are the benefits of various separation of lower incisors; protocol are available at http://www. treatments for ankyloglossia in mechanical problems related to oral effectivehealthcare.ahrq.gov/search- children up to 18 years of age clearance; and psychological stress for-guides-reviews-and-reports/ intended to prevent or address so- in affected individuals. ?pageaction=displayproduct& cial concerns related to tongue Although most ankyloglossia research productID=1991. mobility (ie, speech, oral hygiene, is focused on infant breastfeeding excessive salivation, kissing, spit- issues, concerns beyond infancy also ting while talking, and self- METHODS have been recognized, including esteem)? speech-related issues, such as Search Strategy Inclusion and exclusion criteria were difficulty with articulation, and social We searched Medline through the developed in consultation with concerns related to limited tongue PubMed interface, the Cumulative technical expert panel of clinicians mobility. There may be long-term Index of Nursing and Allied Health and researchers who treat and study feeding sequelae, unrelated to Literature, Embase (Excerpta Medica ankyloglossia. Treatment breastfeeding, such as difficulty with Database), and PsycINFO (psychology effectiveness data were extracted bottle-feeding and deglutition. and psychiatry literature), with no from comparative study designs (ie, Individuals with untreated publication date restrictions by using randomized controlled trials [RCTs], ankyloglossia may experience vocabulary terms and key terms nonrandomized trials, prospective trouble with licking foods, such as related to ankyloglossia and its or retrospective cohort studies). ice cream, kissing, , playing therapies. Reference lists of all Harms data were collected wind instruments, oral hygiene, and included articles and recent reviews comprehensively from all study licking the . Self-esteem or related to ankyloglossia therapies types, including case series and case psychological issues also may be were hand-searched to identify any reports. Each study was reviewed a concern for affected older patients. additional relevant articles. independently by 2 investigators However, the absence of data on the against inclusion criteria (Table 1) natural history of untreated Study Selection with adjudication by a senior ankyloglossia creates uncertainty. We sought studies to answer the investigator as needed. Some propose that a short frenulum following key questions: will elongate spontaneously with Data Extraction • fi progressive stretching and thinning What are the bene ts of various Two investigators independently of the frenulum with age and use, treatments in newborns, infants, extracted data about study design; and thus no treatment is necessary.1 and children with ankyloglossia descriptions of the study populations, However, there are no prospective intended to prevent, mitigate, or interventions, and comparison longitudinal data to support this remedy attributable medium- and groups; and baseline and outcomes assertion. Absence of evidence long-term feeding sequelae, in- data (including harms/adverse makes it difficult to objectively cluding trouble with bottle-feeding, events) by using standardized forms. fi inform parents about the long-term spilling and dribbling, dif culty Principal outcomes of interest for this implications of ankyloglossia, which moving food boluses in the mouth, analysis were feeding issues, complicates the decision-making and deglutition? including dribbling and choking; process, and guidance to date has • What are the benefits of various speech outcomes, including focused exclusively on breastfeeding treatments in infants and children articulation; and social satisfaction. issues. with ankyloglossia intended to Outcomes related to breastfeeding

Downloaded from www.aappublications.org/news by guest on September 25, 2021 e1468 CHINNADURAI et al TABLE 1 Inclusion and Exclusion Criteria social outcomes; there were no – Category Criteria studies of nonsurgical treatments.3 17 Study population Inclusion: Children ages 0–18 with ankyloglossia or Speech Outcomes ankyloglossia with concomitant tight labial frenulum (-tie) Exclusion: Studies with participants with Van der Woude After breastfeeding, speech concerns syndrome, Pierre Robin syndrome, Down syndrome, or were the second most prevalent craniofacial abnormalities were excluded, as were studies of 19 outcome described in the premature infants (,37 wk of gestation ) fi Publication languages Inclusion: English ankyloglossia literature. The speci c Exclusion: Non-English outcomes measured varied among Admissible evidence (study design Included study designs these studies, but were generally and other criteria) RCTs, prospective and retrospective cohort studies, non- related to assessments of RCTs, prospective and retrospective case series, and articulation and intelligibility. crossover studies Case reports to assess harms A speech-language pathologist Other criteria measured speech outcomes in 2 Original research studies providing sufficient detail studies7,12 with the third using regarding methods and results to enable use and parent report.4 Although we looked aggregation of the data and results for them, no studies included data Studies must address 1 or more of the following: • Surgical interventions (simple anterior frenotomy, laser related to sleep-disordered frenulectomy, posterior frenulectomy, Z-plasty repair) breathing, dental issues, or • Nonsurgical treatments include complementary and dysphagia in the nonbreastfeeding alternative medicine (CAM) therapies (eg, craniosacral child. Two cohort studies attempted therapy, myofascial release, and other chiropractic to assess the effectiveness of therapies), lactation intervention, speech therapy, 4,7 physical therapy, oral motor therapy, and stretching frenotomy, and1RCTcompared2 12 /therapy surgical approaches to frenotomy. • Baseline and outcome data (including harms) related to The 1 poor-quality RCT reported interventions for ankyloglossia Relevant outcomes must be able to be extracted from data in improved articulation in patients the papers treated with 4-flap Z-frenuloplasty Data must be presented in the aggregate (versus individual compared with horizontal-to-vertical participant data) frenuloplasty.12 This study did not compare results to no treatment. Both cohort studies were of poor quality, are presented in the full report and in were based on 5 domains: study and both4,7 reported an improvement a separate article. limitations, consistency in direction of in articulation and intelligibility with effect, directness in measuring fi Study Quality Assessment ankyloglossia treatment, but bene ts intended outcomes, precision of in word, sentence, and fluent speech Two team members independently effect, and reporting bias. (Table 2). were not demonstrated. Numerous conducted risk-of-bias assessment of Strength of evidence was determined noncomparative studies reported each study by using forms developed for major intervention-outcome pairs a speech benefit after treating fi by the review team based on the using a prespeci ed approach ankyloglossia; however, these studies 3 Evidence-based Practice Center (EPC) described in the full review. primarily discussed modalities, with methods guidance with input from Data Synthesis safety and feasibility as the main content experts. Discrepancies were outcome, rather than measures of adjudicated through discussion The paucity of research and speech itself.5,6,8,10,13,14,16,18 Given between the assessors to reach heterogeneity obviated the ability to the lack of good-quality studies and consensus or via a senior reviewer. perform any meta-analyses. limitations in the measurement of Results of assessments were then Therefore, characteristics of the study outcomes, the strength of the translated to the AHRQ quality populations and interventions were evidence for the effect of surgical “ ” designation standards of good, summarized and descriptive statistics interventions to improve speech and “ ” “ ” fair, and poor. used to report study outcomes. articulation is insufficient. Strength of evidence of current research was assessed using methods RESULTS Feeding Outcomes established in the AHRQ Effective In all, 15 studies addressed the Three studies provided data Health Care Program’s Methods Guide benefits of treatment intended to specifically on feeding processes for Effectiveness and Comparative improve nonbreastfeeding outcomes other than breastfeeding. One was Effectiveness Reviews.2 Assessments (Fig 1), including feeding, speech, and an RCT11 (fair quality), 1 was a

Downloaded from www.aappublications.org/news by guest on September 25, 2021 PEDIATRICS Volume 135, number 6, June 2015 e1469 TABLE 2 Strength of Evidence Grades and Definitions from lactation consultants (n = 29; Grade Definition control group). Outcomes were based High We are very confident that the estimate of effect lies close to the true effect for this solely on maternal report within 48 outcome. The body of evidence has few or no deficiencies. We believe that the hours of randomization. However, the findings are stable (ie, another study would not change the conclusions). control group was offered, and most Moderate We are moderately confident that the estimate of effect lies close to the true effect for elected to receive, frenotomy within fi this outcome. The body of evidence has some de ciencies. We believe that the that time frame, thus eliminating the fi ndings are likely to be stable, but some doubt remains. “ Low We have limited confidence that the estimate of effect lies close to the true effect for ability to assess medium- to long- this outcome. The body of evidence has major or numerous deficiencies (or both). term” comparative feeding outcomes. We believe that additional evidence is needed before concluding either that the Nonetheless, among pretreatment findings are stable or that the estimate of effect is close to the true effect. bottle-fed infants, 76% had major fi fi Insuf cient We have no evidence, we are unable to estimate an effect, or we have no con dence in problems with dribbling, and 71% the estimate of effect for this outcome. No evidence is available or the body of “ ” evidence has unacceptable deficiencies, precluding reaching a conclusion. had excess wind (gas). Mothers reported significant improvement in Excerpted from Berkman et al 2014.20 feeding in all 8 who received the poor-quality retrospective cohort were included in 2 studies.4,11 In frenotomy and in none who did not. study,4 and the remaining study was summary, the RCT11 randomized The interval to ascertainment of fi a case series.9 All studies were single- infants born with ankyloglossia and outcomes was not speci cally center or single-surgeon studies. Two diagnosed within the first 5 months reported, but outcomes were obtained fi studies were conducted in the United with breastfeeding or bottle-feeding within the rst 4 weeks of life. Kingdom9,11 and 1 study in the problems to either frenotomy (n = The retrospective cohort study United States.4 Comparative data 28) or intensive advice and support compared parent-reported outcomes

FIGURE 1 Disposition of articles identified by the search strategy. aIncludes data from 1 unpublished thesis. bNumbers do not tally, as studies could be excluded for multiple reasons.

Downloaded from www.aappublications.org/news by guest on September 25, 2021 e1470 CHINNADURAI et al at age 3 years for children born in untreated participants. The feeding, and social benefits to the 2010 who (1) received frenotomy for intermediate outcome of improved child. These perpetuated beliefs are tongue-tie (n = 71; frenotomy group), tongue movement or mobility after based on a small body of literature, (2) were offered but declined ankyloglossia repair was assessed in few of which use a comparison group. frenotomy for tongue-tie (n = 15; no- 2 comparative studies: 1 poor-quality The scientific evidence is therefore frenotomy group), and (3) children RCT7,12 and 1 poor-quality cohort insufficient to inform patients, their without ankyloglossia (n = 18; control study.7 The RCT assessed tongue families, and clinicians about the group).4 Three questions rated on mobility by using 2 different surgical potential benefits or harms of a 5-point Likert scale were used to techniques for treating ankyloglossia ankyloglossia treatment when assess a child’s difficulty (1) cleaning and found that both approaches considered for reasons other than his or her teeth with the tongue, (2) significantly improved tongue breastfeeding difficulties. licking the outside of his or her lips, mobility, but that Z-frenuloplasty was Specifically, our review identified and (3) ice cream. For all superior in terms of a measure of a total of 4 comparative studies that questions, the frenotomy group articulation problems.7,12 In the evaluated outcomes other than performed better than the no- cohort study, individuals with breastfeeding (ie, feeding, speech, frenotomy group at age 3 years and untreated ankyloglossia had the social concerns) and none was of good did not differ significantly from the worst tongue mobility followed in quality. Anecdotally, clinicians on our comparison group without order by children with treated research team, as well as key ankyloglossia. P values were ankyloglossia, and those with no informants, report that patients presented without reporting the history of ankyloglossia at present with speech difficulty and central tendency (eg, median, mean) approximately age 6.7 with social issues, such as discomfort or variance (interquartile range, SD) With only 1 poor-quality comparative from which they were calculated. with kissing, but those clinical study, strength of the evidence scenarios are currently unrepresented Therefore, further comparative related to the ability of treatment of description or analysis was not in the effectiveness literature. As such, ankyloglossia to alleviate social there is a lack of evidence base to possible. fi concerns is currently insuf cient. counsel patients and their families on Bottle-feeding and ability to use the Also, with only 3 comparative studies what medium- and long-term tongue to eat ice cream and clean the with small sizes and limitations in the outcomes to expect from surgical or mouth (ie, oral hygiene) improved measurement of outcomes related to other forms of intervention. more in treatment groups in the 2 tongue mobility, we considered the comparative studies that considered strength of the evidence for the effect Only 1 study with comparative poor- these end points,4,11 none of which of surgical interventions to improve quality retrospective cohort data was of good quality. Supplementary the short-term outcome of mobility to addressed feeding issues other than 4 ’ bottle-feedings decreased over time in be insufficient. breastfeeding. The study s the case series.9 With only 2 intervention group received Harms comparative studies, both with frenotomy for ankyloglossia, which fi fi significant methodological limitations, Our assessment of harms is described was identi ed within the rst month including heterogeneous populations in the full report and the other article, of life, and was compared with and measured outcomes, existing data but harms were generally mild to mother-child dyads that were also are insufficient to draw conclusions nonexistent, and included bleeding offered, but declined, frenotomy for about the benefits of surgical that resolved quickly, as would be the same indication in the same time interventions on medium- and long- expected. No harms of treatment period. Although this is a common term or nonbreastfeeding feeding associated with older children’s care decisional dilemma for parents of (ie, bottle) outcomes for infants and were reported. infants with congenital ankyloglossia, children with ankyloglossia. in usual clinical care, surgical intervention is typically not Social Outcomes DISCUSSION considered unless congenital Only 1 poor-quality comparative, Ankyloglossia studies have ankyloglossia co-occurs with retrospective cohort study assessed burgeoned largely secondary to the breastfeeding concerns. Thus, outcomes related to social concerns observed benefit of breastfeeding on treatment of ankyloglossia for other other than speech.4 It reported infant health. However, some types of other feeding difficulties is significantly improved ability to clean practitioners and family also think poorly studied and understood, as the teeth with tongue, licking outside of that frenotomy treatment of preponderance of related literature lips, and eating ice cream in the ankyloglossia with frenotomy has focuses on mitigating those issues treatment group compared with medium- to long-term speech, related to breastfeeding problems.

Downloaded from www.aappublications.org/news by guest on September 25, 2021 PEDIATRICS Volume 135, number 6, June 2015 e1471 Studies providing data on speech group.7 It was performed at a tertiary lack of comparative data, against outcomes were all rated as poor care facility in an Israeli urban center. a backdrop of inadequate natural quality and included an RCT7,12 and 2 Unfortunately, its applicability is history data related to ankyloglossia. retrospective cohort studies.4,7 The limited for reasons similar to those Clinicians and policymakers lack the RCT compared 2 different previously described except that requisite information to properly frenuloplasty approaches for speech-language pathologists counsel parents and children about treatment of children of a mean age of objectively assessed speech by using the benefits and harms of ∼6 years with a tight frenulum a standardized assessment tool. Both ankyloglossia treatment as it pertains effecting articulation or retrospective studies lacked to outcomes other than breastfeeding. intelligibility12 and found that explanations about the rationale for In practice, uncertainty in evidence children treated with either 4-flap initial surgical intervention or reason can be associated with parochialism Z-frenuloplasty or horizontal-to-vertical the parent chose not to intervene and practice variation. With frenuloplasty had significant (eg, tongue-tie severity, breastfeeding a growing emphasis in evidence- improvement in articulation as difficulties). based practice across medical judged by trained speech-language The population studied to evaluate specialties, future research should fi pathologists. This bene t, however, the benefit of ankyloglossia repair for characterize which, if any, tongue-tied fl did not translate into improved uent social concerns included children and infants or children need surgical speech scores. Applicability and adults with wide variation in ages. intervention. It is also notable that fi generalizability of these ndings is Studies were rated as poor quality, despite use of alternative nonsurgical limited because of the small sample were retrospective, and few in treatments in practice, including size, the inadequate characterization number. Outcomes in 1 study were craniosacral therapy and lactation of candidate children, and because assessed by parental report and consultation, no comparative studies specialist pediatric craniofacial subject to recall bias4 and social in the literature assessed their surgeons performed these outcomes assessed were limited to effectiveness. at an urban tertiary care center. licking lips, cleaning teeth with Study Limitations Similarly, cohort studies were tongue, and eating ice cream. The performed solely in urban tertiary care social concerns or implications of This review included only studies centers. One assessed speech outcomes these issues are unclear. No other published in English. Our scan and on 3-year-old children treated for comparative study considered social review of non-English references ankyloglossia as neonates compared concerns. In addition, at least 2 case revealed a high percentage of with those who had untreated series did consider the impact of noneligible items. Given the high ankyloglossia, and a control group ankyloglossia on kissing and playing percentage of noneligible items in without a history of ankyloglossia.4 a wind instrument13 and drooling and this scan (97%), we feel that Pediatric otolaryngologists made the oral hygiene.8 excluding non-English studies did fi not introduce significant bias into diagnosis using prespeci ed diagnostic Limiting these findings was the thereview.Thereviewfocusedon criteria. However, the reason that absence of preprocedure status of comparative studies (studies infants presented for treatment of these patients in these domains and including an intervention and ankyloglossia was not described. how each was assessed. In addition a comparison group). However, we Further limiting its applicability is that to not including a comparison group did identify case series data to outcomes were assessed by using of any type, case series are strongly determine whether it could provide a nonvalidated parent-reported affected by selection bias and are, by support for the comparative findings, telephone survey. Thus, there was no nature, not comparative studies. and to identify potential harms of objective speech evaluation. There is Moreover, patients were selected intervention. a high risk for ascertainment bias; that either by retrospective chart review is, parents of children with or as they presented to Research Gaps ankyloglossia would have a higher otolaryngology clinics. Only surgical index of concern for speech issues than interventions were studied and no Because there are so few available those whose children never had been 2 studies measured the same data on other feeding outcomes, the diagnosed with tongue mobility outcomes. In most, social concerns entire research question related to restriction. The second poor-quality were measured as a secondary feeding outcomes other than retrospective cohort had a relatively outcome. breastfeeding represents a gap and smallsamplesize(n = 23) of children a potential area for future research. ∼6 years of age who were divided into Limitations of Evidence Base Similarly, substantially more research those with treated ankyloglossia, There are glaring deficiencies in this is needed to consider whether untreated ankyloglossia, and a control evidence base. The most salient is treatment of ankyloglossia in infancy

Downloaded from www.aappublications.org/news by guest on September 25, 2021 e1472 CHINNADURAI et al prevents future production parameters. Standardization would and concerns persist into childhood difficulties, as well as whether allow for comparability between related to feeding, speech, and social treatment later in life with frenotomy studies and could allow potential outcomes among children with leads to improvement when speech study data aggregation and meta- ankyloglossia, evidence is sparse on problems exist. To conduct this analysis in future systematic reviews. management of the condition. Very research effectively, methods for Social concerns are difficult to little is known about whether evaluating risk and presence of measure objectively, so there will ankyloglossia treatment, particularly speech production difficulties will likely always be a subjective frenotomy, is associated with positive need to be standardized, and component to measuring these types changes in these nonbreastfeeding outcomes agreed on. Furthermore, an of outcomes. Larger studies that outcomes. understanding of the natural history assess both treated and untreated of speech concerns in children with individuals could provide useful data ACKNOWLEDGMENTS ankyloglossia is of paramount to minimize the potential bias found Ms Tanya Surawicz contributed to importance and is currently lacking, in the existing literature. Similarly, thedatacollectionand as are comparative studies that use future research in objective interpretation. Ms Annette Williams standardized measurement tools for measurement tools, or validated self- and Ms Nila Sathe designed the data speech outcomes. report tools, is needed. collection instruments and Finally, no standard definitions of coordinated and supervised data tongue mobility or established norms CONCLUSIONS collection. Ms Katherine Worley for mobility exist, and further Although individuals and clinicians assisted in the preparation of the research is needed to determine such report anecdotally that challenges manuscript.

FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose. FUNDING: This project was supportedd under contract 290-2012-00009-I from the Agency for Healthcare Research and Quality, US Department of Health and Human Services. The authors of this report are responsible for its content. Statements in the report should not be construed as endorsement by the Agency for Healthcare Research and Quality or the US Department of Health and Human Services. POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose. COMPANION PAPER: A companion to this article can be found on page e1458, online at www.pediatrics.org/cgi/doi/10.1542/peds.2015-0658.

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Downloaded from www.aappublications.org/news by guest on September 25, 2021 Treatment of Ankyloglossia for Reasons Other Than Breastfeeding: A Systematic Review Sivakumar Chinnadurai, David O. Francis, Richard A. Epstein, Anna Morad, Sahar Kohanim and Melissa McPheeters Pediatrics 2015;135;e1467 DOI: 10.1542/peds.2015-0660 originally published online May 4, 2015;

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