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REVIEW

CURRENT
OPINION Optimizing speech outcomes for cleft palate
Michael M. Lindeborg a, Pramila Shakya b, Shankar Man Rai b, and
David A. Shaye a

Purpose of review
Cleft lip with or without palate is one of the most common pediatric birth anomalies. Patients with cleft
palate often have speech difficulties from underlying anatomical defects that can persist after surgery. This
significantly impacts child development. There is a lack of evidence exploring, which surgical techniques
optimize speech outcomes. The purpose of this update is to report on recent literature investigating how to
optimize speech outcomes for cleft palate.
Recent findings
The two-flap palatoplasty with intravelar veloplasty (IVVP) and Furlow double-opposing Z-plasty has the strongest
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evidence for optimizing speech. One-stage palatal repair is favored at 10–14 months of age, while delays are
associated with significant speech deficits. For postoperative speech deficits, there is no significant difference between
the pharyngeal flap, sphincter pharyngoplasty, and posterior pharyngeal wall augmentation. Surgical management
should be guided by closure pattern and velopharyngeal gap but few studies stratify by these characteristics.
Summary
According to recent evidence, the two-flap palatoplasty with IVVP and Furlow palatoplasty result in the best
speech. The pharyngeal flap, sphincter pharyngoplasty, and posterior pharyngeal wall augmentation are
all viable techniques to correct residual velopharyngeal insufficiency. Future research should focus on
incorporating standardized measures and more robust study designs.
Keywords
cleft lip, cleft palate, quality of life, speech, velopharyngeal insufficiency

INTRODUCTION speech outcomes is essential for improving the stan-


Cleft lip with or without palate is the most common dard of care for cleft palate. This article provides an
congenital orofacial defect in the world, and glob- update on recent literature focused on optimizing
ally occurs once every 500–700 births [1]. A cleft speech outcomes in children with cleft palate.
palate develops when the palatal shelves fail to fuse
during the 6th–12th week of pregnancy [2]. Among
STANDARD OF CARE FOR OPTIMIZING
patients who undergo cleft palate repair, up to 5–
SPEECH AFTER CLEFT PALATE REPAIR
40% will have speech deficits because of anatomical
defects that persist after surgery [3,4]. The most Standard-of-care surgical treatment for cleft palate is
common anatomical defect impacting speech in a primary palatoplasty between 10 and 14 months of
cleft palate is velopharyngeal insufficiency (VPI), age. Repairing the cleft palate before phonemic
when the soft palate does not articulate against development supports normal speech production
the posterior pharyngeal wall [5]. This results in and minimizes compensatory articulation errors
persistent air escape from the nasal cavity and nasal [9,10]. The most common techniques include the
resonance during speech production [6,7]. VPI is two-flap palatoplasty with or without intravelar
usually present before surgery, and sometimes post-
operatively as a common complication [6]. a
Department of Otolaryngology, Massachusetts Eye & Ear, Harvard
For patients with cleft palate, speech is an integral Medical School, Boston, Massachusetts, USA and bDepartment of
component of care that directly impacts communi- Burns, Plastic and Reconstructive Surgery, Kirtipur Hospital, Nepal
cation competency, psychological well being, quality Correspondence to Michael M. Lindeborg, Department of Otolaryngol-
of life, and child development [7,8]. In recent years, ogy, Massachusetts Eye & Ear, Harvard Medical School, Boston, 02114,
there has been a greater focus on optimizing speech MA, USA. E-mail: michael_lindeborg@hms.harvard.edu
outcomes during primary palatoplasty or secondary Curr Opin Otolaryngol Head Neck Surg 2020, 28:206–211
speech surgery. Understanding how to optimize DOI:10.1097/MOO.0000000000000635

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Optimizing speech outcomes for cleft palate Lindeborg et al.

&&
speech outcomes. In 2019, Téblick et al. [17 ] per-
KEY POINTS formed a systematic review of 23 retrospective and
 Standard-of-care surgical treatment for cleft palate cohort studies, finding that the IVVP and Furlow
traditionally includes a wide range of surgical techniques were associated with improved speech
techniques; however, procedures targeting the levator outcomes compared with the two-flap palatoplasty,
muscles are needed to correct underlying V-to-Y pushback technique, and von Langenbeck
velopharyngeal insufficiency and speech deficits. palatoplasty. For example, Doucet et al. [18] showed
 The two-flap palatoplasty with intravelar veloplasty and in a cohort of 40 children that palatoplasty with
Furlow double-opposing Z-plasty at 10–14 months of IVVP had decreased rates of misarticulation (15 vs.
age have the strongest evidence for optimizing speech. 55%) and improved intelligibility (75 vs. 30%) com-
pared with two-stage repair without IVVP after
 For persistent velopharyngeal insufficiency, there are no
3 years. The Furlow palatoplasty was also shown
major differences between the pharyngeal flap,
sphincter pharyngoplasty, and posterior pharyngeal to produce better speech outcomes when compared
wall augmentation. with the von Langenbeck or two-flap palatoplasty
techniques [19–22]. Corroborating these results,
 Recent innovations in speech surgical technique include Chorney et al. [23] performed modified Furlow pal-
an autologous fat injection for pharyngeal
atoplasty in 289 patients and found that only 5%
augmentation, modified superior-based pharyngeal
flap, and modified Furlow palatoplasty. required secondary surgery for persistent VPI. When
investigating the two-flap palatoplasty, a small ret-
 Future research should incorporate standard indicators rospective cohort (n ¼ 29) by Alammar et al. [24]
for reporting baseline patient characteristics (e.g. cleft demonstrated that 21% of patients had VPI and
size, closure pattern) and speech outcomes (e.g.
20% developed fistulas. Bruneel et al. [25] published
validated speech evaluation tools, nasoendoscopy).
a 2018 study showing persistent speech deficits after
palatoplasty with IVVP; however, this case series had
a small sample size and did not directly compare
with other palatoplasty techniques. Of note, none of
veloplasty (IVVP) and the Furlow double-opposing these studies stratify by cleft size, which may nega-
Z-plasty. Others include the von Langenbeck tively bias speech results in cohorts with a higher
(bipedicled) palatoplasty, and the V-Y pushback proportion of wider clefts (>9 mm).
(Veau–Wardill–Kilner) [11]. A technique to optimize speech for wider clefts
Past research has investigated differences in is the modified Furlow palatoplasty with a unilateral
postpalatoplasty speech outcomes, but limited evi- buccal myomucosal flap [26,27]. The addition of the
dence has made it difficult to define, which tech- buccal myomucosal flap precludes the need to use
nique is best. The two-flap palatoplasty with IVVP relaxing incisions, which via scarring and growth
resulted in superior speech compared with the V-Y restriction can impact palatal length and velar mus-
pushback as it reconstructs the velar muscle group cle positioning [28]. This technique was supported
and avoids shortening of the palate [12,13]. Yama- by a large cohort study (n ¼ 505) by Mann et al. [28],
nishi et al. [14] found no significant differences in finding that the addition of the buccal myomucosal
speech outcomes between the V-Y pushback and flap for wider clefts produced similar nasal reso-
Furlow palatoplasty techniques. There is conflicting nance scores compared with the standard Furlow
evidence on whether the two-stage palatoplasty palatoplasty in narrower clefts. A bilateral buccal
compromises speech outcomes as it delays complete myomucosal flap has also aided in wide palate repair
palatal closure [12,13,15]. The V-Y pushback tech- and VPI correction, though no recent studies have
nique was found to significantly reduce hypernasal- explored their utility further [29].
ity, though was not significantly better than other With respect to staging palate repair, a 2017
VPI repair techniques [16]. systematic review of 26 studies concluded that there
Overall, there is a dearth of high-quality, compar- was not enough high-quality evidence (primarily
ative speech studies between surgical techniques. Evi- retrospective and nonrandomized studies) to con-
dence has recently emerged that sheds light onto clude whether the one-stage or two-stage palato-
which surgical techniques optimize speech outcomes. plasty was associated with better speech outcomes
for unilateral cleft lip and palate [30]. Recent evidence
favors the one-stage palatoplasy, as cleft repair pre-
PRIMARY PALATOPLASTY: RECENT cedes phonemic development [31]. Kappen et al. [32]
INNOVATIONS AND EVIDENCE established that the two-stage palatoplasty has poor
A number of recent studies have supported the use long-term speech outcomes, with high rates of hyper-
of the IVVP and Furlow palatoplasties for optimal nasality (>30%) and pharyngoplasty (40%). Nyberg

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et al. [33] and Hanai et al. [34] found that one-stage population of syndromic cleft patients (22q11.2
palatal repair leads to significant improvements in deletion), combined palatoplasty with sphincter
compensatory articulation errors, but moderate rates pharyngoplasty was found to produce similar
of persistent VPI. speech outcomes as a pharyngeal flap [48].
Though a number of studies have investigated Repeat procedures for cleft palate repair has also
when to perform palatoplasty, there has been sig- been shown to improve residual VPI. Repeat pala-
&
nificant variability in age cutoffs. Shaffer et al. [35 ] toplasty with reorientation of levator muscles was
found that a late palatoplasty (>13 months), was shown to significantly improve nasal emission,
significantly associated with speech delays when hypernasality, and articulation in patients with
compared with early (<11 months) or standard VPI and evidence of abnormal levator muscle orien-
&&
(11–13 months) timing. VPI and speech sound pro- tation [49 ]. In patients with persistent VPI after
duction were not associated with palatoplasty tim- primary palatoplasty, Wong et al. found that a mod-
& &
ing [35 ]. Pet et al. [36 ] compared internationally ified Furlow palatoplasty with pharyngeal flap was
adopted vs. nonadopted children with cleft palate, found to significantly improve perceptual speech
&
and found that a 10.4 month average delay in assessment and velar closing [50 ]. In small cohort
palatoplasty was significantly associated with mod- studies by Park et al. and Gosain et al., the double-
erate/severe VPI and secondary speech surgery. Bru- opposing Z-plasty was shown to significantly
neel et al. [37] established that delayed palatoplasty improve speech outcomes (i.e. perceptual speech
with IVVP will not correct established resonance assessment scale, resonance, nasal emission, intrao-
and articulation errors that have developed over ral pressure, and social/personal problems) after
time. previous double-opposing Z-plasty (n ¼ 14) and Fur-
Amongst patients who have submucous cleft low palatoplasty (n ¼ 15), respectively [51,52].
palates, cleft surgery was traditionally performed Pharyngeal augmentation using an autologous
only after developing hypernasality. Swanson fat injection (AFI) has gained interest as a minimally
et al. found in a retrospective cohort of over 60 invasive method for VPI treatment. A 2017 review
patients with submucous clefts that a palatoplasty by Nigh et al. [53] explored 15 studies with 251
before 4 years old is associated with less incompe- patients who underwent AFI for VPI, the majority
tent speech and persistent misarticulation [38]. of whom required a velopharyngeal gap closure of at
Overall, the majority of recently published least 50%. AFI significantly improved speech and
articles continue to be comparative cohort studies. nasalance, though cohort sizes were small, and there
Randomized controlled trials would bolster the was a lack of comparative studies [53]. An additional
palatoplasty evidence base to optimize speech out- study with a small cohort (N ¼ 21) also supported
comes. AFI, but there was no long-term speech follow-up
[54].
&
Winters et al. [55 ] recently described a novel
CLEFT SPEECH SURGERY: RECENT approach for the superior-based pharyngeal flap,
INNOVATIONS AND EVIDENCE which bisects the soft palate along the axial plane
If a patient develops VPI and fails to improve with and insets the pharyngeal flap into the soft palate.
speech therapy, the two most common procedures The authors advocate that this technique avoids soft
used to reduce nasal emissions are the pharyngeal palate midline splitting inherent to most pharyng-
flap and the sphincter pharyngoplasty. Procedure oplasty inset techniques. In a cohort of 78 patients,
choice is partially guided by underlying lateral wall there were significant improvements in speech out-
motion, but there is still mixed evidence about comes using validated assessment tools; however,
&
which produces the best speech outcomes [39– future comparative studies are needed [55 ].
&
45]. A 2018 systematic review by de Blacam et al. Denadai et al. [56 ] explored factors associated
&&
[46 ] compared the pharyngeal flap, sphincter with speech outcomes in a cohort of 167 patients
pharyngoplasty, palatoplasty, and posterior pharyn- with repaired cleft palate who underwent posterior
geal wall augmentation in 4011 patients with VPI. pharyngeal fat graft for VPI, and found that a large
There were no significant differences in speech out- velopharyngeal gap and a higher number of previ-
comes or need for further surgery, though 70.7% of ous palatal surgical procedures were associated with
patients had normal resonance and 65.3% had nor- poor speech outcomes. Interestingly, type of pri-
&&
mal nasal emissions [46 ]. Ekin et al. [47] found that mary palatoplasty and preoperative speech deficits
&
postoperative nasalance scores were significantly were not associated with speech outcomes [56 ].
lower in a cohort of 66 children after modified VPI surgical management can be partially
superior-based pharyngeal flap, regardless of the guided by the closure pattern and the size of the
closure pattern (sagittal, circular, or coronal). In a velopharyngeal gap; however, there are no past

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Optimizing speech outcomes for cleft palate Lindeborg et al.

studies that stratify by gap size and closure pattern. more research is needed to explore how speech is
Nam [6] presented a conceptual framework for influenced by these auxiliary surgeries [62].
approaching VPI surgery based on velopharyngeal
gap and closure pattern. The Furlow palatoplasty or
overlapping IVVP with oral Z-plasty may be better NONSURGICAL INTERVENTIONS AND
suited for smaller gap sizes in which there is pre- PATIENT-REPORTED OUTCOMES FOR
dominantly pharyngeal wall movement (sagittal). If SPEECH
there is predominantly soft palate movement (cor- After surgical repair, a multidisciplinary team that
onal), the sphincter pharyngoplasty or combined includes surgeons, speech and language patho-
overlapping IVVP with oral Z-plasty are recom- logists (SLPs), orthodontists, prosthodontists, and
mended. If there is combined movement of the soft audiologists become involved to assure proper
palate and pharyngeal walls (circular), then the speech development [63]. Speech evaluation
overlapping IVVP is suited for smaller gaps, and involves evaluating for inappropriate airflow using
the pharyngeal flap is best for larger gaps [6]. Over- nasoendoscopy, videofluoroscopy, nasometry, pres-
all, more research is needed that investigates speech sure-flow technique, or MRI [45]. SLPs also utilize
outcomes stratified by VPI characteristics. validated speech assessment scales to investigate
aspects of speech including misarticulation, unin-
telligibility, distortion, nasal air escapes, hyperna-
OTHER SURGICAL INTERVENTIONS sality, and glottal stop [64]. Speech therapy is a
Patients with cleft palate also undergo a number of necessary complement to surgery, as past studies
surgeries to optimize hearing, nasal function, and have shown that continued speech therapy after
maxillary development. The impact of these surger- palatoplasty can significantly reduce abnormalities
ies on speech is largely unexplored and remains a immediately present postoperatively [65]. In low-
potential avenue for future investigation. Eusta- resource settings, access to speech therapy has been
chian tube dysfunction is commonly found in expanded through task-shifting and decentralized
patients with cleft palate, and recent evidence sug- outreach programs [66–68].
gests that children with cleft palate have prolonged Patient-reported outcomes for speech have
dysfunction compared with those with cleft lip grown significantly over the past few years
&
alone [57 ]. Of note, the duration of Eustachian tube [7,69,70]. The VPI Effects on Life Outcomes (VELO)
dysfunction was not found to be associated with instrument is one example that has been validated
cleft size or the presence of a cleft-syndrome/ across multiple languages in both low-resource and
&
sequence [57 ]. In regards to its impact on speech, high-resource settings [69,71,72]. Recent efforts
Shaffer et al. [58] discovered an association between have demonstrated that the VELO can measure
hearing loss or multiple tympanostomy tubes and quality of life improvements in speech after cleft
impaired speech development in a large cohort of surgery, including those targeting VPI, such as pha-
737 children, suggesting the importance of close ryngeal flap surgery [73].
audiometry care in patients with cleft palate. Of
note, early tympanostomy tube placement before
palatoplasty did not impact speech outcomes. CONCLUSION
Children with cleft palate often have maxillary Research continues to be limited by inconsistencies
hypoplasia, and maxillary advancement procedures in postoperative speech and VPI assessment. A
may worsen speech by moving the soft palate ante- recent review by Kummer [74] investigated studies
riorly and worsening velopharyngeal sphincter clo- between 1990 and 2014 that reported speech out-
sure. Amongst a cohort of 42 patients at Texas comes for palatoplasty and velopharyngeal insuffi-
Children’s Hospital, Schultz et al. [59] found that ciency. Most studies were not blinded, had
LeFort I advancement resulted in worsening VPI in variability in speech samples used, had primarily
22% of patients. Kobayashi et al. [60] compared a perceptual ratings, and did not often evaluate velo-
small cohort of patients (n ¼ 26) who either under- pharyngeal function using objective measures.
went simultaneous orthodontic premaxillary set- Overall, this significantly limits the ability to inter-
back (OPS) and palatoplasty, OPS and palatoplasty pret studies that compare surgical speech outcomes
at separate times, or OPS with premaxillary osteot- [74]. The International Consortium for Health Out-
omy followed by palatoplasty, and found no signifi- come Measurement published outcomes measures
cant differences in speech. Dentino et al. [61] found for cleft lip/palate in 2016 and represents one pos-
success using a superiorly based pharyngeal flap to sibility for standardizing measures and best practices
improve VPI that developed after maxillary [75]. Future work should incorporate standardized
advancement in children with cleft palate. Overall, measures for speech and VPI, produce higher quality

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