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Oral Maxillofacial Surg Clin N Am 14 (2002) 525 – 537

Secondary cleft surgery and speech


Étoile M. LeBlanc, MS, CCCa,b,c,d,*
a
Private Practice, Speech Science Centre, 75 South Broadway, 4th Floor, White Plains, NY 10601, USA
b
Craniofacial Speech Disorders Center, New York Presbyterian Hospital, 3959 Broadway, New York, NY 10032, USA
c
Babies and Children’s Hospital of New York, USA
d
Department of Plastic and Reconstructive Surgery, Albert Einstein College of Medicine, 1300 Morris Park Avenue,
Bronx, NY 10461, USA

The craniofacial-cleft surgeon searches continu- the best surgical speech outcome [26 – 32]. Divergent
ally for the combination of treatment regimens that results may lead to evolution of the field but also
secure successful aesthetics, dental-skeletal form, and lends to confusion, misdirection and subsequent mis-
speech results in patients with cleft lip and palate. management of many patients. The variability in
Recent developments in innovative surgical proce- current practices and research has been marred by
dures, such as osetodistraction, neuroradiographic methodologic flaws (Table 1). In the past 10 years,
and video imaging, and robotic and minimally inva- from 1992 to 2002, only 13 randomized clinical trial
sive surgical technology [1], have been welcomed studies were performed in clefting disorders world-
additions to the field. We anticipate how they may wide [33]. A retrospect look at the literature reveals
translate into cleft surgery protocols, yet we are still in that researchers continue to conduct and publish
pursuit of practices that will enhance speech out- research based on less than optimal scientific design
comes after primary and secondary cleft surgery. and have yet to harness the multitude of variables that
Many children have successfully developed nor- affect speech outcomes in clefting disorders.
mal intelligible speech after surgical repair; however, Acknowledging and understanding the variables
a review of the literature represents not only what we that influence speech outcomes and limitations
do know but also what must be learned to ensure in controlling these variables have a great effect on
desired speech outcomes. The reported incidence our ability as surgeons and speech pathologists to
of speech problems in cleft lip and palate after provide better management protocols that lead to
primary repair ranges from 5% to 89% depending optimized speech.
on the criteria applied [2 – 10]. This variability in
results leaves levels of confidence of effect and out-
come modest at best. The literature also is replete with Variable I. The speech matrix
conflicting reports on which surgical procedures are
the most effective in primary palatoplasty [11 – 13], at The processes and mechanisms that function to
which age surgery provides the desired speech out- carry out speech are virtually multifactorial, with
come [14 – 17], what effects maxillary advancement numerous variables directly and indirectly affecting
and distraction have on resonance [18 – 25], and its integrity (Table 2).
which secondary pharyngoplasty procedure provides Should any one determinant of the speech matrix
become inoperative, other morphologic components
(epigenetically determined and nondeterministic pro-
cesses) have the capacity to compensate and provide
* Speech Science Center, 75 South Broadway, 4th Floor, obligatory responses temporally and operationally,
White Plains, NY 10601, USA as the demands require [34]. They can provide an al-
E-mail address: craniofacial@msn.com ternative means in an attempt to achieve more or less
(E.M. LeBlanc). the same developmental and functional result, al-

1042-3699/02/$ – see front matter D 2002, Elsevier Science (USA). All rights reserved.
PII: S 1 0 4 2 - 3 6 9 9 ( 0 2 ) 0 0 0 4 1 - 9
526 E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537

Table 1
Methodical flaws in research of speech outcomes in cleft lip and palate
Methodologic design Speech parameters Surgical parameters
Lack of interexaminer and Need for operational definitions Age of surgery
intraexaminer reliability
Small number of subjects Lack of objective speech data Type of surgery
lack of randomized sampling Comparison of speech results Age at assessment
Large propensity of across wide age ranges Multiple surgeons
retrospective studies Inconsistent methods of Differing surgical modifications
Decreased use of matched defining VPI in surgical procedure
control group studies Lack of objective measures of Do not operationally define criteria
Reliance on one-time velopharyngeal function of acceptability
assessment accepted Lack of consistency in preoperative Differing criteria on successful
Lack of consistency in and postoperative assessment tools surgical results
subject pools Do not operationally define what
Uneven subject distribution speech criteria should be accepted
across independent variables Differing criteria on successful
Lack of double blind studies speech results
Limited number of randomized
clinical trials

though perhaps with some degree of anatomic and Each of these systems may interact with one another
physiologic change in function. and many other abnormal speech processes (second-
The capability to speak originates from a genomic ary speech disorders such as developmental articu-
makeup, anatomic structure, and internal and external lation errors, neurologically based articulation
functional demands. Facets that maintain the drive to disorders), which results in greatly compromised
speak include environmental and communicative speech and a complicated paradigm for the craniofa-
needs (ie, communicative intent, social needs, expec- cial-cleft surgeon.
tations). Conversely, anatomic limitations and envi- Compensatory articulation disorder secondary to
ronmental facets (ie, educational services, quality of VPI is the typical articulation disorder associated with
rehabilitative services, poor social esteem, lack of cleft palate. It involves production of sounds in an
familial support) have the potential to discourage what aberrant location of the vocal tract (posterior oral
level of speech integrity is possible in an individual. cavity, hypopharyngeal and supralaryngeal area).
Speech represents the internal functional demand Sound production in this disorder reinforces abnormal
on the craniofacial scaffold and its soft tissue envel- placement and inappropriate manipulation of airflow
ope. If velopharyngeal, laryngeal, palatal, maxillary, and air pressure, which results in less-than-optimal
or mandibular segments are missing, unstable, or in velar and lateral pharyngeal wall movement of the
poor anatomic relationship with one another, as typi- velopharynx. It is possible that VPI is, in part, related
cally presented in clefting, the functions of breathing, and exacerbated by the presence of compensatory
swallowing, and speaking can be impaired [35]. Un- articulation disorders.
derstanding how speech manifests in an individual Velopharyngeal insufficiency may be, in part,
with a cleft lip and palate requires understanding the ‘‘articulatory based.’’ The child ‘‘learns’’ that velo-
dynamic speech matrix. pharyngeal movement is not necessary. The use of the
Speech is made up of the systems of resonance, velopharyngeal mechanism may be avoided, although
articulation, and voice. The ‘‘potential’’ primary closure may be anatomically possible in these pa-
speech disorders associated with cleft lip and palate tients. The child also may use other anatomic struc-
are hypernasality (resonance disorder), compensatory tures to facilitate velopharyngeal closure, such as
articulation disorder secondary to velopharyngeal lingual, pharyngeal muscular, and epiglottic valving
insufficiency (VPI), and obligatory articulation or den- [36,37]. These maladaptive gestures directly affect
tal adaptations (articulation disorders). The underlying perceived resonance quality. Phoneme-specific VPI
cause of obligatory articulation errors is related to den- is another example in which hypernasality is per-
tal and skeletal anomalies, whereas hypernasality and a ceived and velopharyngeal dysfunction is docu-
compensatory articulation disorder are directly related mented; however, it is purely an articulation disorder
to dysfunction of the velopharyngeal mechanism. and, if assessed correctly, can be treated successfully
E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537 527

Table 2
Factors affecting speech outcomes in cleft lip and palate
Genetic Anatomy/Physiology Environmental Speech Surgical
DNA integrity Cleft type Quality of Appropriate Age of surgery
Chromosomal Severity and extent of educational services development Type of procedure
integrity original defect Cultural diversity Timely Experience of surgeon
Type of underlying Family and peer support preventative services Integrity of preoperative
syndrome Family and Articulation integrity assessment
Cognitive/IQ status peer interaction Resonance integrity Presence of fistulas
Hearing status Cultural origin Type of articulation Success in primary
Language status Quality of disorder palate closure
Psychosocial status rehabilitative services Type of Use of
Velopharyngeal integrity Presence of inter- resonance disorder preorthopedic devices
Size of velopharyngeal multidisciplinary Neurologic integrity Use of nasal
gap team care Hearing integrity molding devices
Movement of velar/ Experience of Feeding/Voice integrity Postoperative healing
lateral musculature team members swallowing status and scarring
Type of velophryngeal Stimulating environment Types of objective Length of time
closure for development speech measures between surgery and
Etiology of insufficiency Age at time speech assessment
Cranial base angle of assessment Amount of short- and
Adenoid/tonsillar integrity Quality of management long-term follow-up
Dental-skeletal integrity History of therapeutic Presence of postoperative
Neurologic integrity intervention complications
Presence of other Amount of therapeutic One-stage versus
medical issues intervention two-stage procedures
Growth rates Primary or secondary repair
Orthodontic treatment
Use of nasopharyngoscopy
and procedures
Multiview videoflouroscopy

by speech therapy only. Marginal or touch closure of history, genotypic and phenotypic characteristics of
the velopharyngeal mechanism is often remediated by the syndrome. For example, a child with an isolated
nonsurgical, and/or nonprosthetic management. cleft lip and palate may potentially present with
Articulation and velopharyngeal function are varying degrees of hypernasality, varying types and
inversely related, and a craniofacial speech assess- degrees of severity of compensatory articulation sec-
ment that includes at least nasopharyngoscopy is ondary to VPI, obligatory errors, and transient com-
imperative to the decisions being made preoperatively promised hearing acuity caused by otitis media. A
and postoperatively. The act of speech requires ele- child with Treacher Collins syndrome may potentially
ments from various dimensions of the matrix. Its present with varying degrees of hyponasality accom-
dysfunction, compensatory nature, and rehabilitation panied by possible VPI secondary to asymmetry [31]
depend highly on many factors. or cleft palate, varying types and degree of severity of
obligatory errors or compensatory articulation second-
ary to VPI, and hoarseness, pitch, and loudness
Variable II. Isolated versus syndromic clefting problems (voice disorders) caused by laryngeal asym-
metry [41]. A child diagnosed with 22q11.2 micro-
Isolated clefting versus syndrome-related clefting deletion syndrome presents with hearing, cognitive,
is an important variable to take into account when language, and voice problems and increased use of
discussing speech and surgical outcomes. The attri- compensatory articulation errors (as compared to
butes of the speech disorder in isolated cleft lip and isolated cleft palate). A child diagnosed with Trisomy
palate have the potential to differ significantly as 21 however, presents with mental retardation, upper
compared to a syndrome-related cleft lip and palate airway issues, oral structural anomalies that affect
[38 – 40]. The speech matrix changes in the presence speech in a varying manners [38], and VPI related
of a syndrome, which depends highly on the natural to clefting or neurologic impairment. The surgeon
528 E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537

may increase the chances of appropriate speech and 5. Which surgical procedure is appropriate for the
surgical outcome after surgery if the child’s speech relative dimensions in the velopharynx
profile is well delineated in a manner that determines 6. Delineation of abnormal structure, such as
which component of the disordered speech is directly carotid pulsations associated with 22q11.2
related to the velopharyngeal mechanism, dental skel- microdeletion syndrome
etal anomalies, neurologic impairment, learned behav- 7. Anticipation of the affect of the intended
ior, and other nonstructural speech issues. surgical procedure on desired speech outcome.

The relationship between the surgeon and the


Variable III. The preoperative assessment craniofacial speech pathologist is pivotal for success.
The surgeon who cultivates this relationship and is
The successful surgeon understands the existence willing to take a transdisciplinary role in surgical
of the speech matrix effect and seeks out the appro- management undoubtedly increases the results of
priate speech diagnostic assessment. The use of per- desired outcome. The craniofacial-cleft palate special-
ceptual speech assessment and direct visualization of ist, whether a surgeon or a speech pathologist, cannot
the velopharyngeal mechanism by means of naso- ignore the significance and impact of a comprehensive
pharyngoscopy and multiview videofluoroscopy in- diagnostic evaluation. Many erroneous assumptions
creases the frequency of desired speech outcomes and have been made about velopharyngeal structure and
decreases the chance of postoperative complications. function and sound production based on less than
Indirect instrumentation, such as nasometry and aero- adequate diagnostic regimens, often producing less
dynamic pressure flow studies, provides objective than adequate treatment outcomes. One cannot pro-
measures on airflow and pressure; however, on their vide successful management of hypernasality without
own they provide minimal utility in diagnostic etiol- a comprehensive articulation assessment and a com-
ogy and determination of abnormal physiology of the prehensive assessment of the velopharyngeal mech-
velopharyngeal structure and do not provide distinct anism, which involves at least a nasopharyngoscopy
information on management. or multiview videofluoroscopic study.
Perceptual speech assessment should be conducted
by a well-trained craniofacial speech pathologist who
is able to decipher the speech matrix and provide the Variable IV. The effects of primary palatoplasty
surgeon with vital information in the following areas:
Secondary cleft surgery encompasses a category
1. The type and severity of the resonance dis- that is wide and varied. It includes procedures to
order present improve the appearance of the lip and nose, move
2. The type and severity of the articulation and stabilize the mandible and maxilla, and improve
disorder the function of speech (Table 3). The more severe the
3. Whether therapeutic intervention is needed initial cleft problem, the more likely the need for
4. Whether therapeutic intervention should be secondary or revisional surgery. The individual who
conducted before surgery, in combination with needs secondary cleft surgery is affected by many
surgery, or after surgery more potential variables that affect desired speech
5. The anticipated speech outcome after surgery. outcome than the person who faces initial primary
surgery (Table 4).
Direct visualization of the velopharyngeal mech- The infant who needs a primary lip or palate repair
anism should provide the surgeon with the follow- presents to the surgeon with significantly fewer
ing information: demands on the surgeon: (1) the type of unrepaired
cleft lip and palate (laterality and severity), (2) status
1. The cause of insufficiency of premaxilla and anterior palate and need for presur-
2. To what degree structure contributes to closure gical molding devices, (3) possibility of contributing
or dysfunction (ie, adenoids, tonsils, lin- phenotypic characteristics of an underlying syndrome
gual valving) (ie, airway, cardiac, gastrointestinal anomalies), and
3. To what degree movement of the velar, lateral (4) the possibility of hearing and feeding issues.
pharyngeal walls, posterior pharyngeal wall, The individual who faces secondary cleft surgery
and lingual structure is present (1) generally is much older, (2) may present with less-
4. To what degree the dysfunction noted is related than-optimal primary surgical repair with scarring and
to learned behavior dehiscence, (3) may present at a much later age than
E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537 529

Table 3
Potential speech issues after primary cleft surgery
Speech symptom
Clinical sign Resonance Articulation Voice
Nose
Severe septal deviations, Hyponasal, hypernasal No known effect No known effect
Reduced nasal patency cul de sac resonance

Lip
Tissue deficiency, poor Perceived hypernasality Weak articulatory contact, No known effect
differentiation from weak contact caused by adaptive changes
maxilla asymmetry,
short upper lip, whistle
tip deformity

Hard palate
Fistula repair Hypernasality, nasal rustling, Weak articulatory contact, No known effect
nasal emission, grimacing, middorsal palatal stops,
nasal regurgitation, lingual backing,
nasal emission obligatory errors
Alveolar cleft No known effect

Velum
Velopharyngeal Hypernasality, nasal rustling, Compensatory articulation Hoarseness,
dysfunction nasal emission, nasal turbulence low volume
Marginal closure Mild hypernasality Weak articulatory contact Hoarseness,
low volume
Learned VPI Hypernasality, nasal rustling, Compensatory articulation Hoarseness,
nasal emission, nasal turbulence low volume

surgically desired, (4) presents with dental occlusal deformities do not affect sound production or voice.
and skeletal anomalies, (5) presents with the possibil- Depending on the degree of nasal deformation, how-
ity of learned maladaptive speech behavior, (6) has ever, there could be an effect on resonance. The
other speech problems that may interact and influence individual may present with a hyponasal, hypernasal,
speech disorders related to the clefting, (7) has a or cul de sac resonance quality. Nasal tip and nostril
dysfunctional velopharyngeal mechanism or one at revision is possible in early childhood, although
high risk for dysfunction, (8) may contribute pheno- septal and bony surgery to straighten the nose totally
typic characteristics of an underlying syndrome, (9) should be postponed until full or near full growth of
possibly may have hearing, language, and cognitive the facial features has occurred. Secondary surgery,
issues, and (10) may have affected social-emotional such as straightening the nasal septum, is expected to
integrity. The surgeon who performs secondary cleft have a positive effect on the elimination of hypona-
procedures faces an increased number of concerns that sality (if septal deviation and nasal deformities are the
affect surgical outcome. primary sources of the hyponasality perceived). Tran-
sient resonance issues, such as hyponasality, may be
caused by postsurgical edema, which is expected to
Secondary procedures of the nasal deformity dissipate during the healing process.

Primary repair of a complete or incomplete cleft


lip with its associated nasal deformity presents with a Secondary procedures of lip deformity
potential effect on certain aspects of speech. In the
unilateral cleft lip, the patency of the nasal cavity may Major revisional procedures on the lip are not un-
be affected by nasal collapse, broad flaring nostrils, common. With a wide range of procedures, repair may
asymmetry, shortened columella, and a concomitant occur at almost any age. Secondary correction of the
septal deviation. These anomalies affect the patency cleft lip unilaterally or bilaterally has minimal effect
of the nasal cavity and might affect speech. Nasal on speech sound production. Primary repair of the lip
530 E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537

Table 4
Potential speech issues after secondary cleft surgery
Speech symptom
Clinical sign Resonance Articulation Voice
Lip/nose revisions No known effect No known effect No known effect

Hard Palate
Oronasal fistula repair Hypernasality, nasal Middorsal palatal stops No known effect
emission, nasal
regurgitation, foul odor
Alveolar bone graft Nasal regurgitation, Obligatory errors No known effect
foul odor

Velum
Secondary pharyngoplasty Hypernasality, nasal Compensatory articulation No known effect
emission, nasal
regurgitation, hyponasality
Le Fort I advancement Hypernasality, nasal emission Improved obligatory No known effect
Maxillary osteodistraction Transient hypernasality No known effect
Mandibular osteodistraction No known effect No known effect No known effect

has marginal effects on sound production. Resonance lip, age of the child, and how habitual the articulatory
and voice are rarely—if ever—affected directly or in- adaptation becomes may have positive effects on
directly. Clefting of the lip results in tissue deficiency, these sound errors. Superior retropositioning of the
poor differentiation from the maxilla, asymmetry, and maxilla and mandibular and maxillary advancements
retropositioning of the maxilla from the mandible. reportedly has an effect of stretching the lip and
Depending on the type of cleft (unilateral versus bi- increasing lip tension. Offering the tissue changes of
lateral), severity of the cleft, surgical procedure used, that nature allows lips to approximate in a more
and experience of the surgeon, surgical repair may normalized position for sound production [43]. The
result in scarring, infection, and dehiscence. sound errors found in clefts of the lip are considered to
A repaired short upper lip, a whistle tip deformity, be attempts to balance the relationship between a
lip incompetence, and immobility caused by scarring, functional need (sound production) and insufficient
small prolabium, and an underlying pseudo Class III soft and bony structure. Often this equalization is
malocclusion may affect production of certain sounds maladaptive in nature.
[m, p, b, f, v, w] [41,42]. The effect is often a
maladaptive change, so the intended sounds actually
may be produced with the dental edge and the lower Secondary correction of the palate
lip, which results in a [p] or [b] being perceived as
having weak articulatory contact, or ‘‘sounding soft.’’ Oronasal fistula and unrepaired alveolar clefts
Weak articulatory contact limits the amount of
increased intraoral air pressure behind the lips because An oronasal fistula is noted in 5% to 30% of all
the lips do not achieve a tight seal for the necessary primary palate repairs [44]. There are differences in
duration of time that allows for air pressure to build incidence depending on the type of palatoplasty
behind them, which provides the possible perception performed, the degree of cleft severity, and the sur-
of increased nasality. geon’s experience. In some cases the fistula is left
Production of [f, v] may be affected by a shortened intentionally, which is called initial fistulas (secondary
upper lip and may affect the phonemes because of to intravelar pharyngoplasties); in other cases, fistulas
difficulty in maintaining the correct placement of the develop because of poor healing (recurrent fistulas)
maxillary dentition on the lower lip (labiodental). [45]. It is critical to delineate between fistulas asso-
Production of [w] and some vowels may be affected ciated with unrepaired alveolar clefts and oronasal
by a tight or short upper lip. This effect may be palatal fistulas. The differentiation is important from a
marginal to the untrained ear. Growth and secondary speech perspective. Fistulas caused by an unrepaired
lip revisions of adding bulk and length to the upper alveolar cleft rarely have a direct effect on resonance,
E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537 531

articulation, or voice. Conversely, the size and extent positive effects of fistula repair on speech far out-
of the alveolar cleft may have the potential for indirect weigh the negatives.
speech effects, such as concomitant nasal emission,
weak articulatory contact, and nasal regurgitation. Velopharyngeal insufficiency
Nasal regurgitation, foul odor, and difficulty in eating
in public are often noted as secondary effects [46]. All too often the first question asked when hyper-
Oronasal fistulas have a direct effect on speech. nasality is heard concerns the integrity of the velo-
The effects of an oronasal fistula depend on the pharyngeal musculature. For successful management
location, size, and length of time during sound devel- of hypernasality, however, the first question asked
opment and length of time in which the fistula was should be how much of the nasality heard is related to
present. Hypernasality, nasal emission, and articu- VPI and how much—if at all—is it related to ‘‘learned
lation errors, such as mid-dorsal palatal stops (com- maladaptive sound compensations.’’ Many complicat-
pensatory articulation disorder secondary to VPI), ing factors contribute to the perception of hyperna-
have been associated with oronasal fistulas [39, sality. The surgeon who achieves the desired speech
47,48]. Oronasal fistulas are often the site of resistant outcome successfully (1) identifies which contrib-
sound errors, which may have direct and indirect utory speech factors exist, (2) provides a compre-
effects on velopharyngeal closure [48 – 50]. Leblanc hensive structural and physiologic assessment, (3)
and Eisig [39] noted that long-standing history of determines when to manage the patient surgically,
oronasal fistula was found to have a direct effect on and (4) develops an ‘‘haute couture’’ surgical plan
the velopharyngeal closure in patients who underwent (surgery specifically designed for the patient based on
a Le Fort I advancement. The presence and effect of in-depth preoperative assessment).
fistulas on maxillary osteodistraction are currently Management of VPI depends greatly on the trans-
unknown. Paying attention to the integrity of speech disciplinary diagnostic assessment. This assessment
sounds during the elicited speech sample (with the involves the use of direct visualization techniques,
fistula occluded) provides information on the pres- such as nasopharyngoscopy and multiview video-
ence of articulation errors and how hypernasality is fluoroscopy, measurements of airflow and air pressure
masking the extent of the sound errors as well as through the use of aerodynamic studies, and a cranio-
provides information as to the possible contributions facial speech assessment.
of velopharyngeal dysfunction on the hypernasality The debates expressed in the literature and during
being heard. professional forums that revolve around the issue of
Management of fistulas, especially oronasal fistu- which secondary surgical procedure provides the most
las, is important to undertake as soon as possible if it desired outcome are misplaced. Each procedure has
has been determined that they have detrimental effects the potential to result in positive outcomes. The debate
on speech. Management may include temporary over which procedure is most effective more appro-
occlusion by use of prosthetic obturators [41] and priately revolves around issues of postoperative man-
adhesive patches that adhere to the oral mucosa [51]. ifestations of nasal and airway obstruction. To date, the
In the case of a small fistula (no matter how affected pharyngeal flap presents with a 10% to 13% increased
the speech may be), surgical management may be risk of postoperative airway issues as compared to the
delayed until fistula repair can be performed at the sphincter pharyngoplasty [29,54]. There have been
same time as other surgical procedures to reduce the several reports of airway obstruction and death asso-
number of times one undergoes general anesthesia. ciated with posterior pharyngeal flap surgery and
Large fistulas that directly contribute to hypernasality, obstructive sleep apnea after such surgery [55].
nasal emission, articulation errors, and nasal regur- Many surgeons who use the pharyngeal flap pro-
gitation often require immediate surgery. Surgical cedure do not experience these results. Differences in
closure of oronasal fistulas is common, however its opinion still exist as to whether hyponasality beyond
success is often tenuous because of the presence of the postoperative healing process is considered to be
scar tissue, location, and size [44,52]. normal. Hyponasality does not represent normal res-
There have been few systematic investigations into onance [5,9,36,56].
the effects of the following procedures and the desired The surgical approaches currently available consist
speech outcomes: local turnover flaps, modifications of augmentation of the velum (ie, Furlow double-
of the mucoperiosteal palatal flaps, buccal flaps, and reversing Z-plasty) and altering the structure and
tongue flaps, and the use of pharyngeal flap (a sec- physiology of the pharyngeal musculature (ie, supe-
ondary pharyngoplasty procedure) in the presence of riorly based pharyngeal flaps, sphincter pharyngoplas-
posteriorly placed oronasal fistulas [26,44,53]. The ties, and augmentation pharyngoplasties).
532 E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537

Sphincter pharyngoplasty Secondary correction of the maxilla and mandible

The sphincterplasty acts on the velopharynx by Secondary alveolar bone graft


advancing the posterior pharyngeal wall (dissection of
the posterior faucial pillars, with insertion into the Alveolar bone grafts (primary and secondary) are
posterior pharyngeal wall), reducing the lateral pha- designed to stabilize the segments of the maxilla and
ryngeal wall dimensions (reducing the overall dia- replace missing bone. Alveolar bone grafting typically
meter), and providing a dynamic sphincter action occurs between 9 and 11 years of age, when the
(simulating the natural physiologic action of the velo- permanent lateral incisor or the canine tooth roots
pharynx) [29,35,57 – 59]. Riski et al [58] and Witt are approximately one-third developed. The presence
et al [59] noted that preexisting and persistent com- of appliances (rapid palatal expansion devices) before
pensatory articulation errors, low lateral flap place- bone grafting may affect articulation and resonance
ment, flap dehiscence, and reduced experience of indirectly because of the obstructive nature of the
the surgeon accounted for sphincterplasty failures in appliances affecting lingual placement (affecting spa-
the patients who did not demonstrate an improve- tial relationships) and aerodynamics of airflow and
ment in their resonance quality. Pensler et al [60] pressure for sound production. This is typically a
reported a 4% occurrence of sleep apnea. The sphinc- transient effect [64]. Before the bone graft; a child
ter pharyngoplasty has gained an ever-increasing with a cleft lip presents with dental and occlusal
attractiveness by its ease of procedure, favored anomalies that have the potential to affect speech
speech outcomes, and significantly reduced postop- (obligatory sound errors, weak articulatory contact
erative complications. of sound, possible nasal emission).

Maxillary and mandibular osteodistraction


Pharyngeal flap
Osteodistraction of the mandible has been used for
The pharyngeal flap as a procedure to eliminate
a decade, since it was first introduced in 1992 by
hypernasality has been reported in the literature with
McCarthy et al [65]. Osteodistraction is still relatively
the most vigor and frequency because of its long
new to the flora of utility research endeavors, espe-
history. It has been in use in one form or another
cially when related to speech. To date, few studies have
since 1876 [61]. The success of the pharyngeal flap
examined the effects of speech after distraction
depends on more factors to ensure resonance integrity
[21,22,66]. The current data indicate that maxillary
than that of the sphincterplasty. The pharyngeal flap
distraction of no more than 10 to 15 mm resulted in
relies in part on the mesial movement of the lateral
VPI occurring in transient manner with resolution to
pharyngeal walls, the appropriate width of the flap, the
predistraction status at least 3 to 6 months after surgery
superior placement of the flap, insertion of the free
[21]. These results are consistent with the reported
flap into the velum, the size of the lateral ports, flap
literature on Le Fort I advancements. Theoretically, the
tissue atrophy, and surgeon experience [62]. In addi-
gradual advancement of the maxilla may allow for
tion to concerns of airway obstruction after pharyn-
progressive adaptation in the velopharyngeal mech-
geal flap, questionable resonance results have raised
anism, although this has not been well researched.
concern about transient changes in facial growth
Of the scant literature and personal communica-
patterns in patients who undergo pharyngeal flaps,
tions available on speech outcomes and maxillary
particularly maxillary arch changes, mandible angle
distraction, it is believed that an advancement of
changes, and increased anterior facial height [63].
10 mm or more may result in increased velopharyn-
geal function. Satoh et al [22] noted that seven of eight
Furlow double opposing Z-plasty patients who presented with normal velopharyngeal
function and normal articulation before distraction
The Furlow-plasty has enjoyed much use and were unchanged after distraction. The average ad-
success with submucous and occult submucous cleft vancement ranged from 4.5 to 7.6 mm in 9- to
palates. It also has become increasingly popular as a 11-year-old children, with six subjects presenting with
secondary surgical procedure in the presence of status hypertrophic adenoidal tissue. The patient who pre-
postprimary palatal repair and documented VPI. The sented with borderline velopharyngeal closure before
success depends on the size of the insufficiency, the distraction exhibited increased VPI after distraction.
movement excursion of the lateral pharyngeal walls, Satoh et al [22] also noted that the ratio of velar length
and the presence of compensatory articulation errors. to pharyngeal depth decreased in all eight subjects
E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537 533

(1.06 – 1.76 before distraction and 0.79 – 1.19 after disorder secondary to VPI or secondary articulation
distraction). Guyette [21] reported on the positive disorders, such as developmental or phonologic pro-
change in sound production after distraction. This cessing feature disorder.
finding is supported by several studies on the effect
of Le Fort I advancement and articulation [24]. Velopharyngeal function
Distraction osteogenesis is a term used to denote
several different types of surgical and movement Advancement of the maxilla results in three-
techniques for moving hard and soft tissue in varying dimensional changes of the velopharynx, nasophar-
directions into more normal positions. Because of this, ynx, and hypopharynx. Le Fort I advancement has the
one is cautioned to make assumptions that the effect potential of impacting the integrity of the function of
on speech would be similar to that noted in Le Fort I the velopharyngeal mechanism by modifying its kin-
advancements. One also is reminded that the literature ematic dynamics. One area of relative agreement in
on speech effects after Le Fort I advancements con- the literature is that postoperative hypernasality is
tinues to cause debate because of numerous methodo- likely to occur in patients with maxillary advance-
logic flaws in the research. The research conducted to ments of 10 mm or more. This is a reported finding in
date gave minimal rigor to reliably discussing the one dimension only, however. Adaptive or maladap-
short- and long-term effects of maxillary distraction tive effects or no effects at all on resonance have been
on resonance and articulation. reported [20,39,56 – 74]. Schwarz et al [75] summa-
rized the following findings:
Le Fort I advancement
1. No changes in the normal oral-nasal reso-
Although maxillary distraction has gained increas- nance balance
ing popularity as the procedure to be used for maxillary 2. Changes from normal resonance to hyper-
hypoplasia and retrusion, the Le Fort I advancement nasality
continues to be used actively in many surgeons’ 3. Increase in the severity of hypernasality
protocols. The goal of maxillary repositioning with ap- 4. Decrease in the degree of severity of hyper-
propriate movement of osteotomized units includes nasality
achieving normal occlusion, normal sized and func- 5. Changes from hypernasality to hyponasality
tioning oral and pharyngeal resonance cavities, and 6. Increase in the degree of hyponasality
improved facial proportions. Orthognathic surgery re- 7. Decrease in the degree of hyponasality
quires careful coordination among the surgeon, speech 8. Elimination of hyponasality.
pathologist, and orthodontist. Much debate has oc-
curred over the Le Fort I osteotomy’s effect on speech. The following patient profile has been noted to
increase the potential for undesirable changes in
Articulation velopharyngeal function after Le Fort I advancement
[17,39]:
Crowding of the maxillary teeth, increased spaces
that result from alveolar fistulas, missing incisors, 1. Status after primary pharyngoplasty
crossbites, and pseudoprognathism in cleft lip may 2. Status after secondary pharyngoplasty (based
lead to articulation errors (obligatory disorder) known on pharyngeal flap procedure)
as distortions (frontal and lateral lisping [s, z, sh, ch, 3. Presence or history of marginal or touch
j], lingual retraction [t, d, n], and maladaptive produc- velopharyngeal closure
tion [p, b, f, v]). Dental-skeletal anomalies have 4. Status after long-standing history of oronasal
greater potential to effect sound production if they palatal fistula
exist before or during the period of sound devel- 5. Presence of or suspicion of submucous or
opment (9 months to 4 years) [56]. Correction of occult submucous cleft palate
Class III malocclusion often improves or resolves 6. Presence of hypertrophic adenoid tissue
obligatory sound errors. Published reports on advan- 7. History of hypernasality after adenoidectomy
cing the maxilla forward, with or without simulta- or tonsillectomy
neous sagittal split osteotomies, repeatedly have 8. Presence of compensatory articulation disorder
demonstrated that obligatory errors were eliminated secondary to VPI
if present in most of the reported cases [23,65,67 – 69]. 9. Presence of cranial base anomalies, such as
Le Fort I advancements have minimal to no effect Apert, Crouzon, Pfeiffer, and Treacher Collins
(negative or positive) on compensatory articulation syndromes.
534 E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537

Little attention has been paid in the literature to erations. Historically, we have learned from the
the effects of Le Fort I advancement on sphincter growth of our professions and have moved closer to
pharyngoplasty and furlow pharyngoplasty, the site of providing optimized surgical and speech outcomes for
placement (inferior versus superior) and width of the patients. We have learned that enhancing surgical
pharyngeal flap, or whether there is a concomitant procedures, technique and the learning curve do not
relapse in velopharyngeal function (with or without ensure optimized speech outcomes alone. Speech
secondary pharyngoplasty) in patients who dem- represents a dynamic matrix of elements that interact
onstrate a dental-skeletal relapse. To date, there has with each other and the surgical components. Con-
been no attempt to assess all the potential factors and trolling many of the variables to ensure an optimized
their predictive value in velopharyngeal outcome. outcome is the responsibility of the cleft team. The
One aspect is ensured: extensive preoperative per- team is ideally made up of cleft-experienced disci-
ceptual and direct visualization (via nasopharyngo- plines that work with each other for the benefit of the
scopy) is vital to the success of Le Fort I advancement. patient. The American Cleft Palate-Craniofacial Asso-
Documentation of the risk for velopharyngeal dys- ciation [78] has established minimal criteria on the
function does not change the potential undesired parameters of cleft team care.
resonance outcome. It does lend to appropriate expec- Based on what we do know at the beginning of the
tations of surgical and speech outcomes, however, twenty-first century, a comprehensive preoperative
with the realization that revision or secondary pharyn- speech assessment is fundamental to any cleft surgery.
goplasty is a possibility in 6 to 9 months after Universal standards have been recently proposed for
advancement. It is important that such patients be speech assessment in clefting [5,79], including a
closely monitored longitudinally. The most recent rigorous perceptual assessment, nasopharyngoscopy,
study by Sabry et al [76] noted a low risk of velopha- and multiview videofluoroscopy by a trained cranio-
ryngeal dysfunction in 12 patients who previously facial-cleft speech specialist.
underwent a secondary pharyngoplasty. Six of 9 sub- The type of articulation disorder presented by the
jects presented with a degree of VPI (‘‘bubbling’’) patient has a direct effect on velopharyngeal function,
before advancement. Three subjects reportedly the outcome of secondary pharyngoplasties, maxillary
improved velopharyngeal function, whereas 1 pre- distractions, and Le Fort I advancements. Compen-
sented with increased VPI, as compared to the pre- satory articulation disorder secondary to VPI—in the
operative state. The researchers failed to comment on sheer nature of the sound disorder—directly affects the
the relationship of velopharyngeal function after kinematics of velar and lateral pharyngeal wall move-
advancement and type of secondary pharyngoplasty. ment, which results in less than optimal mobility. In
A small sample and a poorly delineated mix of non- many cases, optimizing sound production before sec-
syndromic and syndromic patients make the results ondary pharyngeal cleft surgery changes the physio-
difficult to interpret. logic mechanism of the velopharynx and necessitates a
different surgical approach. If optimal speech is dif-
Oronasal fistula ficult to obtain at the time that surgery is considered,
having appropriate expectations of velopharyngeal
The presence or long-standing history of palatal function and speech postoperatively is imperative.
oronasal fistula has been noted to have an impact on This is especially important because many secondary
velopharyngeal function after maxillary advancement. pharyngoplasties result in concomitant edema, which
LeBlanc and Eisig [39] and Poole et al [77] noted that leads to hyponasality. This may have a false-positive
patients who presented with velopharyngeal dysfunc- effect, because when the transient edema resolves, one
tion after maxillary advancement exhibited the pres- may perceive hypernasality caused by the presence of
ence or history of fistula. The deleterious effect of the the type of articulation errors.
fistula on the kinematics of the velopharyngeal mech- Postprimary pharyngoplasties have the potential
anism has been supported by the work of Isberg and to place the palate and its function at risk for res-
Henningsson [34]. onance and articulation disorders. Fistulas, regarded
by many surgeons as having minimal effect on speech,
have the potential to impact articulation, velopha-
Discussion ryngeal function, eating, and the outcomes of second-
ary pharyngoplasties, maxillary advancements via
For the surgeon who performs secondary proce- osteodistraction, and Le Fort I osteotomy.
dures on clefting disorders, there is a need for careful Secondary pharyngoplasties are best used when an
balancing of occlusal, aesthetic, and speech consid- ‘‘haute couture’’ approach of assessment and surgery
E.M. LeBlanc / Oral Maxillofacial Surg Clin N Am 14 (2002) 525–537 535

approach is adopted. Surgical proficiency on the [7] Muliken JB. Final operations for the older child born
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air in children with velopharyngeal incompetence.
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