Khe Hở Môi Và Vòm Miệng: Chẩn Đoán Và Quản Lý Điều Trị (Cleft Lip and Palate)

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Part VIII
Facial Growth: Time Is the Patient’s Ally

441
S. Berkowitz (ed.), Cleft Lip and Palate,
DOI 10.1007/978-3-642-30770-6_19, © Springer-Verlag Berlin Heidelberg 2013
19
19.1
Presurgical Treatment
Approaches Used to Manage
the Prominent Premaxilla
Management of the prominent premaxilla in
bilateral cleft lip and palate remains challenging,
and consequently many different approaches
have been tried.
Surgical setback at time of lip repair is one
approach that provides immediate resolution of
the excessive prominence. However, follow-up
studies have demonstrated severe deleterious
effects on midface growth resulting from this
treatment (Bishara and Olin 1972 ; Friede and
Johanson 1974 ; Vargervik 1983 ) .
The fact that most often the midface becomes
underdeveloped over time after surgical setback is
in part due to the inherent growth characteristics
of the premaxilla in bilateral clefts ( Friede and
Pruzansky 1972 ; Latham 1973 ; Vargervik 1983 ;
Trotman and Ross 1993 ; Padwa et al. 1999 ) .
In a study from our center on 63 individuals
with complete bilateral cleft lip and palate, 12
had surgical setback of the premaxilla (Vargervik
1983 ) . The surgical setback had been done before
the patient presented to our center, and details of
the procedures were not available for all of them.
It was shown that all 12 demonstrated very
de ﬁ cient midface growth that gradually became
more signi ﬁ cant with age. It was also shown that
in the other 51 subjects who had conservative
treatment of the premaxilla, the position of the
premaxilla was on the average prominent until
age 12 from which time the maxilla fell behind
compared with normative data (Fig. 19.1 ). The
study also showed that mandibular growth on an
average did not differ from that of a sample of
noncleft subjects (Fig. 19.2 ).
In most centers, surgical premaxillary setback
in the infant has been limited to extreme cases
where taping or other premaxillary manipulation
has not succeeded in reducing the premaxillary
prominence enough to allow lip closure. There
may also be circumstances where the patient
comes for treatment at a later age with an
untreated prominent or vertically overdeveloped
premaxilla where surgical repositioning cannot
be avoided (Murthy 2009 ) . A recent three-center
study reported negative effects at age 12 years of
premaxillary repositioning of the premaxilla at
the time of alveolar bone grafting at an average
age of 9.9 years (Bartzela et al. 2011 ) .
Management of the Premaxilla/
Maxilla in Bilateral Cleft Lip
and Palate
Karin Vargervik and Snehlata Oberoi
K. Vargervik , DDS () • S. Oberoi , DDS
Department of Orofacial Sciences ,
Center for Craniofacial Anomalies,
University of California, San Francisco ,
513 Parnassus Avenue ,
Box 0442 , San Francisco , CA 94143 , USA
e-mail: karin.vargervik@ucsf.edu; sneha.oberoi@ucsf.edu

442
K. Vargervik and S. Oberoi
Less drastic measures to achieve retraction of
the premaxilla prior to lip repair include the
“Latham appliance.” When successful, this may
retract the entire premaxilla to a position between
the lateral segments. This has also been proven to
have a deleterious effect on the forward growth of
the maxilla (Berkowitz et al. 2004 ) . Thus, it
appears that any approach that results in retrac-
tion of the entire premaxilla, including the
anterior nasal spine area, has the potential to con-
tribute to maxillary growth inhibition.
The currently often-advocated nasoalveolar
molding therapy may also succeed in bringing the
entire premaxilla back to a position between the
lateral segments (Grayson and Cutting 2001 ) .
This will facilitate a tension-free lip repair
and may also allow periosteoplasty if desired.
However, most centers are not advocating
bclp +
surg.
bclp (6)
bclp
contr.
Years
18
16
14
12
10
8
6
4
80
85
90
95
TM – ANS
mm
100
Fig. 19.1 A graph
depicting forward growth of
the maxilla in three groups
of individuals with bilateral
cleft lip and palate and a
group of controls (Reprinted
with permission from
Vargervik ( 1983 ) )
Years
18
16
14
12
10
8
6
4
95
105
115
125
mm
TM – PGN
135
bclp (6)
bclp
contr.
Fig. 19.2 Mandibular length
increase (Reprinted
with permission from
Vargervik ( 1983 ) )

443
19 Management of the Premaxilla/Maxilla in Bilateral Cleft Lip and Palate
periosteoplasty or bone grafting in infancy as
this also has been shown to contribute to maxil-
lary growth inhibition and to interfere with later
expansion. Many studies since the procedure was
ﬁ rst used by Skoog in 1965 have shown this.
A recent study again documented the deleterious
growth effects of periosteoplasty/bone grafting in
infancy (Hsin-Yi Hsieh et al. 2010 ) . Long-term
positive effects on growth, esthetics, and parent
satisfaction from any presurgical infant orthope-
dics have not yet been documented, according to
a recent systematic review of the literature (Uzel
and Alparslan 2011 ) .
19.2
Presurgical Management
of the Prominent Premaxilla
in Our Center
Surgeons prefer a position of the premaxilla that
allows lip repair without excessive tension. In
most cases in our center, molding of the lower
portion and tipping of the premaxilla have proven
to reduce the prominence enough to allow a good
surgical outcome. In years past, we made a small
acrylic cup ﬁ tted to the premaxilla. It had four
wires for elastic traction to buttons on a bonnet
(Fig. 19.3 ). Currently we use taping directly to the
premaxilla as we have found this to be easier for
parents to manage, and the results are similar. Lip
taping to retract a prominent premaxilla that is in
centric position is shown in Fig. 19.4 . It is impor-
tant that traction does not allow the premaxilla to
tip to one side. If the premaxilla presents asym-
metrically, this should be corrected. An example
is shown in Fig. 19.5a–e . The taping is continued
until lip surgery is done at about 10 weeks. Two to
three visits with the clinic nurse is usually needed
during the lip taping period. The purpose of the
treatment is easily understood and the procedure
easy to follow. It costs little in time and supplies,
is generally not uncomfortable, and does not
require arm restraints. If judged advantageous,
Fig. 19.3 Retraction of prominent premaxilla with appliance attached to a bonnet
Fig. 19.4 Lip taping of prominent but centrally posi-
tioned premaxilla

444
K. Vargervik and S. Oberoi
Fig. 19.5 ( a ) Newborn with asymmetric prominent premaxilla. ( b ) Bringing the premaxilla to midline with taping to
one side only. ( c ) After centralizing, symmetric retraction. ( d ) Immediately before lip repair at 10 weeks
a
b
c
d

445
19 Management of the Premaxilla/Maxilla in Bilateral Cleft Lip and Palate
nasal conformers are used in combination with
the taping (Fig. 19.6 ). If there is concern of lateral
segment collapse behind the prominent premax-
illa, a palatal plate is used with the lip taping.
19.3
Postsurgical Management
of the Premaxilla/Maxilla
in Our Center
The repaired lip will further mold the alveolar
process of the premaxilla. Due to the molding of
the alveolar process, the primary incisors will be
retroclined and may erupt into an anterior crossbite
(Fig. 19.7 ). We generally start the orthodontic
preparation for alveolar bone grafting when the
6-year molars have erupted. At that time, the
permanent incisors are erupting as well. They
will also be retroclined even before eruption and
will often erupt into an anterior crossbite. At this
time, usually around age 7, an expander is placed
to correct the position of the lateral segments.
The appliance may be ﬁ tted with labial arms with
hooks for elastics to a face mask for maxillary
protraction when needed (Fig. 19.8 ). This also
helps to correct an anterior crossbite.
Uprighting of the incisors is easily accom-
plished if the upper portion of the premaxilla
has remained prominent. A boy with repaired
complete bilateral cleft lip and palate is shown
through expansion with crossbite correction,
alignment of maxillary central incisors, bone
grafting, and lower ﬁ rst bicuspid extraction,
waiting for canine eruption to start phase II orth-
odontic treatment (Fig. 19.9a–e ). The canines
are erupting forward to contact with the central
Fig. 19.6 Premaxillary retraction and nasal conformers
Fig. 19.7 Primary dentition with slight anterior and
lateral crossbites
Fig. 19.8 Maxillary expansion with posteriorly hinged
ﬁ xed expander, labial hooks for face mask traction
e
Fig. 19.5 (continued) ( e ) Age 2 years, lip closed at
10 weeks, palate at 11 months

446
K. Vargervik and S. Oberoi
incisors and will be substituted for the missing
lateral incisors (Oberoi et al. 2009 ) .
We prefer to close the spaces for congenitally
missing lateral incisors by canine substitution if
it is possible without compromising maxillary
arch length. Three additional cases are shown
from infancy to adulthood and completion of
treatment. Figure 19.10a–f is shown to demon-
strate management of an over-extruded premax-
illa. This can be a very dif ﬁ cult problem to correct
and should be intercepted whenever possible
(Vargervik 1983 ; Meazzini et al. 2010 ) .
a
b
c
e
d
Fig. 19.9 ( a ) Mixed dentition stage, ready for maxillary
expansion and central incisor crossbite correction in prep-
aration for alveolar bone grafting. ( b ) After expansion and
bilateral bone grafting. ( c ) Lateral head ﬁ lm before incisor
uprighting. ( d ) Lateral head ﬁ lm after incisor correction.
( e ) Cone beam CT showing bone ﬁ ll in alveolar defects
and mesial eruption of canines into lateral incisor
position

447
19 Management of the Premaxilla/Maxilla in Bilateral Cleft Lip and Palate
Figure 19.11a–e demonstrates treatment com-
plicated by loss of one maxillary central incisor
and replacement on an implant (Pena et al. 2009 ).
In some cases, it is clear from an early age that
the maxilla will be hypoplastic to a degree that
will require surgical advancement of the maxilla
a
c
d
e
f
b
Fig. 19.10 ( a ) Over-extruded premaxilla being intruded
before alveolar bone grafting. ( b ) Panorex before treat-
ment start. ( c ) Lateral head ﬁ lm before treatment start.
( d ) Lateral head ﬁ lm after treatment completed. ( e ) Panorex
after treatment completed. ( f ) Final occlusion

448
K. Vargervik and S. Oberoi
a
b
c
d
e
a
Fig. 19.11 ( a ) Infant with a very small prolabium and
moderately prominent premaxilla. ( b ) Occlusion at age
6 years. ( c ) Maxillary right central incisor malformed and
in ectopic position, requiring removal. ( d ) Panorex at end
of treatment, age 18 years. Note implant for right central
incisor. Both lateral incisors congenitally missing, canine
substitution. ( e ) Occlusion at end of orthodontic
treatment

449
19 Management of the Premaxilla/Maxilla in Bilateral Cleft Lip and Palate
at growth completion (Oberoi and Vargervik 2005 ;
Oberoi et al. 2008 ) . This is most often associated
with multiple missing teeth and with cleft syn-
dromes. In those cases, it may not be advantageous
to correct an anterior crossbite or maintain space
for missing lateral incisors as the maxilla will be
advanced surgically at growth completion. Phase I
orthodontic treatment should still be done to align
the segments, followed by alveolar bone graft-
ing. This will facilitate canine eruption in a mesial
direction (Oberoi et al. 2010 ) . It will also result in
a one-piece maxilla at the time of Le Fort advance-
ment. Phase II orthodontic treatment should then be
postponed until 2–3 years before anticipated maxil-
lary surgery. Such a case is shown in Fig. 19.12a–f .
Le Fort advancement was done at age 19.
In examining records between 1970 and 1990,
we found that 13.3 % of all nonsyndromic bilateral
cleft lip and palate patients treated in that period
had maxillary advancement surgery. This com-
pares to a 25 % average reported in the literature
(Vargervik et al. 2009 ).
f
h
g
Fig. 19.11 (continued) ( f ) Occlusion after prosthetic treatment. ( g ) Lateral head ﬁ lm at end of treatment and growth.
( h ) Pro ﬁ le after completion of treatment including ﬁ nal lip and nose revision

450
K. Vargervik and S. Oberoi
a
b
c
e
f
d
Fig. 19.12 ( a ) Infant with bilateral cleft lip and palate
immediately after bilateral lip repair. ( b ) Primary denti-
tion before maxillary expansion and bone grafting.
( c ) Phase II orthodontic treatment after bone grafting with
closure of spaces for congenitally missing lateral incisors.
( d ) Lateral head ﬁ lm at completion of growth, before Le
Fort I maxillary advancement. ( e ) Pro ﬁ le before advance-
ment, age 17 years. ( f ) Lateral head ﬁ lm after maxillary
advancement at age 17½ years

451
19 Management of the Premaxilla/Maxilla in Bilateral Cleft Lip and Palate
19.4
Maxillary Growth in Repaired
Clefts
Most all individuals with a repaired unilateral or
bilateral cleft demonstrate some degree of maxil-
lary de ﬁ ciency. Growth studies have yielded
con ﬂ icting results. Some claim that cleft-related
maxillary de ﬁ ciency is an intrinsic primary defect
while others maintain that it is primarily a result of
surgical repair, resulting from lip or from palate
repair or both. This has been presented in more
detail in an earlier publication (Oberoi et al. 2008 ) .
19.4.1 Future Direction for Growth
and Treatment Studies
Superimposition of lateral cephalograms has been
the standard method for quanti ﬁ cation of changes
from treatment or by growth. Differentiating dento-
facial changes caused by treatment from those
induced by growth is not possible with either two-
dimensional or three-dimensional (3D) superimpo-
sition methods. However, comparison of treated
and untreated controls by using 3D regional super-
impositions using cone beam computed tomogra-
phy has been shown to have the potential to assess
bone displacements and bone remodeling of skel-
etal and soft tissue facial components relative to
relatively stable structures such as the cranial base.
Assessment by this method may improve our abil-
ity to quantify treatment and growth changes and
lead to better differentiation between intrinsic and
iatrogenic effects on growth and development.
References
Bartzela T, Katsaros C, Ronning E et al (2011) A longitu-
dinal three-center study of craniofacial morphology at
6 and 12 years of age in patients with complete bilat-
eral cleft lip and palate. Clin Oral Investig. doi: 10.1007/
s00784-011-0615-y
Berkowitz S et al (2004) A comparison of the effects of
the Latham-Millard procedure with those of a conser-
vative treatment approach for dental occlusion and
facial aesthetics in unilateral and bilateral complete
cleft lip and palate: part 1. Plast Reconstr Surg
113:1–18
Bishara SE, Olin WH (1972) Surgical repositioning of the
premaxilla in complete bilateral cleft lip and palate.
Angle Orthod 42:139–147
Friede H, Pruzansky S (1972) Longitudinal study of
growth in bilateral cleft lip and palate from infancy to
adolescence. Plast Reconstr Surg 49:392–403
Friede H, Johanson B (1974) A follow-up study of cleft
children treated with primary bone grafting. Scand
J Plast Reconstr Surg 8:88–103
g
h
Fig. 19.12 (continued) ( g ) Pro ﬁ le 3 years after advancement. ( h ) Final occlusion showing intact alveolus and canine
substitution, age 20½ years, 3 years postsurgical advancement

452
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Grayson BH, Cutting CB (2001) Presurgical nasoalveolar
orthopedic molding in primary correction of the nose,
lip, and alveolus of infants bone with unilateral and
bilateral clefts. Cleft Palate Craniofac J 38:193–198
Hsin-Yi Hsieh C, Wen-Ching Ko E, Kuo-Ting Chen P, Huang
C-S (2010) The effect of gingivoperiosteoplasty on facial
growth in patients with complete unilateral cleft lip and
palate. Cleft Palate Craniofac J 47:439–446
Latham RA (1973) Development and structure of the pre-
maxillary deformity in bilateral cleft lip and palate.
Brit Plast Reconstr Surg 26:1–11.
Meazzini MC, Lematti L, Mazzoleni F, Rabbiosi D,
Bozzetti A, Brusati R (2010) Vertical excess of the
premaxilla in bilateral cleft lip and palate patients: a
protocol for treatment. J Craniofac Surg 21:499–502
Murthy J (2009) Primary bilateral cleft lip repair with
management of premaxilla without preoperative ortho-
pedics. J Craniofac Surg 20:1719–1722
Oberoi S, Vargervik K (2005) Hypoplasia and hypodontia
in van der Woude syndrome. Cleft Palate Craniofac
J 42:459–466
Oberoi S, Chigurupati R, Vargervik K (2008) Morphologic
and management characteristics of individuals with
unilateral cleft lip and palate who required maxillary
advancement. Cleft Palate Craniofac J 45:42–49
Oberoi S, Chigurupati R, Gill P, Hoffman WY, Vargervik
K (2009) Volumetric assessment of secondary alveolar
bone grafting using cone beam computed tomography.
Cleft Palate Craniofac J 46:503–511
Oberoi S, Gill P, Hatcher D, Vargervik K (2010) Three-
dimensional assessment of the eruption path of the
canine in individuals with bone grafted alveolar clefts
using cone beam computed tomography. Cleft Palate
Craniofac J 47:507–512
Padwa BL, Bagheri SA, Mulliken JB (1999) Children
with repaired bilateral cleft lip/palate: effect of age at
premaxillary osteotomy on facial growth. Plast
Reconstr Surg 104:1261–1269
Pena W, Sharma A, Vargervik K et al (2009) The role of
endosseous implants in the management of alveolar
clefts. J Pediatr Dent 47:318–321
Skoog T (1965) The use of periosteal ﬂ aps in the repair of
cleft of the primary palate. Cleft Palate J 2:332–339
Trotman CA, Ross RB (1993) Craniofacial growth in
bilateral cleft lip and palate: ages six years to adult-
hood. Cleft Palate Craniofac J 30:261–273
Uzel A, Alparslan ZN (2011) Long-term effects of presur-
gical infant orthopedics in patients with cleft lip and
palate: a systematic review. Cleft Palate Craniofac
J 48:587–595
Vargervik K (1983) Growth characteristics of the premax-
illa and orthodontic treatment principles in bilateral
cleft lip and palate. Cleft Palate J 20:289–302
Vargervik K, Oberoi S, Hoffman WY (2009) Team care
for the patient with cleft: UCSF protocols and out-
comes. J Craniofac Surg 20:1668–1671

Part IX
Presurgical Orthopedics

455
S. Berkowitz (ed.), Cleft Lip and Palate,
DOI 10.1007/978-3-642-30770-6_20, © Springer-Verlag Berlin Heidelberg 2013
20
20.1
Introduction
More than 60 years after its introduction by the
Scottish prosthodontist McNeil ( 1950 ) , neonatal
maxillary orthopedics still remains a controver-
sial part of the comprehensive care for cleft lip
and palate patients. The therapy, also known as
presurgical or early orthopedic treatment, presur-
gical or infant orthopedics, early maxillary ortho-
pedics, or more recently nasoalveolar molding,
was rapidly adopted by many centers around the
world, although at that time there was no scienti ﬁ c
evidence for the claimed bene ﬁ ts nor were the
possible adverse effects properly investigated
(Pruzansky 1964 ; Prahl-Andersen and Meijer
1979 ; Witzel 1990 ) . In Europe, in the year 2000,
about 54 % of the operational centers used neo-
natal maxillary orthopedics (Shaw et al. 2000 ) .
Many different types of neonatal maxillary
orthopedics have been described in the literature.
A wide range of appliances has been designed for
this purpose with pin-retained active appliances
at one end of the spectrum (Georgiade et al. 1968 ;
Latham 1980 ) and passive appliances at the other
(Hotz and Gnoinski 1976 ) . Arbitrarily, they fall
into three main categories: active, semi-active,
and passive appliances. Active appliances are
constructed to apply a force to the maxillary seg-
ments to move them into the desired direction by
using an active force delivery system like springs
and screws. Additional anchorage can be obtained
by pins that are driven into the maxillary bone
holding the plate in position (Harkins 1960 ;
Georgiade 1964 ) . Semi-active appliances are
constructed by sectioning the dental cast and
reorienting the maxillary segments in a more
favorable position. The plate is made on the
reconstructed cast and will force the palatal seg-
ments into the predetermined direction, when
placed in the oral cavity. External strapping
across the cleft can be part of the treatment proto-
col. These are the McNeil ( 1950 ) and Burston
( 1959 ) type of appliances. Passive appliances that
are combined with extraoral strapping also ﬁ t
into this category (Huddart 1967 ) . Passive appli-
ances are supposed to induce arch alignment dur-
ing growth by grinding away material in de ﬁ nitive
areas of the plate to ensure a proper spontaneous
development of the segments. The plate is held in
position by suction and adhesion only, and no
extraoral strapping is applied. The so-called
Zürich approach as proposed by Hotz ( 1969 ) and
Neonatal Maxillary Orthopedics:
Past to Present
Anne Marie Kuijpers-Jagtman and Charlotte Prahl
A. M. Kuijpers-Jagtman , DDS, Ph.D. ()
Department of Orthodontics and Craniofacial Biology ,
Cleft Palate Craniofacial Unit, Radboud University
Nijmegen Medical Centre ,
6500 HB , Nijmegen , The Netherlands
e-mail: orthodontics@dent.umcn.nl
C. Prahl , DDS, Ph.D.
Department of Orthodontics ,
Academic Centre for Dentistry ,
Amsterdam , The Netherlands
Orthodontist Cleft Palate Team ,
Free University Medical Centre ,
Gustav Mahlerlaan 3004 ,
Amsterdam 1081 LA , The Netherlands
e-mail: c.prahl@acta.nl

456
A.M. Kuijpers-Jagtman and C. Prahl
Hotz and Gnoinski ( 1976 ) is the most well-known
representative of this kind of neonatal maxillary
orthopedics.
Over the years, neonatal maxillary orthope-
dics has given rise to emotional debates between
advocates and opponents of the procedure. At the
annual meeting of the American Cleft Palate-
Craniofacial Association held in St. Louis in
1990, the thesis “Presurgical orthopedics is by
and large a waste of time” was discussed during
the so-called Great Debate. During the 8th
International Congress on Cleft Palate and
Craniofacial Anomalies in Singapore in 1997, a
comparable debate took place, discussing the
statement “Is presurgical infant orthopedics more
a luxury than a necessity?” Before and after the
debates, the audiences voted equally in favor or
against the statements. It was considered a major
problem that hardly any sound research ﬁ ndings
were available regarding the effects of neonatal
maxillary orthopedics. And even nearly 50 years
after publication of one of the most talked about
critical reviews on presurgical orthopedics and
primary bone grafting by Samuel Pruzansky from
Chicago, what he stated still holds generally true:
“The procedures advocated might be defended
on the basis that continuous exploration for new
and better methods is warranted and deserves
support. While such research may be justi ﬁ ed,
despite the costs incurred, it is incumbent on the
investigator to document his results in a scienti ﬁ c
manner. Instead, we have been fed opinion, anec-
dotal pap, wishful thinking, and empirical trivia”
(Pruzansky 1964 ) .
20.2
Early History of Neonatal
Maxillary Orthopedics
Before the modern school of neonatal maxillary
orthopedics began, facial binding or adhesive
tape strapping was used centuries ago to narrow
the cleft before surgery (Winters and Hurwitz
1995 ) . A variation on these external bindings that
were all used to reduce the cleft width was the
method of Brophy ( 1927 ) . He wired both ends of
the cleft alveolus together in order to reduce the
cleft by tightening the wire, after which lip
surgery could be performed. All procedures,
which were at that time mainly done by prostho-
dontists or by the surgeons themselves, were
based on the never proven assumption that a nar-
row, well-aligned cleft would be easier to repair
with less undermining and less mobilization of
soft tissues. A narrower cleft would also lead to
less tension in the repaired lip. These claims have
been repeated over and over again since McNeil
started to advocate presurgical orthopedics half a
century ago (McNeil 1950, 1954, 1956 ) , although
he used another argument to perform this treat-
ment. He adopted the theory of the anatomist
Scott ( 1956, 1958 ) , who suggested that the pala-
tal segments being detached from the downward
and forward growth of the cartilaginous nasal
septum, the so-called growth center, remained
de ﬁ cient and retruded in the face. In his opinion,
this resulted in the midfacial de ﬁ ciency often
observed in cleft lip and palate patients. By mold-
ing the palatal segments into the anatomical cor-
rect position by using a series of acrylic plates,
McNeil believed that this would produce a nor-
mal maxilla while reducing the alveolar and pala-
tal cleft at the same time.
Much confusion with respect to the effect has
arisen, because the morphology of the maxilla of
newborn children with clefts varies from col-
lapsed maxillae to wide open clefts of more than
15 mm. Obviously, different treatment mechanics
have to be used in these cases. Burston ( 1959,
1965, 1971 ) advocated a passive plate to start
with to facilitate feeding and letting the child
adapt to the plate. A following plate should be
constructed by sectioning and aligning the seg-
ments of the maxilla on the plaster models before
constructing the acrylic plate. These plates were
used with external strapping. The plates were
ﬁ tted with “wings,” the chief purpose of which
was to enable the parents to insert and remove the
plates and not as stated later to aid retention.
McNeil and Burston claimed that soft tissues
overlying the hard palate were stimulated to grow
and that neonatal maxillary orthopedics could
control and modify the postnatal development of
the maxilla. These two practitioners have had a
tremendous impact on the early treatment of

457
20 Neonatal Maxillary Orthopedics: Past to Present
children with clefts and led the way for the idea
of bone grafting in conjunction with early ortho-
pedic treatment (see also Sect. 20.4.1 ).
20.3
Claimed Bene ﬁ ts of Neonatal
Maxillary Orthopedics
There has been consensus since the early 1950s
that a multidisciplinary team within a center can
best treat children with cleft lip and palate. The
team should function as an organization with a
general policy for the treatment, and each member
of the team should have insight into the different
aspects of treatment. The orthodontic discipline
has been part of such organizations from the start,
because in the majority of cases, dental arch dis-
tortion and a constricted maxilla were thought to
be the result of the surgical reconstruction of the
lip. Neonatal maxillary orthopedics aims at secur-
ing a good maxillary arch form in acceptable rela-
tionship with the mandible and to restore normal
oral function. McNeil ( 1950, 1954, 1956 ) and
Burston ( 1959, 1965, 1971 ) claimed that the
deformation seen at birth was partly due to the
original lack of tissue and a further complication
of growing tissues having become mal-related
consequent to cleft formation. McNeil ( 1954 )
stated that the most important aspect of preopera-
tive treatment of cleft palate infants was control
and correction of the lateral segments in unilateral
and bilateral conditions and that the effect of bone
stimulation plays an important role in this correc-
tion. Forces which are within the limits of biologi-
cal tolerance are said to stimulate bone growth if
they are applied to particular regions and in such
a direction that they can be regarded as intensi ﬁ ed
normal forces.
The attainment of an end-to-end position of
the alveolar processes before lip operation was
the ultimate goal for neonatal orthopedics per-
formed by all dental practitioners in the past. It
has been shown that it is possible to narrow the
cleft and to achieve an anatomical correct posi-
tion of the maxillary segments at the time of lip
surgery (McNeil 1954 ; Burston 1959 ; Rosenstein
1969 ; Robertson 1971 ; Brogan and McComb
1973 ; Huddart 1974 ; O’Donnell et al. 1974 ; Hotz
and Gnoinski 1976 ; Huddart 1979 ; Prahl et al.
2001 ; Yamada et al. 2003 ) . It also improves the
angulation of the palatal shelves to a more hori-
zontal position (Huddart 1987 ; Hochban and
Austermann 1989 ; Mishima et al. 2000 ; Mishima
et al. 2001 ) . However, the question remains of
whether these short-term effects prior to primary
surgery have any bene ﬁ cial effect on overall
treatment outcome in the long term.
Although neonatal maxillary orthopedics
originally was instituted to restore the normal
anatomy and to guide growth and development of
the maxillary segments, only after the introduc-
tion of presurgical orthopedics did the orthodon-
tic discipline try to justify early intervention for
other reasons. Prahl et al. ( 2001 ) summarized the
arguments of the proponents of the use of infant
orthopedics who state that this approach allows a
more normalized pattern of deglutition, prevents
twisting and dorsal position of the tongue in the
cleft, improves arch form and position of the alar
base, facilitates surgery, and improves outcome in
general. Other alleged bene ﬁ ts that became “en
vogue” are reduction of posterior cleft width, pre-
vention of initial collapse after surgery, prevention
of crossbites, straightening of the nasal septum,
facilitation of feeding, less danger of aspiration,
better speech development, better nose breath-
ing, better middle ear conditions, less extensive
orthodontic treatment at later ages, and a posi-
tive psychological effect on the parents (Hotz and
Gnoinski 1976, 1979 ; Dorf et al. 1985 ; Oblak
1986 ; Weil 1987 ; Gnoinski 1990 ; Ball et al. 1995 ;
Mishima et al. 1996a, b ; Kozelj 1999 ; Reid 2004 ) .
The opponents of neonatal maxillary orthopedics
stated that neonatal maxillary orthopedics is a
complex and expensive therapy that is ineffec-
tive and unnecessary (Pruzansky 1964 ; Prahl-
Andersen and Meijer 1979 ; Pruzansky and Aduss
1964 ) . Parents are obliged to travel frequently to
the treatment center and are given the burden of
compliance (Prahl et al. 2001 ) . Furthermore, it is
stated that neonatal maxillary orthopedics restricts
maxillary development (Pruzansky 1964 ; Kramer
et al. 1992 ) and in ﬂ uences speech negatively due
to delayed surgery of the hard palate inherent to
neonatal maxillary orthopedics (Bardach et al.
1984 ; Witzel et al. 1984 ) .

458
A.M. Kuijpers-Jagtman and C. Prahl
Unfortunately, we have to conclude that
despite more than half a century of research into
neonatal maxillary orthopedics, the results of
these studies remain inconclusive, mainly due to
shortcomings in the research design. Most stud-
ies were nonrandomized, retrospective in nature,
have a small sample size, and lack a (random-
ized) control group or used historical controls.
Furthermore, treatment protocols are often not
properly described, and the competence of the
professionals who performed the treatment is not
always well documented. Also, frequently, no
clear outcome measures are given, and confound-
ing variables are not taken into account.
20.4
Speci ﬁ c Types of Infant
Orthopedics
20.4.1 Kernahan Rosenstein
Procedure
An obvious problem after realignment of the
maxillary segments by either neonatal orthope-
dics or lip surgery alone is of course to hold the
realigned segments in position. For this purpose,
bone grafting, as a primary procedure at or around
the time of lip repair, was ﬁ rst utilized in the late
1950s in Europe (Nordin and Johansson 1955 ;
Schmid 1955 ) . Great emphasis was placed on the
importance of this procedure in stabilizing the
realigned maxillary segments (Robertson 1983 ) .
In 1977, Berkowitz concluded in his state-of-the
art report that the bene ﬁ ts of presurgical orthope-
dic therapy (PSOT) were not proven and that pri-
mary bone grafting was detrimental to midfacial
growth (Berkowitz 1977 ) . At that time, many cli-
nicians who previously advocated primary bone
grafting abandoned the procedure because of its
negative effect on maxillary and midfacial growth
(Jolleys and Robertson 1972 ; Robertson 1973 ;
Friede and Johanson 1974 ) . After 1975, several
additional centers con ﬁ rmed midfacial growth
inhibition and discontinued the use of primary
bone grafting (Friede and Johanson 1982 ; Pfeifer
1986 ; Reichert and Manzari 1990 ; Lilja et al.
1996 ; Smahel et al. 1998 ; Russell et al. 2011 ) .
Over the years, a few centers continued to use
neonatal maxillary orthopedics in combination
with primary bone grafting. One of these is the
Children’s Memorial Hospital Cleft Palate Clinic,
Chicago (Dado 1990 ) . The Rosenstein appliance
is a passive plate that is inserted prior to lip sur-
gery. Then, the lip is closed and the arch segments
are molded until they are in butt alignment, after
which the segments are stabilized by a small sub-
periosteal onlay rib graft. The plate is retained for
6–8 weeks postgraft, and the palate is usually
closed at or before 12 months of age (Kernahan
and Rosenstein 1990 ; Rosenstein 2003 ) . In com-
plete bilateral cleft lip and palate, the appliance
covers the lateral segments, holding them in posi-
tion while an extraoral elastic band and later on
the restored lip molds the premaxilla backwards.
Since 1965, Kernahan and Rosenstein have
used this approach, claiming that there is a princi-
pal difference with other approaches both in
sequence and technique of the procedures and that
this is critical to its success (Kernahan and
Rosenstein 1990 ) . In 2003, Rosenstein and
coworkers reported the long-term results in a sam-
ple of 135 patients, or about 50 % of the cases that
were seen in their clinic since 1965 (Rosenstein
et al. 2003 ) . In their paper, they give an overview
of the comparative studies with other centers that
have been performed. These studies show that the
growth in the Chicago sample was as good as for
any other sample treated without primary bone
grafting. However, evaluation bias cannot be ruled
out as there is an attrition rate of 50 % of the origi-
nal sample. In his multicenter study, Ross ( 1987 ) ,
and later on Trotman et al. ( 1996 ) , found less
favorable results showing that patients who under-
went primary bone grafting on average had less
protrusive maxillae than the nongrafted sample,
while the mandible compensated for this by down-
ward and backward rotation, resulting in an
increase of lower anterior facial height.
The recently published Americleft study
(Russell et al. 2011 ) throws some new light on
the results of this procedure. In this retrospective
audit of ﬁ ve North American cleft centers, there
was only one center that employed primary alve-
olar bone grafting with infant orthopedics as well
as secondary surgical revisions completed prior

459
20 Neonatal Maxillary Orthopedics: Past to Present
to 8 years of age. The outcome of this center was
less favorable than centers using much simpler
and less burdensome protocols. Although it is not
possible to detect a cause-effect relationship from
a nonrandomized retrospective study, Center B in
the Americleft study has discontinued use of pri-
mary bone grafting in its infant management pro-
tocol, due to the failure to detect any measurable
bene ﬁ ts and the possibility of detrimental effects
on maxillary development.
20.4.2 Latham-Millard Pinned
Appliance
In the Millard-Latham method of neonatal maxil-
lary orthopedics, forces are applied using a
pinned palatal appliance to manipulate mechani-
cally the maxillary segments into close approxi-
mation, followed by alveoloperiosteoplasty and
lip adhesion (Millard and Latham 1990 ) Latham
based his treatment concept on the facial growth
hypothesis of Scott ( 1956, 1958 ) as mentioned
earlier, and he encouraged Millard to use it in
complete unilateral and bilateral clefts. The aim
of the procedure is “to carry the interrupted
embryonic process to normal completion” by
maxillary alignment, stabilization of the align-
ment along with tunneling of the alveolar cleft
with periosteum, and reconstruction of the nasal
ﬂ oor to support the alar base (Millard 1994 ) .
It was many years before the ﬁ rst longer-term
studies were published about this procedure.
Berkowitz ( 1996a ) found that of a group of 32
UCLP patients at 6 years of age, 23 cases had an
anterior cross bite due to the displacement of the
premaxillary portion of the larger maxillary seg-
ment and the scar tissue resulting from the perios-
teoplasty. Comparable results were found analyzing
dental casts of 63 patients with complete clefts
showing a greater percentage of anterior crossbites
at 6 years but becoming less at 9 years of age com-
pared to a lip adhesion alone group (Millard et al.
1999 ) . Lukash et al. ( 1998 ) also found anterior and
lateral crossbites after 6 years of age in most UCLP
cases, but in BCLP cases, arch form and occlusion
as well as midfacial growth were considered
acceptable. However, they did not present data or
statistical analysis. The ﬁ ndings in BCLP patients
are in contrast to the data from Berkowitz ( 1996a,
b ) , who showed in a BCLP group of 14 patients
treated by Millard and Latham that all patients had
midfacial retrusion at 9 years of age and in 50 % of
the cases the premaxilla was retruded with the
anterior teeth in crossbite.
In 2004, Berkowitz and coworkers ( 2004 )
published an extensive report on their ﬁ ndings in
30 UCLP patients and 21 BCLP patients treated
with the Millard-Latham procedure at the South
Florida Cleft Palate Clinic. These patients were
compared to patients who were treated without
neonatal maxillary orthopedics and alveolope-
riosteoplasty. Both in complete UCLP and BCLP,
they found a higher frequency of anterior cross-
bite and (except at 3 and 12 years of age) also for
buccal crossbite compared to the treatment alter-
native without neonatal orthopedics and alveolo-
plasty. The only other long-term follow-up study
found in the literature reports more anterior open
bites and posterior crossbites in 55 UCLP and
BCLP patients treated with the procedure com-
pared to a control group without the procedure
(Henkel and Gundlach 1997 ) .
In analyzing the results of the published stud-
ies on the Latham-Millard procedure, the prob-
lem is that besides neonatal maxillary orthopedics,
infant periosteoplasty is always performed as
well as a speci ﬁ c sequence of related operations.
For example, in a study comparing two groups
that had the same Latham infant orthopedics pro-
cedure but a different timing of lip and palate
repair as well as another technique of hard palate
closure, better results were found in the group
with the modi ﬁ ed surgical protocol (Latham
2007 ) . With respect to infant perioplasty, it must
be emphasized that their technique of periosteo-
plasty is more limited with less undermining of
the periosteum on the maxilla than the original
one, described by Skoog ( 1965 ) . However, infant
periosteoplasty seems not to be widely performed
in most European centers anymore, as its expec-
tations have not been ful ﬁ lled (Hellquist 1982 ;
Hellquist et al. 1983 ; Hellquist and Svärdström
1986 ; Rintala and Ranta 1986 ; Fara et al. 1990 ;
Smahel and Müllerova 1994 ) . Nevertheless, in
the United States, the Millard-Latham method

460
A.M. Kuijpers-Jagtman and C. Prahl
continues to attract attention despite the fact that
its bene ﬁ ts are questionable (Kuijpers-Jagtman
and Long 2000 ) . Based on the long-term results
reported by Henkel and Gundlach ( 1997 ) and
Berkowitz et al. ( 2004 ) , we conclude that there is
reason to be at least suspicious of the Millard-
Latham procedure or to even abandon it.
20.4.3 The Zürich Approach
In most other clinics in Europe, neonatal maxillary
orthopedics started in Zürich in the mid-1950s
based on the treatment principles of McNeil.
After the ﬁ rst long-term evaluation, it became
clear that forced approximation of the maxil-
lary segments was not advisable. Consequently,
the procedure was greatly modi ﬁ ed to what is
known now as the “Zürich approach” (Hotz and
Gnoinski 1976 ; Hotz et al. 1986 ) . According to
Hotz and Gnoinski, the primary aim of neona-
tal orthopedics is not to facilitate surgery or to
stimulate growth as postulated by McNeil, but
to take advantage of intrinsic developmental
potentials. Since 1969/1970, early maxillary
orthopedic treatment was essential in Zürich,
while surgical intervention was postponed in
order to minimize subsequent growth distur-
bance, create optimal conditions for the max-
illary segments to develop their entire growth
potential, maintain or improve arch form, and
control effects of surgical lip closure. The appli-
ance used is a passive plate of compound soft
and hard acrylic resin, and it is worn 24 h a day
for about 16–18 months, when the soft palate is
closed surgically. The hard palate is closed after
5 years of age (Hotz 1969 ; Hotz and Gnoinski
1976 ) . During the course of treatment, the lip is
closed at about 6 months of age. The posterior
extension of the plate that extends to the uvula
must be carefully adapted to the speci ﬁ c anat-
omy of the patient. Arch alignment is achieved
by grinding away the acrylic in speci ﬁ c areas.
Figure 20.1 shows a patient who was treated
according to these principles.
According to the scienti ﬁ c use at that time,
conclusions regarding the effects of treatment
were mainly based on observations: orthopedic
guidance together with optimal timing of surgery
has bene ﬁ cial effects. Later evaluations claimed
better results with two-stage palatal closure on
speech; whether or not the presurgical treatment
had an in ﬂ uence could not be demonstrated (Hotz
and Gnoinski 1976, 1979 ; Gnoinski 1982 ; Van
Demark et al. 1989 ) . In 2009, longitudinal cepha-
lometric results were published of a sample of 29
patients with BCLP from 5 years to the end of the
growth period (Gnoinski and Rutz 2009 ) . All 29
patients had been treated in Zürich according to
the same protocol, operated on by the same sur-
geon and treated by the same orthodontist. The
authors conclude that the multidisciplinary con-
cept of maintaining the initially protrusive posi-
tion of the premaxilla by means of a passive plate
at the newborn and infant stage, as well as using
surgical procedures with limited retrusive effect,
proved to be correct in the long run: at the young
adult stage, the ANB angle remained positive for
almost all patients except for those with multiple-
tooth agenesis in the upper arch.
We can conclude that the treatment principles
of Dr. Hotz and Dr. Gnoinski have had a tremen-
dous impact on cleft palate treatment, especially in
Europe. Unfortunately, in the last two decades, not
much has been published about the results of the
Zürich approach, and the earlier papers have no
strict research design that allows us to draw evi-
dence-based conclusions about the effectiveness
of neonatal orthopedics. It might very well be pos-
sible that the surgical timing and sequencing are
the decisive factors in the ﬁ nal treatment result.
20.4.4 Nasoalveolar Molding Grayson
After nearly 40 years of neonatal maxillary
orthopedics, the treatment started to drift away
from its traditional aims and techniques with the
publications from the cleft palate team at New
York University Medical Center in the 1990s
(Grayson et al. 1993 ; Cutting et al. 1998 ) . To theirs
and many others’ experience, it is very dif ﬁ cult to
reach a good result in the neonatal treatment of
cleft lip and palate due to the actual anatomical
deformity of the nose, which includes abnormal
nasal cartilage morphology, deviated nasal septum

461
20 Neonatal Maxillary Orthopedics: Past to Present
Fig. 20.1 ( a – e ) Patient with a complete unilateral cleft
lip and palate at birth ( a ), after presurgical orthopedic
treatment according to the Zurich approach ( b ), after lip
closure ( c ), frontal view at the age of 15.8 years ( d ), and
palatal view at 15.8 years after secondary bone grafting
and orthodontic treatment ( e )
a
c
e
d
b
and columella, asymmetry of the alar base, and a
short or even absent columella, depending on the
type of cleft. In addition, ﬁ bers of the orbicularis
oris muscle run superiorly along the margins of
the cleft towards the nasal tip, while in bilateral
clefts, muscle tissue is often lacking in the prola-
bium, which is seated directly at the short colu-
mella. Therefore, Grayson and Cutting ( 2001 ) ,
who have used the Latham-Millard (POPLA)
procedure for 15 years but did not note why it
was abandoned, emphasize the importance of
presurgical correction of the nasal cartilage and
soft tissue deformity, which can be achieved by
a combination of nasal and alveolar orthopedic
molding. This therapy, now called NAM (nasoal-
veolar molding) or PNAM (presurgical nasoal-
veolar molding), makes a distinct difference with
the other types of infant orthopedics, as described

462
A.M. Kuijpers-Jagtman and C. Prahl
previously (Grayson et al. 1993 ; Cutting et al.
1998 ; Grayson and Cutting 2001 ; Barillas et al.
2009 ) . In NAM, nasal stents are added to the
alveolar molding plate. The molding plate itself
is mainly used to approximate the alveolar seg-
ments and retract the protruding premaxilla in
CBCLP in order to reduce the nasal deformity
to a degree that enables the start of more precise
nasal molding with stents (Grayson and Maull
2006 ; Santiago and Grayson 2009 ) . Retention of
the appliance in the mouth is secured by tapes
on the cheeks, which engage the intraoral plate
with orthodontic rubber bands. The appliance
aims to improve nasal tip projection, and sep-
tal and lower lateral cartilage position, before
cleft repair. In bilateral cleft lip and palate, the
nasal stents are projected to gradually lengthen
the de ﬁ cient columella. A longitudinal study in
BCLP patients on nasal morphology after NAM
and primary alveolar surgical repair (gingivope-
riosteoplasty) showed that near normal nasoalve-
olar morphology was reached until 12.5 years of
age compared to reference values from the litera-
ture (Gar ﬁ nkle et al. 2011 ) . However, this paper
does not discuss the effects on the facial pro ﬁ le
and anterior occlusion.
The use of NAM is controversial because of
reported midfacial recessiveness by other NAM
users and polarizing professionals in the ﬁ eld of
cleft lip and palate as happened before concerning
the use of regular infant orthopedics. The claimed
bene ﬁ ts of the procedure are (a) improved long-
term nasal esthetics, (b) a reduced number of nasal
surgical procedures, (c) a reduced need for second-
ary bone grafts if gingivoperiosteoplasty (GPP) is
applied, (d) no larger growth disturbance than is
found for other well-established procedures, and (e)
savings in cost. In a series of papers that are dis-
cussed in their 2001 paper, the New York group
attempted to substantiate these claims (Grayson and
Cutting 2001 ) . On the other hand, Berkowitz ( 2009 )
argues that NAM + GPP compromises future facial
growth. This was con ﬁ rmed in a retrospective study
showing that midfacial growth was affected more
negatively in a group with NAM + GPP than in a
NAM non-GPP group measured at the age of 5
(Hsieh et al. 2010 ) . Whether there was a negative
effect by NAM alone could not be ruled out.
Opponents also suggest that NAM places an extra
burden on the family that already must adapt to hav-
ing a newborn with a cleft (Sischo et al. 2012 ) .
However, as for all other types of neonatal
maxillary orthopedics, the research design for
studies into the effect of NAM was not adequate
to show scienti ﬁ c evidence for these claims so far.
In line with the DUTCHCLEFT trial as described
below, a two-group randomized controlled clini-
cal trial would be the preferred design to investi-
gate the effect of NAM before deciding whether
to accept or abandon this therapy. Santiago and
Grayson ( 2009 ) recognize this as they state that
“Although these bene ﬁ ts have been demonstrated
in multiple clinical publications there is no doubt
that a need for long-term and perhaps federally
supported clinical trials exists.” Nevertheless, in
the USA, NAM is now employed in 37 % of 117
teams that have a case load of more than 25
patients annually (Sischo et al. 2012 ) .
20.5
The DUTCHCLEFT Study
20.5.1 Background
As noted above, neonatal maxillary orthopedics
was developed and introduced on theoretical
grounds, and it became part of the treatment proto-
col in many centers although the actual effective-
ness had never been tested nor were the possible
adverse effects properly looked into. In Nijmegen,
we have performed neonatal infant orthopedics
according to the Zürich approach for over 20 years,
but our clinical experience has convinced us that in
unilateral cleft lip and palate patients, neonatal
orthopedics is at best not necessary and at its worst
could have negative side effects, even if consider-
ing only the cost-effectiveness. Gradually, we
reached equipoise regarding whether or not to per-
form neonatal infant orthopedics. Being strong
supporters of evidence-based care in general, and
for cleft lip and palate patients in particular, a ran-
domized controlled clinical trial was a next logical
and inevitable step to try to solve the controversy
regarding neonatal orthopedics.
After obtaining funding for a ﬁ rst period of 3
years, a prospective randomized clinical trial to

463
20 Neonatal Maxillary Orthopedics: Past to Present
investigate the effect of neonatal maxillary
orthopedics in children with a complete UCLP,
named “DUTCHCLEFT,” was begun in 1993 in
three academic Cleft Palate Centers in the
Netherlands: the Cleft Palate Centers of Radboud
University Nijmegen Medical Center, Free University
Medical Centre Amsterdam, and Erasmus Medical
Centre in Rotterdam.
The following outcome variables were studied:
(A) General effects: in ﬂ uence on feeding, body
length and weight gain, parents’ satisfaction
(B) Surgical and orthodontic effects: duration of
lip surgery, esthetic outcome, maxillary arch
form and dimensions, maxillofacial growth
(C) Speech and language development: prelin-
gual sound production, early speech and lan-
guage development, intelligibility
(D) Cost-effectiveness: medical and nonmedi-
cal costs
Results for feeding, general body growth, maxil-
lary arch dimensions, facial growth, nasolabial
appearance, speech and language development,
and cost-effectiveness have been published in a
series of papers between 1998 and 2009 and will
be discussed below.
20.5.2 Experimental Design
The study was set up as a prospective two-arm
randomized controlled clinical trial in three
academic cleft palate centers in the Netherlands.
The Institutional Review Boards of the three
hospitals approved the study protocol. An exten-
sive description of the design of the study is given
by Prahl et al. ( 2001 ) .
The intake started in January 1993 and ended in
June 1996. In total, 54 babies (41 boys, 13 girls)
entered the trial, 27 in each group. The patient
inclusion criteria were complete UCLP, infants
born at term, both parents Caucasian and, because
of the speech assessments, ﬂ uent in the Dutch lan-
guage, and trial entrance within 2 weeks after birth.
The exclusion criteria were other congenital mal-
formations (except for syndactyly) and soft tissue
bands. The parents of eligible infants were informed
about the trial, and written informed consent was
obtained from all participants. A computerized
balanced allocation method was used in order to
reduce imbalance on relevant prognostic factors
between the two groups. Patients were allocated
based on birth weight (<3,300 or >3,300 g) and
alveolar cleft width (<8 mm, between 8 and 12 mm,
or >12 mm). Treatment allocation was concealed.
A computer program assigned the infant to the
group with infant orthopedics (= IO + ) or without
infant orthopedics (= IO − ).
Lip surgery was performed at 18 weeks of age
according to the Millard technique. The soft palate
was closed at the age of 52 weeks according to a
modi ﬁ ed von Langenbeck procedure. Neonatal
maxillary orthopedics was performed by means of
passive plates, starting within 2 weeks after birth.
The plate was fabricated on a plaster cast and con-
sisted of compound soft and hard acrylic. The plate,
with a small extension into the cleft nose, covered
the palate and the alveolar ridges and obtruded the
cleft of the hard and soft palate. The plate was worn
24 h a day. IO + children returned to the clinic every
3 weeks to have their plates adjusted by grinding at
the cleft margins to ensure proper approximation
of the maxillary segments. Maxillary growth indi-
cated the necessity for a new plate. After surgical lip
closure, the plate was relieved in the frontal area and
reinserted the same day. Checkup visits were now
planned every 4–6 weeks. The plate was worn until
surgical closure of the soft palate. Children in the
IO − group did not wear plates. They visited the clinic
for an extra checkup at the age of 6 weeks as well as
before and after lip repair and soft palate closure.
20.5.3 General Effects
Available information in the literature of the
effects of IO on feeding and subsequently on gen-
eral body growth seemed to be inadequate, and
therefore, feeding variables and the anthropomet-
ric variables weight and length were measured in
DUTCHCLEFT. The feeding variables were
measured using questionnaires that were given to
the parents when the child was 0–2 weeks of age
and at 3, 6, 15, and 24 weeks of age. The ques-
tions concerned: amount of food, duration of the
feeding, feeding frequency, feeding velocity
(time measurement), feeding method, breast milk

464
A.M. Kuijpers-Jagtman and C. Prahl
or formula, and feeding problems. We also used
questionnaires to measure the mother’s satisfac-
tion in motherhood (Prahl et al. 2008 ) .
Mother’s satisfaction was comparable for both
groups. Only slight feeding problems were expe-
rienced, but there were no signi ﬁ cant differences
between babies who wore a plate and babies with-
out a plate. Feeding velocity increased with time
from 2.9 to 13.2 ml/min in the IO − group and from
2.6 to 13.8 ml/min in the IO + group; no signi ﬁ cant
differences were found between groups. Weight-
for-age, length-for-age, and weight-for-length
( z -scores) did not differ signi ﬁ cantly between
groups, but overall the UCLP infants in both
groups had signi ﬁ cantly lower mean z -scores for
weight-for-age and height-for-age than the refer-
ence during the ﬁ rst 14 months of life and had
lower mean values for weight-for-length after
soft palate closure (Prahl et al. 2005 ) .
The data show that neonatal orthopedics with
the aim to improve feeding and the consequent
nutritional status in infants with UCLP can be
abandoned. In addition, parents’ satisfaction
proved not to be an argument to perform neonatal
orthopedics.
20.5.4 Surgical and Orthodontic
Effects
The purpose of this part of the DUTCHCLEFT
trial was to evaluate the effect of neonatal orthope-
dics on maxillary arch form and arch dimensions
from birth on and occlusion of the deciduous den-
tition in UCLP children, to evaluate cephalomet-
ric facial growth, and to rate facial and nasolabial
appearance.
Maxillary arch dimensions were evaluated by
means of plaster casts of the upper jaw at the ages
of 0, 15, 24, 48, and 58 weeks and 1–1/2, 4, and
6 years. The maxillary casts were analyzed three-
dimensionally by means of the Re ﬂ ex Microscope
(Re ﬂ ex Measurement Ltd., Somerset, UK). In
addition, contact or collapse of the maxillary seg-
ments, at the age of 11/2 year, were scored.
Figure 20.2a , b shows a series of models from the
study. Cephalometric radiographs were analyzed
at 4 and 6 years of age. Facial appearance on full
face photographs and photographs showing only
nose and mouth was rated at different time points
until 6 years of age.
The results show that neonatal orthopedics
does reduce the alveolar cleft width before lip
surgery, which means that at the time of lip clo-
sure, the cleft width in children treated with plates
was narrower than in the other group. However,
after lip closure in both groups, the alveolar cleft
width diminished further, and at the time of soft
palate closure, the maxillary arch dimensions in
the two groups were comparable. The same holds
true for the palatal cleft width that was also
reduced during the orthopedic treatment, but after
lip closure no signi ﬁ cant differences were found
between the groups anymore. Furthermore, when
a plate was used, the palatal vault ﬂ attened. An
explanation for this ﬁ nding could be that the plate
keeps the tongue out of the cleft, allowing the
palatal shelves to ﬂ atten in the treated group.
Once the soft palate was operated, the effect of
treatment disappeared, and at 1–1/2 year of age,
the shape of the palatal vault was comparable for
both groups (Prahl et al. 2001 ) . Neonatal ortho-
pedics also did not prevent collapse of the maxil-
lary arch (Prahl et al. 2003 ) . The long-term results
until 6 years of age showed no signi ﬁ cant differ-
ences between the IO + and IO − groups for maxil-
lary arch dimensions (Bongaarts et al. 2006 ) . In
addition, the 5-year-old index (Atack et al. 1997 ) ,
which is a measure for the jaw relationship, over-
jet, overbite, presence of cross bites, and sagittal
occlusion were comparable in the two groups
(Bongaarts et al. 2004 ) .
Facial morphology at 4 and 6 years of age also
did not differ between the two groups, although it
has to be mentioned that at 6 years of age some
attrition of the original sample had taken place as
about 20 % of the patients was lost to follow-up
(Bongaarts et al. 2009 ) . At younger ages, there
was no difference in facial appearance between
the groups (Prahl et al. 2006 ) . At the age of
4 years, IO had a positive effect on full facial
appearance of UCLP children, but at the age of 6,
only professionals saw a positive effect of IO on
the nasolabial photographs. This seems to be
rather irrelevant since patients deal with laymen
in their daily life (Bongaarts et al. 2008 ) .

465
20 Neonatal Maxillary Orthopedics: Past to Present
Fig. 20.2 ( a , b ) Series of
study casts of two patients
from the DUTCHCLEFT
trial. The infant whose
models are shown in
( a ) received no infant
orthopedics; the infant
whose models are shown in
( b ) received infant
orthopedics. The ﬁ ve stages
are (from upper to lower
model) 0, 15, 24, 48, and
58 weeks of age
a
b

466
A.M. Kuijpers-Jagtman and C. Prahl
We concluded that infant orthopedics has only
a temporary effect on maxillary arch dimensions
which does not last beyond soft palate closure. It
also does not improve the jaw relationship and
occlusion in the deciduous dentition, and it has no
measurable effect on facial morphology and facial
and nasolabial appearance until 6 years of age.
Therefore, from the orthodontic and surgical point
of view, neonatal maxillary orthopedics could be
abandoned.
20.5.5 Effect on Speech
Evaluation of speech and language development
showed that at the age of 1 year, the children who
wore plates presented enhanced production of
alveolar sounds in babbling; however, at the age
of 1 and 1/2 years when the plate was no longer
used, consonant production in babbling was com-
parable in the two groups (Konst et al. 1999 ) .
Reports on the later speech and language devel-
opment of the children in the DUTCHCLEFT
study showed a limited effect on speech (Konst
et al. 2003a, b, c ) . The speech results at 2.5 years
of age showed differences in intelligibility
between the groups. In two different experiments,
untrained listeners as well as experienced speech
and language therapists gave higher intelligibility
ratings to the children who formerly used plates
(Konst et al. 2003c ) . However, data obtained by a
transcription task indicated no differences in actual
intelligibility (Konst et al. 2000 ) , but compared to
their non-cleft peers, the children with clefts were
signi ﬁ cantly less well understood.
At 2.5 years of age, the phonological develop-
ment of the IO + children was normal or delayed,
whereas most children who were not treated with
a plate had abnormal development. Half a year
later, it appeared that the IO + children had acquired
more initial consonants than the IO − group (Konst
et al. 2003b ) . In the same age groups, the IO + chil-
dren used longer sentences than the IO − children,
indicating that their grammatical development was
more advanced. At the age of 6, no differences in
expressive language skills between the two groups
were found (Konst et al. 2003a ) . Hence, neonatal
maxillary orthopedics did not have long-lasting
effects on language development.
Regarding the claimed bene ﬁ ts of neonatal
maxillary orthopedics on speech development in
unilateral cleft lip and palate children, it can be
concluded from this trial that there is a bene ﬁ cial
but rather limited effect until the age of 2 and
1/2 years. However, irrespective of neonatal max-
illary orthopedics, the speech of children with
clefts remains far behind that of their non-cleft
peers. Due to lack of funding, the speech samples
at the age of 6 years have not yet been analyzed.
20.5.6 Cost-Effectiveness
Nowadays, cost-effectiveness is an important issue,
as costs of health-care interventions are often topic
of debate. Especially in cases of reimbursement
decisions, cost-effectiveness estimates can be use-
ful. Together with the results of clinical effective-
ness studies, cost-effectiveness information can
be used to determine whether a certain treatment
should become available to patients. The main
principle of cost-effectiveness analysis is to esti-
mate the costs and the treatment outcome (= effec-
tiveness), compared to an alternative treatment
(Drummond et al. 1997 ) . Based on this explicit
comparison, the difference in costs is related to
the difference in effectiveness between alterna-
tive treatments. A prospective study in a random-
ized clinical trial design, such as DUTCHCLEFT,
in combination with an economic evaluation is
a good vehicle to analyze the cost-effectiveness
(Severens et al. 1998 ; Cunningham 2001 ) .
In DUTCHCLEFT, a cost-effectiveness analysis
was planned for all three research areas that were
mentioned above (see A, B, C). For the cost-
effectiveness analysis, a so-called societal point of
view was used, indicating that cost of treatment was
based on real prices rather than fees. Besides this, a
differential approach was used, aiming to calculate
the difference in cost between yes or no neonatal max-
illary orthopedics. A more extensive description of the
cost analysis methods used in DUTCHCLEFT can be
found in our earlier publications (Severens et al. 1998,
1999 ) , which together with the publication by
Cunningham [99] provide a good starting point to
learn more about economic health evaluation.
In a preliminary report, the short-term cost-
effectiveness of neonatal maxillary orthopedics

467
20 Neonatal Maxillary Orthopedics: Past to Present
was based on the duration of the operation for sur-
gical lip closure (Severens et al. 1998 ) . However,
this effectiveness parameter has little to do with
effectiveness in terms of clinical outcome, but at
that time, as the trial was still running, no other
effectiveness variables were available. In 2004,
we published data on cost-effectiveness of neona-
tal maxillary orthopedics compared to no such
treatment, focusing on speech outcome at 2 and
1/2 years of age (Konst et al. 2004 ) . The patients’
speech was assessed by a panel of ﬁ ve speech
therapists with experience in assessment of cleft
lip and palate speech. At the time of evaluation,
full data of both groups were not yet available. In
each group, the data of ten children could be ana-
lyzed. The cost-effectiveness analysis required
that the effects of treatment were expressed in one
general effect measure. Therefore, the listeners
were asked to rate speech quality on a 10-point
scale. A detailed description of the full perceptual
evaluation is given by Konst et al. ( 2003c ) .
The group that was treated with neonatal
orthopedics had a signi ﬁ cantly better rating for
speech (IO + : 3.52, SD 1.75; IO − : 2.18, SD 0.62).
The total costs of neonatal maxillary orthopedics
were of course higher in the treatment group. The
resulting cost-effectiveness ratio for IO + versus
IO − was EUR 1041.00 for 1.34 point of speech
improvement. Relative to the total costs spent on
the management of children with clefts, the
ﬁ nancial investment to obtain this speech
improvement is rather limited. Furthermore, the
additional costs of neonatal maxillary orthope-
dics might be partly outweighed by the costs pre-
vented for speech therapy in later years. This
needs to be investigated further when the 6-year
data of all children have been analyzed.
Conclusions
In 1991, the National Institute of Dental
Research (NIDR) sought grant applications to
conduct prospective and/or retrospective clini-
cal trials evaluating treatment procedures for
nonsyndromic, unilateral cleft lip and palate.
The background information was that treat-
ment sequence, timing, methods, and surgical
techniques were all controversial. The long-
term impact on maxillofacial growth and
speech was considered to be of primary
importance. This initiative re ﬂ ected the lack
of evidence with which treatments were car-
ried out up to this point in time. This also
holds true for neonatal infant orthopedics.
Prior to the start of the randomized clinical
trial of the effect of presurgical orthopedics in
UCLP, we attempted to perform a meta-analy-
sis of the existing literature, but this proved
not to be possible due to the lack of random-
ized clinical trials, inconsistent outcomes and
result reporting, missing data, small sample
sizes, confounders, covariates, and publica-
tion bias. This problem has not been solved
yet. A recent Cochrane review could only
identify three randomized clinical trials into
the effect of neonatal plates on feeding (Bessell
et al. 2011 ) . The available information on
bilateral cleft lip and palate patients is even
more sporadic. This was the main reason to
start the DUTCHCLEFT trial.
Based on the results of DUTCHCLEFT so
far, we conclude that neonatal maxillary ortho-
pedics in unilateral cleft lip and palate patients
as performed in this trial is not necessary for
feeding, parents’ satisfaction, or orthodontic
and surgical reasons. Regarding speech, a
positive but very limited effect was found until
the age of 2 and 1/2 years, but the speech of
the children with clefts remained far behind
that of their non-cleft peers anyway. Relative
to the total costs of the treatment of a UCLP
patient, the ﬁ nancial investment to reach this
effect was rather limited. However, it is ques-
tionable whether this limited effect is impor-
tant enough to justify neonatal orthopedics. It
should also be taken into consideration that
the 6-year results for speech due to lack of
funding have not yet been analyzed.
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DOI 10.1007/978-3-642-30770-6_21, © Springer-Verlag Berlin Heidelberg 2013
21
21.1
Dental Occlusion
One of the most widely debated areas in the
treatment of cleft lip and palate involves the use
of presurgical orthopedics and periosteoplasty
with lip adhesion (POPLA) designed by Ralph
Latham (orthodontist) and D. Ralph Millard, Jr.
(plastic surgeon) (Millard 1980, 1986 ) . They
contend the POPLA procedure is superior to a
conservative nonpresurgical orthopedic treatment
(non-POPLA) for producing more aesthetically
appealing lip/nose surgery, while still allowing
for good midfacial growth and dental occlusion
in complete unilateral cleft lip and palate
(CUCLP) and complete bilateral cleft lip and pal-
ate (CBCLP) patients. This study compares the
dental occlusion of POPLA with conservative,
nonpresurgical
orthopedic
treatment
(non-
POPLA), which Millard had been using between
1960 and 1980 (Berkowitz 1996a, b ) . POPLA
proponents and others, who may not resort to a
lip adhesion but who still use the same presurgi-
cal orthopedic procedure designed by Latham in
CBCLP patients for forcefully retracting the pro-
truding premaxilla, favor the attainment of
improved facial aesthetics and palatal arch align-
ment soon after birth with or without periosteo-
plasty. They speculate that the early aesthetic
bene ﬁ ts will remain as the face grows and devel-
ops (Millard and Latham 1990 ; Dufresne and So
1992 ; Millard et al. 1998 ; Mulliken 2001 ) .
In CUCLP and CBCLP, presurgical orthope-
dics is usually followed by periosteoplasty in the
expectation that the resulting bone bridging cre-
ated will avert the need for secondary alveolar
bone grafts at a later date.
It is not a simple or a lightly assumed task to
offer a brief challenging the rationale for presur-
gical orthopedics and periosteoplasty for infants
with complete unilateral or complete bilateral
cleft lip and palate. Advocates of the POPLA
concept, or a variant of it, are few but well
respected. Their proposed goal of making things
right and whole as early as possible seems sensi-
ble and has great emotional appeal.
This ﬁ rst of a two-part serial retrospective
dental occlusal and facial study covers more than
40 years of recording with serial dental casts
and lateral cephaloradiographs, the sequential
in ﬂ uences of both the POPLA and conservative
non-POPLA procedures on palatal development,
and the anterior and buccal dental occlusion.
Part one of this study uses serial dental casts to
determine the extent of anterior and buccal cross-
bites. Detailed dental occlusal analyses of these
A Comparison of the Effects of the
Latham–Millard POPLA Procedure
with a Conservative Treatment
Approach on Dental Occlusion and
Facial Aesthetics in CUCLP and CBCLP
Samuel Berkowitz and Martha Mejia
S. Berkowitz , DDS, M.S., FICD
Adjunct Professor, Department of Orthodontics , College
of Dentistry, University of Illinois , Chicago , IL , USA
Clinical Professor of Surgery and Pediatrics (Ret),
Director of Research (Ret), South Florida Cleft
Palate Clinic, University of Miami School of Medicine ,
Miami , FL , USA
Consultant (Ret), Craniofacial Anomalies Program ,
Miami Children’s Hospital , Miami , FL , USA
e-mail: sberk3140@aol.com
M. Mejia , DDS (*)
Division of Plastic Surgery , Miami Children’s Hospital ,
W-3100 SW 62nd Ave , Miami , FL , USA

474
S. Berkowitz and M. Mejia
records test the ef ﬁ cacy of the POPLA procedure
compared to the non-POPLA treatment employed
by Millard and Berkowitz from 1960 to 1980
prior to the POPLA treatment years.
Part two of this study will analyze facial
changes using serial lateral cephaloradiographs.
21.1.1 Method and Materials
The complete unilateral and bilateral clefts of the
lip and palate cases, treated by either presurgical
orthopedics (POPLA) or the nonpresurgical ortho-
pedics (non-POPLA) procedure, were chosen from
the ﬁ les of the longitudinal facial and palatal growth
studies of the Miami Craniofacial Anomalies
Foundation of patients from the South Florida Cleft
Palate Clinic, the University of Miami School of
Medicine. Dr. Ralph Millard, Jr., performed lip,
nose, and palatal surgery in both test samples.
Secondary alveolar bone grafts and maxillary and/
or mandibular osteotomies and maxillary distrac-
tion osteogenesis were performed by S.A. Wolfe.
Samuel Berkowitz documented growth changes
with dental casts, lateral cephaloradiographs, pan-
orexes, and photographs and performed all treat-
ment
orthodontics
other
than
presurgical
orthopedics (Millard et al. 1988, 1998 ) . Berkowitz
treated a number of children in the POPLA group
who had extensive anterior crossbites starting when
they were about 9 years of age so that beginning at
this age, there is a reduction in the frequency of the
cases with anterior crossbites.
21.1.1.1 POPLA: Presurgical
Orthopedics with Lip Adhesion
(Millard et al. 1998 ) (Fig. 21.1a , b )
Ralph Latham supervised the plastic surgery
residents in the manipulation of the palatally
pinned presurgical orthopedic appliance. Another
orthodontist later performed the same relatively
simple procedure. Because of the training and
close supervision involved in the treatments
given, there was relatively little variation in this
procedure over the years covered by the POPLA
data. In CBCLP, the appliance mechanically
expands the lateral palatal segments, allowing for
the retraction of the protruding premaxilla into
position within the alveolar arch (Figs. 21.2 , 21.3 ,
21.4 , 21.5 , and 21.6 ). In CUCLP cases, the
mechanical forces bring the premaxillary portion
of the larger segment mediopalatally, and in most
instances, the lesser segment is carried forward
2–3 mm to make contact with each other
(Fig. 21.5a , b ). Afterward, the ﬂ oor of the nose is
surgically closed, and a periosteoplasty is per-
formed to permit the migration of alveolar osteo-
blasts to bridge the alveolar gap space. After the
Fig. 21.1 ( a ) CBCLP. Latham’s
presurgical orthopedic appliance.
The elastic chain creates the
activating forces to retract the
premaxilla while expanding the
palatal segments. The posterior
segment is pinned to the palate
for approximately 2 weeks. The
premaxillary pins, which are
pulled by the elastic chain, are
positioned anterior to the
premaxillary vomerine suture.
( b ) CUCLP. Latham’s presurgical
orthopedic appliance. The screw
knob controls the movement of
the pinned appliance. The
premaxilla is bodily rotated
mediopalatally while the cleft
lesser segment is moved
anteriorly approximately 2–3 mm
to make contact with the smaller
cleft segment
a
b

475
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
a
b
Fig. 21.2 ( a – j ). ( a ) Newborn. A small asymmetric protruding premaxilla. ( b ) After premaxillary retrusion. Lip adhesion
followed by de ﬁ nitive lip surgery and gingivoperiosteoplasty (POPLA). ( c ) Midfacial de ﬁ ciency due to retruded premaxilla
c

476
S. Berkowitz and M. Mejia
f
h
i
j
g
d
e
( d ) Retruded premaxilla showing
some missing incisor teeth. ( e ) Orthodontic appliances
placed to correct anterior crossbite and missing incisor
spaces. Protraction facial mask with Class III mechanics
was unsatisfactory in correcting this problem. The use of
protraction facial mask was not successful in reducing
midfacial de ﬁ ciency due to upper and lower facial growth.
This necessitated the use of maxillary distraction osteo-
genesis ( f , h ) Osteogenic appliance. ( g ) After maxillary
advancement. ( i , j ) Following distraction osteogenesis to
advance the maxillary denture. ( i ) After protraction facial
mask – no changing. ( j ) Distraction osteogenesis to tip
occlusion – now hypernasality is present
Fig. 21.2 (continued)

477
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
Fig. 21.3 ( a , b ) Serial
cephalometric tracings showing
the stability of the midfacial
recessiveness even after the use
of a protraction facial mask.
There was no change in the
Class I buccal occlusion since
the orthopedic forces were
directed to only advance the
retruded premaxilla. ( b )
Postmaxillary distraction
osteogenesis. Due to server
hypernasality, maxilla
advancement was discontinued
Fig. 21.4 ( a ) Lateral cephalometric tracing of a newborn
with a complete bilateral cleft lip and palate shows the loca-
tion of the premaxillary vomerine suture ( PVS ) posterior to
the protruding premaxilla. ( b ) Frontal computed tomography
scan of a patient with complete bilateral cleft lip and palate
who was treated with the presurgical orthopedics, gingivope-
riosteoplasty, and lip adhesion protocol. Only the left palatal
segment is fused to the premaxilla with a bone bridge. Note
the premaxilla “telescoping” at the premaxillary vomerine
suture ( arrow ), the junction with the nasal septum
Fig. 21.5 ( a ) Palatal radiograph of a patient with com-
plete bilateral cleft lip and palate who was conservatively
treated at birth with a head bonnet with an elastic strip
over the protruding premaxilla. The premaxillary vom-
erine suture was still open years later. ( b ) Palatal radio-
graph after premaxillary retraction with a Latham
appliance shows a synostosis of the premaxillary vom-
erine suture

478
S. Berkowitz and M. Mejia
premaxillary retraction, a lip adhesion is per-
formed followed 6–8 months later by de ﬁ nitive
lip surgery with “forked” ﬂ aps.
21.1.1.2 Non-POPLA Conservative
Treatment of CBCLP and CUCLP
The protruding premaxilla in CBCLP is ventro-
ﬂ exed by the forces generated by a head bonnet
with elastic strip positioned across the prolabium,
followed by lip adhesion surgery. No attempt is
made to bodily retract the premaxilla and place
it within the alveolar arch. Palatal cleft clo-
sure using a von Langenbeck procedure with a
modi ﬁ ed vomer ﬂ ap is performed between 18 and
36 months, depending on the size of the cleft space.
In non-POPLA as well as POPLA cases, palatal
expansion is sometimes used at 5–6 years of age to
correct the buccal crossbite in both groups.
Fig. 21.6 Presurgical orthopedics,
periosteoplasty, and lip adhesion
(POPLA). The premaxilla was
retruded and positioned within the
arch. Followed by periosteoplasty,
the palatal cleft was closed at 2
1/2 years using a von Langenbeck
procedure with a modi ﬁ ed vomer
ﬂ ap 9–13 years: The use of a
protraction facial mask to advance
the premaxilla was unsuccessful.
This was followed by midfacial
distraction osteogenesis
0–3
2–0
0–5
0–7
0–11
9–5–0
10–4–0
11–0–0
12–0–0
13–0–0

479
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
Orthodontics between 8 and 10 years of age
on POPLA and non-POPLA patients is used to
align the anterior teeth. In non-POPLA cases, it
is performed prior to bilateral or unilateral sec-
ondary alveolar bone grafting. In POPLA cases,
the anterior crossbite correction with full orth-
odontic appliances and a protraction facial mask
is initialized at 8–9 years when one or both of the
permanent central incisors are erupted. An
attempt at this age is made to correct the poste-
rior occlusion. None of the test groups were in
the Class III buccal occlusion at this age.
Standard orthodontics follows to align all the
permanent teeth. In CUCLP and CBCLP, lip
adhesion is mostly performed at 3 months in
both series followed by de ﬁ nitive lip surgery at
6–8 months of age.
21.2
Discussion
Millard and Berkowitz have been associated
since 1961 with the South Florida Cleft Palate
Clinic, where presurgical orthopedic treatment
(PSOT) was not being used. Millard has always
been a strong supporter of the importance of seri-
ally documenting treatment outcomes with lat-
eral cephalometric radiographs, dental casts,
panorexes, and facial/intraoral photographs. To
that end, they have worked diligently.
After 20 years of performing nonpresurgical
orthopedics (non-PSO) prior to the utilization of
the POPLA treatment procedure, Millard pub-
lished an operational plan to investigate and
record facial–palatal changes when utilizing the
Latham procedure (Millard et al. 1988 ) . Today,
Berkowitz has assembled an extensive collec-
tion of serial records that have always been
available for review by any interested party. Kai
Henkel ( 1997 ) , a visiting professor of plastic
surgery from Rostock, Germany, after reviewing
the POPLA treatment serial case records, pub-
lished a review stating that the procedure resulted
in unsatisfactory facial aesthetics and dental
function.
After 40 years of recording facial, palatal, and
dental growth changes in POPLA and non-
POPLA cases, the authors of this brief believe
that criticism is in order for a clinical procedure
whose outcome results have now been adequately
reviewed using serial objective records.
It is unfortunate that a controversy still exists
as to the utility of POPLA or similar treatment
with or without lip adhesion 20+ years after its
introduction and after the critiques to which it
has been subjected (Berkowitz 1996a, b ) . First,
Georgiade and Latham ( 1975 ) and Latham
(Latham 1970, 1973, 1980 ) have failed to publish
any outcome studies. More recently, Millard and
Latham have published a limited outcome report
using Berkowitz’s palatal cast records (Berkowitz
1996a, b ) . Millard and Latham’s coauthors
Huifen, Spiro, and Morovic performed linear
measurements of the changing palatal size rather
than analyze the relative growth of the opposing
jaws by reviewing the dental occlusion.
Dufresne and So, in their chapter supporting
the presurgical orthopedic procedure for CBCLP
and CUCLP patients, referred only to Millard’s
original introductory statements of the POPLA
procedure in Cleft Craft (Vol. III) (Millard 1980 )
and listed no other supporting references, yet
advocate its use (Dufresne and So 1992 ) . Cutting
and Grayson (Cutting et al. 1998 ; Cutting and
Grayson 2000 ) limited their PSOT report to the
effect of periosteoplasty in successfully producing
bone bridging of the alveolar cleft, yet no
mention was made of its effect on the dental
occlusion.
Berkowitz ( 1996a, b ) , in a preliminary report,
compares Millard’s POPLA with Millard–
Berkowitz’s conservatively (non-POPLA) treated
CBCLP outcomes. That comparative outcome
study con ﬁ rmed the negative effects of POPLA
on facial aesthetics and dental occlusion. The
study presented herein expands on that report.
In 1996, Berkowitz and Latham were asked
by the American Cleft Palate Craniofacial
Association program committee to debate the
utility of this procedure at their annual meet-
ing in San Diego, California. At that meet-
ing, Berkowitz and LaRossa (plastic surgeon)
presented long-term case reports that were
critical of the procedure, but no supporting case
studies were forthcoming from Latham and
Morales (plastic surgeon).
Many clinicians who advocate the use of
Latham’s or any other presurgical orthopedics

480
S. Berkowitz and M. Mejia
(PSO) state that one of the appliance’s bene ﬁ ts is
to prevent “collapse” of the lateral palatal seg-
ments. The frequent use of the word “collapse” to
describe palatal arch relationships after neonatal
lip surgery is unfortunate and needs to be better
understood. The term is misleading for it con-
jures up an unwarranted sense of foreboding.
The word “collapse” was introduced into the
cleft-treatment lexicon in the 1960s by some sur-
geons and orthodontists to describe the palate’s
physical state after uniting of the lip and causing
medial palatal movement of overexpanded palatal
segments. They evidently did not realize that in
complete lip and palatal clefts, the resulting
medial palatal movement (molding) is bene ﬁ cial
because it reduces palatal cleft size and corrects
the overexpanded palatal segments relationship.
Serial studies have shown that overlapping palatal
segments in the deciduous and mixed dentition
are of no clinical importance.
The word “collapse” implies that this condi-
tion is bad and should be prevented. However,
after years of analyzing serial dental casts, many
orthodontists have concluded that establishing lip
muscle continuity leads to good geometric palatal
changes no matter their temporary neonatal
geometric relationship. Overlapped segments do
not impede normal palatal growth, and in most
cases, such overlapped segments can easily be
properly realigned using relatively simple ortho-
dontics. Cleft segments in posterior crossbite at
an early age are not indicative of future palatal
maldevelopment. Since the width of the cleft
space in ﬂ uences the type and timing of surgical
palatal closure, one would prefer to have a small
cleft space prior to palatal surgery to reduce the
possibility of creating growth-inhibiting scarring
while producing a normal palatal vault space.
In contrast to conservative non-POPLA cases,
POPLA-treated patients require extensive and
costly orthodontic treatment to correct the ante-
rior crossbites, regain lateral incisor spaces, and
achieve upper to lower anterior arch congruency.
Often the degree of facial and palatal distortion
is so extensive in POPLA cases that additional
surgical intervention is necessary at a later age.
Some parents have reported their children’s
experiencing psychosocial problems due to the
lack of peer acceptance of the concave facial
pro ﬁ le.
POPLA in CBCLP (Table 21.1 ): The bodily
retracted protruding premaxilla is retracted and
placed in excellent alignment within the alveolar
segments (Fig. 21.1a , b ). Since no lateral ﬂ exion
of the nasal septum is seen in cat scans, it was
concluded that the premaxilla is “telescoped”
posteriorly at the premaxillary vomerine suture
(PVS) (Fig. 21.4 ). The bending at the PVS is also
seen in POPLA-treated CUCLP (Fig. 21.5 ). The
PVS is not observed in follow-up palatal POPLA
radiographs (Fig. 21.5 ).
The diagram (Fig. 21.7 ) is designed to show
the error of palatal segmental movements in
POPLA CUCLP cases. Our 3D analysis of the
CUCLP palatal arch changes demonstrates that
the premaxillary portion of the noncleft segment
is brought mediopalatally while the smaller cleft
segment is advanced, resulting in the loss of the
lateral incisor space. This explains why an ante-
rior dental crossbite is most likely to result.
Millard ( 1980 ) has written that premaxillary
retraction and periosteoplasty might have a nega-
Table 21.1 Number of cases at each age level in presurgical and nonpresurgical orthopedics treatment groups
Approximate age of participant
3 years
6 years
9 years
12 years
Total sample
Unilateral cleft
POPLA treatment
30
43
34
18
125
Non-POPLA treatment
51
54
46
33
184
Bilateral cleft
POPLA treatment
21
20
15
9
64
Non-POPLA treatment
49
49
40
35
173
POPLA presurgical orthopedics gingivoperiosteoplasty and lip adhesion

481
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
tive effect on palatal growth, but he nevertheless
believes this trade-off is acceptable to obtain
early aesthetics, to close the ﬂ oor of the nose, and
to avoid the need for secondary alveolar bone
grafting.
The desire of some surgeons to establish a
child’s well-balanced, aesthetically pleasing lips
and nose soon after birth is understandable.
However, extensive facial growth studies make
clear that this should not be the top priority – that
is, at the cost of good midfacial growth, dental
occlusion, and speech at adolescence (Berkowitz
1996a, b ; Pruzansky 1953 ; Pruzansky et al. 1973 ;
Handelman and Pruzansky 1968 ; Friede and
Pruzansky 1972 ; Friede 1973, 1977, 1978 ; Friede
and Morgan 1976 ; Aduss et al. 1973 ; Vargervick
1983 ; Semb 1991 ) . All of these goals should be
possible without sacri ﬁ cing.
This comparative study contradicts the belief
that well-designed lip/nose surgery with Latham’s
PSO performed on newborns to achieve early
facial aesthetics while damaging palatal growth
sites will lead to excellent adult facial aesthetics
and dental function. But, should the face fail to
grow well, the supporters of POPLA suggest that
it can be easily corrected without far-reaching
consequences such as developing poor self-
esteem as a result of having a retrusive midface.
This condition does not lend itself to easy correc-
tion by midfacial surgery alone. Extensive post-
surgical psychosocial therapy may be necessary
in children with inadequately formed faces
(Tobiasen 1996 ) .
In POPLA cases, a buccal crossbite mostly
involved the deciduous cuspid. The percentage of
anterior crossbite cases increases with time
(Fig. 21.29a , b ). In some POPLA cases, when the
premaxilla is not placed precisely within the arch,
less bone bridging and fewer anterior crossbites
occur. None of the cases show a Class III buccal
(posterior) occlusion. The anterior dental cross-
bite in POPLA CBCLP and CUCLP is always
due to the manipulated premaxilla’s retrusive
position, which is never self-correcting or could
be orthodontically corrected in the deciduous or,
in most cases, even in the permanent dentition. In
vertically growing faces, the anterior crossbite is
orthodontically correctable in most cases. When
the mandible grows forward, as it does in most
faces, the anterior crossbite worsens requiring
LATHAM’S APPLIANCE
What it is expected to do
Fig. 21.7 ( a ) According to Latham, the mode of action
of the appliance used in complete unilateral cleft lip and
palate is the medial movement of both segments toward
each other (Dufresne and So 1992 ) . ( b ) None of our uni-
lateral cleft cases, when activated with the Latham appli-
ance, is shown below. The premaxillary portion of the
noncleft segment is bent mediopalatally, while the lesser-
cleft segment is carried slightly forward, into contact

482
S. Berkowitz and M. Mejia
often a maxillary advancement surgery to create
a proper dental overjet.
A typical CBCLP case of serial cephalometric
tracings (Figs. 21.20 and 21.28 ) shows an early
retrusive midface in the deciduous dentition, cre-
ating an anterior crossbite with a concave facial
pro ﬁ le that worsens with time. An extensive
cephalometric report will be forthcoming in part
two of this overall study. In these CBCLP and
CUCLP cases, the upper and lower face gradu-
ally grows forward, but the palatal length between
the ﬁ rst molars and incisors remains constant
(Figs. 21.30 , 21.31 , and 21.32 ). In most cases,
LeFort I surgery or maxillary advancement with
distraction osteogenesis is necessary, as it was in
this CBCLP case. In the POPLA series, maxil-
lary distraction osteogenesis is seldom utilized in
CUCLP. This surgery results in immediate hyper-
nasality due to an increase in pharyngeal depth
leading to velopharyngeal incompetency (VPI).
The hypernasality slowly diminishes in some
cases within 1 year, but is not completely absent
in all of these cases.
In CBCLP, 57 % of POPLA patients, but only
18 % of the non-POPLA-treated cases, exhibit an
anterior crossbite at 6 years of age (Figs. 21.29 ,
21.30 , and 21.31 , Table 21.1 ). Posterior (buccal)
crossbites are not always related to POPLA
(Fig. 21.29 ). The treatability of buccal crossbites
is also in ﬂ uenced by the extent of scarring cre-
ated by the surgical cleft closure procedure. If a
buccal crossbite is present, some may be easily
corrected by 6–9 years of age in both test groups.
As in CBCLP, the CUCLP shows slight anterior
palatal growth. Most palatal growth occurs poste-
riorly to accommodate the developing molars
(Figs. 21.30 , 21.31 , and 21.32 ).
21.3
POPLA CUCLP (Figs. 21.5 , 21.8 ,
21.9 , 21.10 , 21.11 , 21.12 , 21.13 ,
21.14 , 21.15 , and 21.23 ,
Table 21.1 )
Early anterior dental crossbite in the deciduous
dentition is associated with the loss of the lateral
incisor space brought on by PSOT. This results
from the mediopalatal positioning of the premax-
illary portion of the larger noncleft segment,
which is brought into contact with the forward,
positioned lesser-cleft segment. The following
periosteoplasty creates extensive bone bridging
in over 80 % of the cases.
POPLA patients have greater transverse pos-
terior arch width earlier than what is observed in
non-POPLA cases. This is due to the palatal appli-
ance preventing the neonatal overexpanded lateral
segments from molding together and closing off
most of the palatal cleft space. There is no clinical
advantage for maintaining this increased palatal
width at this early age since POPLA and non-
POPLA cases will eventually attain ideal buccal
occlusion after relatively simple orthodontics.
An anterior dental crossbite with some degree
of midfacial retrusion occurs in 60 % of the
CUCLP POPLA cases by 6 years of age
(Fig. 21.29a , Table 21.1 ), while only 17 % of non-
POPLA-treated cases experience an anterior cross-
bite (Fig. 21.29b , Table 21.1 ). In some POPLA
cases that did not result in an anterior crossbite, the
lateral segments are not in contact at the time of
periosteoplasty. Slight or no bone bridging with a
good lateral incisor space is associated with good
incisor overjet with no midfacial retrusion. The
loss of the lateral incisor space can be anticipated
since alveolar bone is lacking in all cleft alveoli at
birth, and for any bone bridging to occur, the alve-
olar segments need to be placed in contact. In these
cases, the posterior palate is unaffected by PSO
and is usually in a Class I or Class II occlusion
(never in Class III) at 6 years of age.
Anterior dental crossbite correction in POPLA-
treated CUCLP and CBCLP cases requires the
advancement of the premaxilla in CBCLP or
the premaxillary part of the noncleft segment in
CUCLP. In CBCLP POPLA cases, the premax-
illary segment, as a result of the early palatal
manipulation and periosteoplasty of the closed-
over cleft lateral incisor spaces, cannot accom-
modate the impacted lateral incisor, if present
(Fig. 21.9 ). This space cannot be regained in the
mixed dentition and only after extensive ortho-
dontics in the permanent dentition in 50 % of the
cases (Figs. 21.16 , 21.17 , 21.18 , 21.19 , 21.20 ,
21.21 , 21.22 , 21.23 , 21.24 , 21.25 , 21.26 , 21.27 ,
and 21.28 ).

483
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
a
c
f
g
d
e
b
Fig. 21.8 ( a ) After appliance activations: lip and nasal
distortions reduces when the palatal segments are moved
together. ( b ) Alveolar segments in contact periosteoplasty
is then performed. ( c ) Lip adhesion to establish muscle
continuity and mold the overexpanded palatal segments in
a preparation for a de ﬁ nitive lip surgery at 6 months of
age. There are instances when defective lip and nose sur-
gery can be performed without ﬁ rst performing a lip adhe-
sion. ( d ) The absence of the upper right and left central
incisors bringing the segments together, causes the right
segment to move too far medially into a crossbite. ( e ) The
x-ray of the maxillary incisor area shows the missing
centrals and laterals of the permanent incisors. ( f , g ) Note
that the upper lip is slightly retruded when compared to
the lower lip. This is associated with the retraction of the
premaxillary portion of the noncleft palatal segment
Conclusion: Even if the maxillary incisor teeth are con-
genitally missing, it is important to keep all cleft sites
open either for the eruption of the permanent or replace-
ment teeth. This might necessitate the placement of a tem-
porary tooth bearing palatal plate. Had periosteoplasty
been done, the correction of the anterior crossbite would
have been delayed. These cases represent the need to
avoid replacing bony bridges across the cleft area

484
S. Berkowitz and M. Mejia
e
a
d
f
g
b
c
h
j
i

485
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
Fig. 21.9 ( a ) Distorted nostrils and lip due to aberrant
muscle pull. ( b ) Lip adhesion reduces nasal and lip distor-
tion. ( c ) Rotation advancement, the de ﬁ nitive lip closure.
( d – g ) Facial photographs at different ages. Note: The verti-
cal facial growth pattern creates an elongated antero-facial
height. As a result, the midface does not appear to be retru-
sive. A facial protraction mask was utilized with intraoral
palatal expander to advance the maxillary incisors ( h , i , j )
Even though there was extreme crowding of the maxillary
anterior segment, the vertical facial growth pattern neutral-
ized the obtaining of midfacial recessiveness. Lip/nose revi-
sion were excellent in creating symmetry
a
b
c
d
e
f
g
i
h
Fig. 21.10 ( a , b ) The retracted premaxillary portion of
the noncleft segment was brought mediolaterally, block-
ing out the right lateral incisor space. The superior torque
to both palatal segments created an anterior crossbite by
the forces generated by the Latham appliance. ( c , d ) The
crossbite became more noticeable when the deciduous
teeth were lost and the permanent incisors began to erupt.
( e ) Vertical elastics were used to extrude the upper ante-
rior teeth. ( f , g ) The displaced anterior teeth were brought
into alignment. The maxillary central incisors were
advanced, opening the lateral incisor space. ( h ) The thin
bony alveolar bridge created by the periosteoplasty per-
mitted the right and the left palatal segments to be moved
later and the lateral incisor space opened. ( i ) A lower inci-
sor was extracted, and the incisors were retracted in order
to create an overbite and overjet. An anterior open bite
resulted due to the original torque of the palatal segments.
The correction of the alignment of the upper incisor teeth
was not stable even though an upper retainer was worn.
Comment: Occlusal stability is dependent on the position
of the basal bone

486
S. Berkowitz and M. Mejia
Fig. 21.11 ( a ) Facial changes. These show recessive midfaces at 4, 6, and 9 years of age. ( b ) Intraoral occlusal photo-
graphs showing anterior dental crossbites at 4 and 9 years of age
SNA = 75
SNPO = 75
NAPO = 180
ANB = 0
a
SNA = 75
SNPO = 75
NAPO = 180
ANB = 0
SNA = 75
SNPO = 81
NAPO = 189
ANB = 6
Fig. 21.12 ( a ) Superimposed facial polygons show that
midfacial recessiveness does not change between 6 and 9 years
of age. However, with greatly increased mandibular growth,
the facial concavity worsens. Maxillary distraction is being
planned to improve facial aesthetics and peer acceptability.

487
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
Fig. 21.13 Case BR 74. Complete unilateral cleft lip and
palate. Severe anterior crossbite with crowding of incisor
teeth and partial closure of the lateral incisor space.
Treatment plan: uncrowd and advance incisor teeth using
a protraction facial mask
N
A
GO
6yr
4yr
9yr
PO
S
b
( b ) Lateral cephalometric tracings show midfacial recessiveness worsens with facial growth
Fig. 21.12 (continued)

488
S. Berkowitz and M. Mejia
0–1
2–0
2–0
9–6
BR
BR–74
BR–74
74
1–0
Fig. 21.14 Case BR 74. Latham–Millard presurgical
orthopedics periosteoplasty with lip adhesion (POPLA)
procedure. Serial casts re ﬂ ect the creation of a severe
anterior crossbite and anterior dental crowding. 0–1: at
1 year after the POPLA treatment, the premaxilla portion
of the larger segment was brought palatally by the presur-
gical orthopedics. 2–0: 2 years of age. Severe anterior
crossbite with partial closure of the right lateral incisor
spaces. Palatal cleft still open. 2–6: 2 years and 6 months
of age. Palatal cleft space closed with a von Langenbeck
and modi ﬁ ed vomer ﬂ ap. Severe anterior crossbite with
closure of the right lateral incisor space. No posterior
crossbite. Comments: One can predict that the anterior
crowding will become more severe with the eruption of
the permanent incisors. The advancement of the anterior
teeth is dependent upon premaxillary advancement, which
is hindered by the bonny bridge created by the periosteo-
plasty. A protraction facial mask will be necessary to
advance the premaxilla (see facial photographs)
Fig. 21.15 ( a ) Case BR 74. Complete unilateral cleft lip
and palate. Severe anterior crossbite with crowding of the
maxillary incisor teeth as a result of retracing the premax-
illary portion of the larger segment. This created a con-
cave facial pro ﬁ le (cephalometric analysis)
6 years
6/30/1997
SNA = 74
SNB = 76
ANB = −2
SNPO = 77
NAPO = 187
9 years
6/26/2000
SNA = 71
SNB = 74
ANB = −3
SNPO = 76
NAPO = 190
10 years
CASE # BR-74
a
5/19/2001
6 years
9 years
10 years
SNA = 70
SNB = 75
ANB = −5
SNPO = 77
NAPO = 192

489
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
Fig. 21.16 Case BT 80. Complete
bilateral cleft lip and palate. The
protruding premaxilla was set back
and aligned within the arch using the
POPLA procedure. By 4 years of age,
a severe anterior crossbite resulted. At
10 years of age, the teeth in the
unerupted severely ventro ﬂ exed
premaxilla still failed to erupt. This
necessitated the placement of a gold
chain on the imbedded central incisors.
Hooks were placed on the chains with
elastics extending to a Delaire-type
facial mask. Treatment is still
underway
b

( b ) Case BR 74. Periapical
radiograph. 7 1/2 years of age. Severe dental
crowding of the maxillary anterior teeth
Fig. 21.15 (continued)

490
S. Berkowitz and M. Mejia
4 years
6/16/1997
SNA = 86
SNB = 83
ANB = 3
SNPO = 84
NAPO = 173
8 years 3 months
1/15/2001
SNA = 86
SNB = 84
ANB = 2
SNPO = 85
NAPO = 178
8 years 11 months
9/8/2001
SNA = 88
SNB = 84
ANB = 4
SNPO = 85
NAPO = 174
9 years
1/12/2002
SNA = 86
4 years
9 years
8 years and 3 months
SNB = 84
ANB = 2
SNPO = 85
NAPO = 180
8 years and 11 months
Fig. 21.17 Lateral
cephaloradiographs
show
the
unerupted severely ventro ﬂ exed premaxilla in crossbite.
The superimposed cephalometric tracing shows the con-
tinued midfacial growth keeping up with the other facial
areas, resulting in a ﬂ at facial pro ﬁ le. One can conclude
that growth at the premaxillary vomerine suture is still
active. However, the bony bridge between the lateral pala-
tal segments and the premaxilla prevents the premaxilla’s
proper alignment and eruption into position
Fig. 21.18 Case BK 36. Bilateral cleft lip and palate.
The protruding premaxilla was retruded and aligned
within the arch using the POPLA procedure. At 7 years
and 4 months of age, an anterior dental crossbite resulted.
A protruding lower lip is due to the permanent mandible
and ﬂ aring incisor teeth

491
21 A Comparison of the Effects of the Latham–Millard POPLA Procedure
Fig. 21.19 Case BK 36. Bilateral cleft lip and palate. The
corrective treatment plan was designed to advance the
crowded anterior maxillary incisors with facial protrac-
tion mechanics and position the cuspids into the missing
lateral incisors spaces. Extraction of the mandibular ﬁ rst
bicuspids and retracting the incisors will help to reduce
the crossbite without the need to advance the upper inci-
sors. This case is still under treatment. Case BK 36.
Bilateral cleft lip and palate. The corrective treatment plan
was designed to advance the crowded anterior maxillary
incisors with facial protraction mechanics and position the
cuspids into the missing lateral incisors spaces. Extraction
of the mandibular ﬁ rst bicuspids and retracting the inci-
sors will help to reduce the crossbite without the need to
advance the upper incisors. This case is still under
treatment