Ortho CLP Lessons of The 60 Years PDF

STATE OF THE ART
Orthodontic Treatment of the Patient With Complete Clefts of Lip,

Alveolus, and Palate: Lessons of the Past 60 Years
ROSS E. LONG JR., D.M.D., M.S., PH.D.
GUNVOR SEMB, D.D.S., DR. ODONT.
WILLIAM C. SHAW, F.D.S., PH.D.
This review paper summarizes 60 years of progress in the orthodontic care
of patients with complete clefts of the lip, alveolus, and palate. The progress
and evolution of orthodontic treatment for patients with complete clefts has
taken a slow and sometimes circuitous route to present-day standards. Nonetheless, in spite of this history of slow and inefficient scientific and clinical
progress, review of the literature of the past 60 years does, in fact, reveal that
progress has been made. This progress has not only been in the area of improved surgical and orthodontic techniques but also relative to the scientific
weaknesses of past decades. The investigations of more recent years seem to
be moving in a direction of better documentation, stricter methodologies, longer-term follow-up, larger sample sizes, etc. However, there remains much
work to be done. Only by adopting a more critical approach will protocols be
defined that can achieve optimal outcomes, while minimizing the burden of the
orthodontic treatment through elimination of superfluous intervention.
KEY WORDS: bone grafting, cleft lip and palate, dental development, dental
occlusion, orthodontic treatment
While orthodontics, as a specialty of dentistry, dates to the

turn of the previous century, the role of the orthodontist on
the cleft palate team has become more clearly defined only
over the past 60 years. Examination of the literature over this
period seems to suggest the appearance of key milestones each
decade that have contributed significantly to the improvement
in orthodontic treatment outcomes for patients with clefts.
It is also of note that no discussion of orthodontic progress
could be complete without an understanding of the essential
dependence of orthodontics on surgery. In no other realm of
orthodontic treatment is the success or failure of treatment so
inextricably tied to surgical capabilities and outcomes as it is
in the management of patients with clefts. As will be seen
below, most of the improvements in orthodontic treatment outcomes over the past 60 years are actually related to improve-
ments in surgical techniques rather than any significant innovations in orthodontic appliances.
This review summarizes this progress in orthodontics as it
has contributed to the current concepts and controversies of
orthodontic treatment of the patient with complete clefts of lip
and palate. This review of the literature of the past 60 years
is intended to be representative but not encyclopedic. In addition, because the effectiveness of the orthodontic approaches
to the problems of complete clefts of lip and palate have not
been evaluated through randomized control trials (Roberts et
al., 1991; Shaw et al., 1996), at this point it is impossible to
state that one approach is unequivocally better than another.
However, these methodological shortcomings notwithstanding,
a review of the recent history of these milestones may help
explain the current state of the art in orthodontic treatment and
point to future directions to continue our progress.
MILESTONES
Dr. Long is Director of the Lancaster Cleft Palate Clinic and Head of Orthodontics and Research of the Lancaster Cleft/Craniofacial Program, Lancaster, Pennsylvania; Assistant Professor of Orthodontics, Department of Orthodontics, Albert Einstein Medical Center, Philadelphia, Pennsylvania; and
Assistant Professor of Orthodontics, Department of Orthodontics, University of
Maryland Dental School, Baltimore, Maryland. Dr. Semb is a senior lecturer
in craniofacial anomalies and Head of the Dental Unit in the Department of
Plastic Surgery, and Dr. Shaw is Chairman of the Department of Orthodontics,
University of Manchester, Manchester, United Kingdom.
Reprint requests: Ross E. Long Jr., D.M.D., Ph.D., Lancaster Cleft Palate
Clinic, 223 North Line Street, Lancaster, Pennsylvania 17602. E-mail
rlong@supernet.com.
IN THE IMPROVEMENT OF
ORTHODONTIC
TREATMENT OUTCOMES
1940s: Development of the Team Concept
The concept of team treatment was developing simultaneously in Europe and the United States in the 1930s and
1940s. For example, in Denmark, a law was passed in 1937
stating that a single national team should provide care for all
Danish individuals affected by clefts and should perform regular multidisciplinary follow-up. Interestingly, one of the cen533-1
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Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6
tral figures in this milestone in the United States was an orthodontist, Herbert K. Cooper. Because of the traumatic nature
of primary surgery at the time and lack of coordinated interdisciplinary care, the subsequent dentofacial deformities exceeded most orthodontic treatment capabilities (Harkins,
1960). Thus, many early publications by these and other orthodontists at the time were as much focused on the need for
interdisciplinary teamwork (Cooper, 1942, 1953) as they were
on orthodontic techniques (Harvold, 1947, 1949). Following
the efforts of these orthodontists and other pioneers in the field,
the American Cleft Palate Association was formed in 1943,
with the mission of promoting the interdisciplinary care of
patients with clefts.
1950s: Understanding of the Role of Scar Tissue in Cleft
Dentofacial Deformity
A second milestone was the documentation by orthodontists,
participating on newly formed teams, of the severe three-dimensional maxillofacial growth disturbances in patients with
repaired clefts of lip and palate (Harvold, 1947, 1954; Graber,
1949, 1954; Slaughter and Brodie, 1949; Pruzansky, 1954).
When compared with early reports of near-normal maxillofacial growth in unoperated cleft populations (Ortiz-Monasterio
et al., 1959; Mestre et al., 1960), this irrefutable evidence of
significant growth differences drew attention to the amount and
location of scar tissue from primary surgery as the causative
factor. This awareness and subsequent improvements in primary surgical techniques contributed substantially to a decrease in the maxillary growth deformities that previously precluded the successful use of routine orthodontic approaches
(Bergland, 1967).
1960s: Development of Presurgical Orthopedics and
Primary Bone Grafting
Early descriptions of preoperative alignment of cleft maxillary segments were actually reported at the beginning of the
1950s (Kjellgren, 1949; McNeil, 1950). However, interest in
this technique of presurgical orthopedics increased through the
1960s (Burston, 1958; Huddart, 1961, 1967; Hotz, 1969; GrafPinthus and Bettex, 1970; Robertson and Hilton, 1971), when
it also became linked to primary bone grafting (Nordin, 1957;
Johanson and Ohlsson, 1961; Rosenstein 1963, 1969; Brauer
and Cronin, 1964). Most importantly, the early proponents of
these primary alveolar repairs identified the potential benefits
of restoration of the cleft alveolar ridge, most notably the improved bone support for cleft-adjacent teeth and the ability to
eliminate the need for prosthetic replacement of missing lateral
incisors. However, this new approach was not without its detractors (Pruzansky, 1964) who cautioned against jumping on
bandwagons based on short-term results presented as retrospective case series. Subsequently, early randomized control
trials and long-term evaluations of maxillary growth and malocclusion following primary bone grafting demonstrated additional problems from the early alveolar repair, dampening
the initial enthusiasm for this surgical and orthopedic approach

(Rehrmann et al., 1970; Jolleys and Robertson, 1972; Friede
and Johanson, 1974). While some particular variations of primary grafting and presurgical orthopedics have been reported
to provide the benefits without the growth risks (Rosenstein et
al., 1972, 1982), the concern over growth problems from this
early surgery remain (Ross, 1987c).
Another alternative to primary bone grafting was also introduced in this period. Skoog (1965) described the use of local
periosteal flaps with the intention of promoting bone formation
in the cleft site. Although a subsequent publication reported
bone formation without the facial growth problems (Hellquist
and Ponten, 1979), periosteoplasty did not become widely popular. A variation of periosteoplasty, the gingivoalveoloplasty
or gingivoperioplasty, has been reintroduced (Millard, 1980;
Delaire, 1989) and has been shown to promote bone formation
in some patients (Santiago et al., 1998) with evidence of minimal short-term growth effects (Wood et al., 1997). However,
the long-term effects on facial growth and consistent avoidance
of bone grafting have not yet been determined.
1970s: Development of Mixed Dentition Bone Grafting
and Emphasis on Mixed Dentition Treatment
The landmark publications of Boyne and Sands (1972,
1976), describing bone-grafting surgery as a later procedure,
renewed interest in the orthodontic benefits to be derived from
this surgical technique. Improved bone support for cleft-adjacent teeth and the ability to eliminate the need for prosthetic
replacement of missing lateral incisors have been documented
byholm et al., 1981; Hall and Posnick,
by many authors (A
1983; Turvey et al., 1984; Enemark et al., 1985, 1987; Bergland et al., 1986a, 1986b; Helms et al., 1987; Lilja et al., 1987;
Paulin et al., 1988). Delaying the procedure until a majority
of maxillary growth has been completed seems also to have
minimized or eliminated the potential damaging growth effects
of bone grafting when carried out at younger ages (Ross,
1987c; Enemark et al., 1987; Semb, 1988; Levitt et al., 1999).
To derive maximum benefit from alveolar bone grafting, it was
recommended that the graft be placed prior to eruption of the
permanent canine and coordinated with mixed dentition ortho byholm et al.,
dontic treatment (Waite and Kersten, 1980; A
1981; El Deeb et al., 1982; Enemark et al., 1985, 1987; Bergland et al., 1986a, 1986b; Paulin et al., 1988).
1980s: Development of Orthognathic Surgical Approaches
for Maxillary Deficiency
Development of the techniques for maxillary osteotomies
promoted by Obwegeser (1966, 1969) and Bell (1973, 1975),
and especially the surgical advancement of the cleft maxilla,
further expanded orthodontic treatment capabilities by allowing for simultaneous correction of skeletal and dental deformities (Converse et al., 1974; Braun and Sotereanos, 1980;
Tideman et al., 1980; Schendel and Delaire, 1981; Westbrook
et al., 1983; Poole et al., 1986). The more recent application
Long et al., ORTHODONTIC TREATMENT
of distraction osteogenesis to cleft-related maxillary deficiencies has similarly expanded orthodontic expectations, especially when integrated with protraction orthopedics (Molina,
1998; Figueroa and Polley, 1999).
1990s: Emphasis on Intercenter Audits and Outcome
Documentation
By the 1990s, the array of surgical treatment approaches had
become enormous, with little reliable information available
upon which to base rational decision making in choosing one
method over another (Semb and Shaw, 1998). In a recent survey of 201 European cleft centers, 194 different surgical protocols were in use for the primary closure of a complete unilateral cleft lip and palate alone (Shaw et al., 2000). With this
number of approaches possible, the inadequate research methods of previous decades left little chance of identifying specific
individual procedures that could be shown to be unequivocally
superior to others (Spriestersbach et al., 1973; Roberts et al.,
1991; Shaw et al., 1996; Semb and Shaw, 1998). However, an
approach using rigorous comparisons of outcomes from different centers, marked a significant change in direction from
previous methodologies. By evaluating outcomes that impacted on orthodontic treatment (occlusion, maxillary growth)
among six European centers with different surgical and orthodontic protocols, Mlsted et al. (1992) and Mars et al. (1992)
were able to draw general conclusions about the relationship
between surgical protocol and orthodontics. Most notably, the
outcomes found most favorable for routine nonsurgical orthodontic finishing appeared to be in centers with simple surgical
and orthodontic approaches (Shaw et al., 1992a, 1992b). While
clearly not capable of identifying the individual procedures
within a total surgical and orthodontic protocol that are responsible for favorable or unfavorable outcomes, intercenter
comparisons have become a key part of quality improvement
programs, leading to international agreements on standardized
documentation (Shaw et al., 2000).
CHARACTERISTIC ORTHODONTIC PROBLEMS
COMPLETE CLEFTS OF LIP AND PALATE
IN
Detailed analysis of large groups of individuals with unoperated clefts reveals characteristic underlying variations in facial form that occur independently of surgical interference
(Mars and Houston, 1990; da Silva et al., 1992, 1993; Capelozza et al., 1993). Complete clefting of the lip and palate even
without surgery is associated with mandibular retrusion, increased gonial angle and increased lower anterior facial height,
and a small but relatively protrusive maxilla with short upper
posterior facial height. The additional adverse influence of surgery is evident mostly in the anteroposterior and transverse
dimensions of the maxilla.
Maxillary Skeletal Retrusion
Many patients with repaired complete clefts exhibit varying
degrees of maxillary sagittal deficiency, which increases with
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age as the cleft maxilla fails to maintain normal growth rates.

In unilateral clefts, this deficiency is often evident at early ages
and is progressive over time (Dahl, 1970; Ross, 1987a; Enemark et al., 1990; Paulin and Thilander, 1991; Semb, 1991b;
Smahel and Mullerova, 1994). In bilateral clefts, the appearance of the deficiency may be delayed slightly because of the
initial prominence of the premaxilla but frequently appears by
adolescence (Dahl, 1970; Friede and Pruzansky, 1985; Semb,
1991a; Trotman and Ross, 1993; Heidbuchel et al., 1994; Semb
and Shaw, 1996). Some studies have identified these facial
growth disturbances to be greater in males (Ross, 1987a; Paulin and Thilander, 1991; Semb, 1991b), and the failure of other
investigators to find similar sex differences in maxillary
growth up to the age of 10 years (Krogman et al., 1982) may
further substantiate the importance of the final adolescent
growth period in revealing the full extent of the dysplasia. This
retrusive growth pattern represents one of the orthodontists
greatest challenges and early recognition of skeletal imbalance
may avoid an unnecessary burden of fruitless early orthodontic
treatment of a skeletal problem.
Transverse Skeletal Dysplasias
The change in the relationship of the cleft alveolar segments
following primary lip and palate repair has been well documented (Subtelny, 1955; Pruzansky and Aduss, 1964; Mazaheri et al., 1967, 1971; Aduss and Pruzansky, 1968; Harding
and Mazaheri, 1972; Berkowitz et al., 1974; Ross, 1975; Vargervik, 1981, 1990; Berkowitz, 1990; Honda et al., 1995,
Heidbuchel et al., 1998). With varying degrees of medial rotation and collapse of the skeletal elements, the cleft maxilla
is predisposed for transverse deficiencies. However, some studies have shown significant collapse only 50% of the time at 4
years of age, with many patients exhibiting only minor medial
rotation limited to the cleft site itself (Vargervik, 1990; Mazaheri et al., 1993). Furthermore, the predictability of this collapse, and its relationship to the development of future posterior crossbite, has also been shown to be uncertain (Schwartz
et al., 1984). This probably accounts for the less common appearance of clear transverse deficiencies in the primary dentition, as opposed to later stages of development (Bergland and
Sidhu, 1974; Cooper et al., 1979; Olin, 1990). However, exacerbation of these initial transverse problems occurs with additional dental eruption. They appear to be related to the proximity and amount of scarring adjacent to the alveolar ridge
that create a further lingual deflection of the erupting permanent dentition, aggravating the initial segment collapse (Kremenak et al., 1967; Ross and Johnston, 1972) Also, Ishiguro
et al. (1976) demonstrated an additional progressive narrowing
of the basal maxilla relative to the mandible, not through further segment collapse but through a lack of normally expected
incremental width gains on the cleft side in unilateral clefts
and on both sides in bilateral clefts.
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Vertical Skeletal Dysplasias

As mentioned above, abnormalities in vertical maxillary
growth also appear to be associated with corresponding changes in mandibular growth direction even in the unoperated cleft
condition. This opening rotation of the mandible may be exacerbated by additional primary and secondary surgical procedures, but the resultant mandibular retrusion can occasionally mask a maxillary deficiency, albeit at the expense of a
bimaxillary retrognathic facial profile (Long and McNamara,
1985; Ross, 1987c; Trotman et al., 1996). Finally, this lower
face height excess combined with a concomitant deficiency in
maxillary height and possible impedance of posterior tooth
eruption has been reported to lead to an excessive freeway
space in patients with clefts (Ross and Johnston, 1967).
Abnormalities in Dental Development
Abnormalities in dental development have been described
by a number of authors. They all can be related to initial disturbance and disruption of the embryonic process of dental
lamina formation, most notably in the area of failed merging
and fusion of the medial nasal, lateral nasal, and maxillary
processes (Bhn, 1950, 1963; Ross and Johnston, 1972; Ranta,
1986; Long, 1998). Although the reported frequencies of various anomalies may vary, the overall pattern and types of
anomalies reported have been remarkably consistent over the
past 60 years.
The clinical effects of these embryonic failures include
missing teeth, supernumerary teeth, hypoplastic teeth, dysmorphic teeth, and impacted teeth (Bhn, 1950, 1963; Jordan
et al., 1966; Kraus et al., 1966; Ranta, 1986; Semb and
Schwartz, 1997). The tooth most commonly affected is the
lateral incisor. Its absence appears to be in the range of 10%
to 20% in the primary dentition, and 30% to 50% in the permanent dentition (Bhn, 1950, 1963; Jordan et al., 1966; Ranta, 1972, 1986, 1990; Ross and Johnston, 1972; Suzuki et al.,
1992). When the lateral incisor is present, it can occur on
either side of the cleft with near-normal form or be rudimentary or malformed, especially in cases in which there are supernumeraries on both sides of the cleft (Bhn, 1950, 1963;
Ross and Johnston, 1972).
In addition to the problems with lateral incisor development,
other dental anomalies that affect orthodontic treatment occur
with greater frequency in the cleft population. Jordan et al.
(1966) reported that 54% of their cleft sample demonstrated
dental anomalies as opposed to 15% in the noncleft population.
Ranta (1983, 1986, 1990) has reported a significantly higher
incidence of congenitally missing teeth other than those adjacent to the cleft site (e.g., second premolars and maxillary
lateral incisors on the noncleft side). Generalized delayed dental development on the sides of the maxillary arch affected by
complete clefts has also been reported (Fishman, 1970; Solis
et al., 1998). Overall tooth size has been shown to be smaller
in patients with clefts (Foster and Lavelle, 1971; Peterka and
Mullerova, 1983; McCance et al., 1990). Impaction of per-
manent canines adjacent to cleft sites has been shown to occur

at a much higher frequency in patients with unilateral complete
cleft lip and palate than in the noncleft population (Semb and
Schwartz, 1997). Finally, a higher frequency of first molar and
incisor impaction has been reported (Bjerklin et al., 1993), and
the prevalence of dental caries has been found to be increased
in children with clefts (Bokhout et al., 1996). These dental
anomalies all impact upon orthodontic treatment planning decisions such as timing of orthodontic intervention, timing of
bone grafting, sequencing of orthodontic and surgical treatment, extraction of teeth, space opening for prosthetic replacements versus space closing, etc.
Dental Occlusion and Alignment
Numerous authors have summarized the most common
problems with occlusion and alignment (Ross, 1975; Cooper
et al., 1979; Subtelny, 1990; Vargervik, 1990). Most of these
are a reflection of the initial intrinsic deformities of the cleft
itself as well as postsurgical changes, and certain characteristics seem to be pervasive in their appearance, regardless of the
specifics of prior treatment. Posterior crossbites are frequent,
varying in severity from single tooth to total segment involvement and becoming worse in the permanent dentition. Maxillary incisor rotations, lingual inclination, and frequent crossbites are typical also. Finally, significant arch asymmetry and
maxillary dental midline deviations are all common characteristics of unilateral clefts (Semb and Shaw, 2000). Of greatest
interest from a historical perspective is not that the orthodontic
problems with occlusion and alignment just mentioned represent currently unique challenges. Rather, they are the very
same problems described by orthodontists for the past 60 years
(Harvold, 1947; Cooper, 1951; Pruzansky, 1954; Ricketts,
1956; Subtelny, 1957). However, while our surgical and orthodontic progress seems not to have eliminated many of them,
the relative severity of the problems seems to have decreased
at many centers, leading to the possibility of more successful
orthodontic treatment results for a larger percentage of patients.
Finally, with regard to the evaluation of dental occlusion
and alignment and future orthodontic treatment difficulties, the
development of rating schemes such as the Goslon Yardstick
(Mars et al., 1987) and the 5 year yardstick (Atack et al.,
1997, 1998) represent significant contributions. Their use in
numerous investigations such as the Eurocleft Study (Mars et
al., 1992), the Scandcleft study (Friede et al., 1991), and the
nationwide study of standards of surgical outcome in the U.K.
(Sandy et al., in press) have confirmed their validity, reliability,
and utility.
ORTHODONTIC TREATMENT PROCEDURES
WITH COMPLETE CLEFTS
FOR
PATIENTS
Benefit Versus Burden

The literature of the past 60 years is filled with case reports
and case series that appear to demonstrate the successful man-
agement of numerous cleft-related orthodontic problems

through surgical, orthopedic, or orthodontic treatment. In addition, given the continuous and often progressive nature of
orthodontically related problems over the entire growth and
dental eruption stages of the child with a cleft, treatment recommendations for nearly every conceivable age can be found.
Therein lie two of the most basic problems in evaluating the
efficacy of orthodontically related treatment protocols: (1) a
lack of long-term, methodologically sound evaluations of treatment results and (2) the failure to consider the total burden of
treatment imposed on the patient versus the benefits expected
through multiple phases of orthodontic intervention. Both of
these aspects were stressed in the 1993 report by the American
Cleft PalateCraniofacial Association, Parameters for Evaluation and Treatment of Patients with Cleft Lip/Palate or Other
Craniofacial Anomalies. In the list of Fundamental Principles it is clearly stated that . . . it is the responsibility of
each team to monitor both short-term and long-term outcomes
. . . . . . . Longitudinal follow-up of patients, including appropriate documentation and record keeping, is essential (p. 6).
In discussion of Dental Care, it is stressed that continuous
active orthodontic treatment . . . should be avoided (p. 18).
An additional point in the same document is that teams (and
team members) should see sufficient numbers of patients
each year to maintain expertise in diagnosis and treatment
(p. 5). This has also been stressed as an issue of growing
importance by other authors (Shaw et al., 2000) since the orthodontic management of patients with complete clefts has become a unique subspecialty calling for special skills and experience that are unlikely to be mastered through occasional
involvement.
Presurgical Orthopedic Treatment
This topic has been covered in depth in a separate review
article. Of interest however, is that comparisons of patients
treated at the same center with and without presurgical orthopedics (for geographical reasons) have not shown benefit from
the intervention (Huddart, 1974; Ross and MacNamara, 1994).
The resurgence in enthusiasm for this approach seems to have
been driven by the development of appliances that provided
more direct and precise control of the maxillary segments both
prior to and following surgery (Latham, 1980). Furthermore,
the inclusion of nasal molding extensions on to the basic orthopedic appliance has expanded the treatment objectives to
include improved nasal esthetics following primary repair
(Grayson et al., 1999; Maull et al., 1999). However, of all
orthodontic protocols presently in use, the value of these approaches remains considerably uncertain because of the common problems described above: no long-term evaluations of
treatment results, no controlled studies, and failure to demonstrate that the additional burden of treatment inherent in
these methods produces long-term benefits sufficiently better
than those available through less costly and less involved procedures. Although some preliminary attempts have been made
to address these issues (Wood et al., 1997), it is incumbent on
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proponents of these techniques to continue careful and standardized documentation, to carry out rigorous intra- and intercenter audits of treatment outcomes, and to participate in randomized control trials. To date, the only randomized trial of
presurgical orthopedics reported did not reveal any benefits but
did document the economic burden of the treatment (KuijpersJagtman and Prahl, 1996).
Primary Dentition Treatment
Orthodontic intervention in the primary dentition has occasionally been recommended over the past 60 years, though
less so in recent years. Of interest is the fact that routine primary dentition treatment seems to have been more frequently
emphasized in the decades during which the severe three-dimensional maxillary deformities in clefting were being exposed. Suggested treatment at that time ranged from full banding (Pierce et al., 1956) to routine arch expansion (Harvold,
1949; Pruzansky, 1954, 1955; Subtelny and Brodie, 1954; Subtelny, 1957, 1966; Olin, 1966; Fishman, 1969). The goals and
proposed benefits of this phase of treatment, mentioned by
these authors, included improvement of alveolar development
in the cleft site by unlocking overlapped maxillary segments; improvement in speech development and nasal breathing by expanding the maxilla and providing more tongue
space; improvement in masticatory function by elimination of
crossbite; and improvement in future permanent tooth eruption
and alignment. If one considers the fact that the more severe
maxillary deformities seen in these earlier decades probably
resulted in an earlier-aged appearance of significant maxillary
arch deformities and malocclusion, it is logical that primary
dentition intervention would have received greater emphasis.
Although some authors have continued to stress the need
for routine primary dentition treatment (Subtelny, 1990), others have suggested treatment only for severe functional problems (Ross, 1975; Cooper et al., 1979; Aduss and Figueroa,
1990; Olin, 1990). Still others saw no benefit to primary dentition intervention at all (Bergland, 1973; Bergland and Sidhu,
1974; Semb and Shaw, 2000). It seems likely that these differing beliefs may be partially related to the decreasing severity of primary dentition problems that occurred naturally over
time as primary surgical procedures improved. However, the
strongest argument against primary dentition treatment is that
it does not pass the burden versus benefit of treatment test.
There is currently no evidence that the additional treatment
provided at this age either eliminates mixed dentition intervention or can provide improved capabilities or results not
possible through a single phase of treatment in the mixed dentition. Primary dentition treatment also calls for lengthy posttreatment retention adding significantly to the overall period
when the child must wear appliances.
Mixed Dentition Treatment
Over the past two decades, the scope of mixed dentition
treatment has been broadened and refined. Many of the skeletal
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discrepancies and dental irregularities mentioned above are

amenable to mixed dentition intervention. Numerous authors
have stressed the beneficial effects on future dental and skeletal development through the elimination of functional and
structural problems at this developmental stage (Ross and
Johnston, 1972; Vargervik, 1981, 1990; Ohkiba and Hanada,
1989; Subtelny, 1990; Rygh and Tindlund, 1996). The most
commonly mentioned procedures include maxillary expansion
to correct the reduced transverse dimension; incisor alignment
and proclination to remove crowding, rotations, and anterior
crossbites; and maxillary protraction to reduce maxillary retrusion. These procedures tend to be coordinated carefully
around alveolar bone grafting (Vig and Turvey, 1985; Vig et
al., 1996).
Incisor Alignment
Alignment of severely malpositioned incisors in the mixed
dentition of children with complete clefts has been recommended for decades because of its positive effect on self-esteem (Olin, 1960; Cooper et al., 1979; Semb 1990; Subtelny,
1990). Simultaneous correction of anterior crossbite when due
to retroclination of the maxillary incisors has been recommended for the additional benefit of maximizing anterior development of the maxillary dentoalveolar process (Ross and
Johnston, 1972; Vargervik, 1981; Subtelny, 1990). The methods used to accomplish this range from removable appliances
to partial fixed appliances (Cooper et al., 1979). The most important issue involving the sequencing of this procedure prior
to bone grafting relates to the danger of perforating the thin
lamina of alveolar bone covering the roots of teeth adjacent to
the cleft site (Turvey et al., 1984; Vig and Turvey, 1985; Vig
et al., 1996). However, with careful evaluation of the bone
support for cleft-adjacent teeth and proper control of root angulation away from the cleft site, this presurgical alignment is
possible in many cases and can be economically achieved concomitant with pregrafting expansion when this is also necessary (Semb, 1990; Long, 1995).
Maxillary Expansion
The need for maxillary expansion in many patients with
complete clefts has been recognized in nearly all publications
related to orthodontic treatment over the past 60 years (Harvold, 1949; Subtelny and Brodie, 1954; Pruzansky, 1954,
1955; Olin, 1966; Bergland, 1973; Ross, 1975; Cooper et al.,
1979; Vargervik, 1981; Vig and Turvey, 1985; Aduss and Figueroa, 1990). Although many of these early publications indicated the need for expansion in nearly all cases, a more recent report found this need in only 25% of 409 patients with
complete clefts (Semb, 1990). The proposed benefits of mixed
dentition maxillary expansion have been described identically
to those listed for expansion in the primary dentition. Interestingly, most authors recommending infant presurgical orthopedics and routine primary dentition expansion also acknowledge the need for additional expansion in the mixed dentition.
This would seem to indicate that management of maxillary

width at those earlier periods does not routinely preclude the
need for additional expansion later, raising again the question
of the benefit versus burdens of these additional phases of
treatment, at least with regard to arch width.
A practical benefit of expanding maxillary segments prior
to bone grafting is the ease with which skeletal movement and
segment rotation can occur (Vargervik, 1978) with appliances
as simple as a removable quad helix (Bergland et al., 1986a,
1986b). With fixed jack screwtype expanders, slow expansion
of the appliance can also accomplish segmental expansion because of the absence of a complete midpalatal suture in cases
with complete clefts (Long, 1995). The absence of the midpalatal suture also means that pregrafting expansion accomplishes skeletal segmental movement at the expense of increasing the cleft width (Long, 1995; Long et al., 1995). While
creating a cleft width that exceeds the soft tissue coverage
capabilities could negatively impact on bone graft results
(Long et al., 1995), the majority of reports in which presurgical
expansion was carried out have shown successful bone grafting results (Johanson et al., 1974; Turvey et al., 1984; Bergland et al., 1986a). A final consideration using presurgical expansion is the possibility of uncovering covert oronasal fistulas, which could then be repaired simultaneously with bone
graft placement (Long, 1995; Vig et al., 1996), eliminating the
need for a separate fistula repair procedure.
A final area of concern relates to the exact timing of graft
placement during the total mixed dentition treatment phase. A
number of publications have suggested that the position and
stage of development of the permanent cuspid is the most important diagnostic feature in making this determination. Waite
byholm et al. (1981), El Deeb et al.
and Kersten (1980), A
(1982), Troxell et al. (1982), Turvey et al. (1984), Bergland et
al. (1986a, 1986b), and Sindet-Pedersen and Enemark (1993)
have all used this method in suggesting optimal timing for
graft placement. All have shown good results with this approach, although each used slightly different criteria (root formation, crown position, etc.). Delay of graft placement until
after completion of cuspid eruption has also been shown to
produce inferior results (Sindet-Pedersen and Enemark, 1985;
Bergland et al., 1986a; Helms et al., 1987). However, Long et
al. (1996) were unable to find any correlation between cuspid
positioning, from unerupted to fully erupted in the cleft site,
and subsequent bone graft success when using their more detailed radiographic measurements. Finally, it should also be
mentioned that the foregoing concern over graft timing and
cuspid positioning is based on situations with missing or unusable lateral incisors. Rygh and Tindlund (1996), Lilja et al.
(2000), and Semb and Shaw (2000) have emphasized the need
to consider earlier alveolar repair in situations in which a lateral incisor is present and requires additional bone support for
further normal development and eruption. At present it is uncertain as to how much younger this alveolar repair can be
done before the possible benefits of improved support for lateral incisors is offset by the risks of growth disturbances found
following primary bone grafting.
Maxillary Protraction Orthopedics

The desire to improve developing maxillary skeletal retrusion is also a treatment modality that has been recommended
for this mixed dentition treatment phase. Delaire (1971) was
one of the earliest proponents of the use of protraction face
frame therapy to attempt nonsurgical correction of the maxillary deficiency (Delaire, 1971; Delaire et al., 1972). Subtelny
(1980), Sarnas and Rune (1987), Buschang et al. (1994) also
described patients treated successfully with this approach. A
substantial follow-up of cases has been reported by Tindlund
and colleagues (Rygh and Tindlund, 1982, 1996; Tindlund,
1989, 1994; Tindlund and Rygh, 1993; Tindlund et al., 1993).
While providing evidence in the short-term of statistically significant changes in anteroposterior position when starting treatment on patients 6 years of age, these authors also described
the actual dimensional changes having taking place, which varied from patient to patient. Some of these changes, on average,
approached clinical significance, with maxillary advancements
averaging 2 mm to 3 mm after 12 to 15 months of treatment.
However, this response was found in only 63% of the sample,
with the poor response group (27% of the sample) showing
only 0 mm to 1 mm advancement over the same time period.
A combined average of about 1.5-mm advancement over 12
to 15 months raises the question of the clinical significance
and true long-term benefits of this procedure. With the possibility that following discontinuation of the appliance, the future growth of these patients will continue to be maxillary
deficient, the degree to which this procedure has reduced or
eliminated the need for future orthognathic surgery has not
been determined. As a result, some authors have not recommended the routine use of this approach (Semb and Shaw,
2000) inasmuch as it may also fail to pass the benefits versus
burden of treatment test.
Finally, a combination of maxillary protraction procedures
with distraction osteogenesis of the maxilla has been proposed
more recently. By using either a removable protraction face
frame (Molina, 1998) or a rigid external distraction device (Figueroa and Polley, 1999) and carrying out simultaneous corticotomy cuts in the buccal cortex of the maxilla, the latter
authors have shown case reports that demonstrate significant
maxillary movement in relatively short periods of time (maxillary advancement of 7 mm to 8 mm after 3 to 4 months of
treatment). Although there are currently no long-term evaluations concerning stability and permanent benefits, the possibility exists that by increasing the benefits (greater maxillary
advancement) and reducing the burdens (shorter treatment
time), this combination could overcome the shortcomings of
standard nonsurgical protraction methods. On the other hand,
if further maxillary advancement is still required later in a
majority of patients, the benefits of early distraction become
more dubious.
Permanent Dentition Treatment
As a result of the milestones listed above, the description
of permanent dentition treatment has changed dramatically
533-7
over the past 60 years. These changes have been most adequately characterized by Bergland et al. (1986a). In their description of progress in orthodontic treatment, these authors
characterized the period before 1945 as the Prosthodontic
Era because of the extreme nature of the dentofacial deformities following the traumatic nature of primary surgery that
was carried out at that time. Final permanent dentition treatment rested largely in the hands of the prosthodontist rather
than the orthodontist. With improvements in surgery and interdisciplinary team management, and a subsequent decrease
in maxillary growth problems, Bergland et al. described the
period of 1945 to 1975 as the Orthodontic/Prosthodontic
Era. Although the problems encountered fell more within the
capabilities of orthodontic treatment, prosthetic replacement of
missing teeth across the cleft site was still required following
arch expansion. More recently, with the introduction of reliable
bone grafting, the current phase has been labeled as the NonProsthodontic Challenge. With coordinated team management, excellent primary surgery, favorable sagittal growth patterns unrestricted by excessive scar tissue, proper management
of the maxillary segment collapse, successful and timely bone
grafting, and normally erupting dentition, many authors report
an increasing frequency of permanent dentition treatment that
is possible using the common approaches for routine noncleft
orthodontics (Olin, 1990; Subtelny, 1990; Aduss and Figueroa,
1990; Rygh and Tindlund, 1996; Semb and Shaw, 2000).
Management of the Dentition Adjacent to the Previous
Cleft Site
Since the advent of routine and successful bone grafting,
space closure in the cleft site has become a desirable and
achievable goal to eliminate the need for artificial replacement
teeth. Long-term evaluation in patients without clefts has
proved the superiority of orthodontic space closure over restorative treatment (Nordquist and McNeill, 1975). Although
the conditions for this to be feasible are not always present
multiple missing teeth, poor maxillary growth (Bergland et al.,
1986a, 1986b; Ramstad and Semb, 1997), when attempted,
most authors have reported high rates of success. Turvey et al.
(1984) reported that in a sample of 24 patients evaluated after
final orthodontic treatment, 50% had the space for missing
lateral incisors closed by aligning the cuspid next to the central
incisor. One of the largest samples of bone-grafted cleft sites
examined reported a 90% success rate in closing space when
grafts were placed before cuspid eruption (Bergland et al.,
1986a). It is likely that a major reason for the frequent ability
to close spaces relates to a corresponding high rate of natural
cuspid eruption through the graft. Although an earlier study
by El Deeb et al. (1982) reported only a 27% spontaneous
eruption rate for cuspids, other reports since then have shown
consistently higher rates: Troxell et al (1982) 295%; Turvey
et al. (1984) 297%; Enemark et al. (1985) 292%; Bergland
et al. (1986a) 285%; Long et al. (1995) 295%. It is logical
that the normal mesial eruption path of the cuspid, combined
533-8
with a missing lateral incisor, would lend itself to a spaceclosing, cuspid-substitution approach to treatment.
In the minority of cases in which space closure is not possible, the use of adhesive bridgework has become a treatment
of choice (Ramstad, 1998) because of the additional root support for abutment teeth provided by the bone graft (Turvey et
al., 1984). However, the use of implants in the grafted alveolar
ridge has been considered (Rygh and Tindllund, 1996; Vig et
al., 1996; Lilja et al., 1998). Several case reports of implants
to a grafted cleft site have been published (Verdi et al., 1991;
Takahashi et al., 1997). The longest follow-up (3 years), a case
series of 14 is that of Kearns et al. (1997). These authors
reported a 90% success rate of implants in grafted cleft alveolar ridges. Interestingly, all were late secondary grafts in the
permanent dentition, and 6 of the 14 patients required augmentation grafts to provide adequate bone in the cleft site.
Also of note was the fact that the longer the time span between
graft and implant placement, the more likely was the need for
augmentation. From this it would appear that mixed dentition
grafts, unoccupied by a mesially erupting cuspid, would most
likely be unsuitable for implants without first undergoing ridge
augmentation close to the time of implant placement.
Another possibility that has been reported is transplantation
of a lower premolar to the upper arch (Hillerup et al., 1987;
Ramstad and Semb, 1997; Semb and Schwartz, 1997), best
results being reported when root development is one-half to
three-quarters finished. Long-term results of teeth transplanted
in patients without clefts have been very favorable (Schwartz
et al., 1985a, 1985b; Andreasen et al., 1990a, 1990b, 1990c,
1990d).
Residual Transverse Deficiencies
In spite of earlier management of the transverse problems
described above, the possibility of encountering remaining
problems with maxillary constriction and inadequate buccal
overjet have been mentioned by almost all of the authors cited
above. Of primary importance since the advent of bone grafting is the decreasing ability to carry out pure skeletal segmental expansion once the alveolar cleft is repaired. Long (1995)
reported successful separation of the remaining interpremaxillary portion of the midpalatal suture using rapid jack-screw
expansion in the permanent dentition of unilateral clefts. The
same approach taken with bilateral clefts was found to be unsuccessful, possibly related to early obliteration of the interpremaxillary suture in the bilateral condition (Delaire and Precious, 1986). Thus, it is critical to complete as much segment
expansion in the mixed dentition prior to bone grafting as possible.
Unfavorable Class III Skeletal Pattern
In spite of the improvements in facial growth results seen
in the past 60 years, Rosss (1987a, 1987b, 1987c, 1987d)
report based on 1600 lateral cephalometric radiographs from
15 centers worldwide still suggests an incidence of maxillary
retrusion requiring end-stage orthognathic surgery of approximately 25%. Similarly, Mars et al. (1992) in their evaluation
of dental study models of 9-year-old patients from six different
centers found a range of 10% to 50% of patients showing clear
indications of requiring eventual orthognathic surgery, depending on the individual center. While these percentages represent an improvement over the percentages that would have
been found in the 1940s and 1950s, they still represent a significantly greater incidence than that found in the noncleft population (1%). Fortunately, the development of effective orthognathic surgical techniques in the 1970s and 1980s, mentioned earlier in this review, has provided orthodontics with
the means to complete treatment of almost all cases with a
nonprosthodontic approach (Posnick et al., 1990; Turvey et
al., 1996; Wolford and El Deeb, 1998).
The specifics of the evolution of these surgical techniques
is outside the scope of this review. The publications most relevant to orthodontic treatment planning relate to those evaluating the stability of maxillary advancement in patients with
clefts (Posnick and Ewing, 1980; Stoelinga et al., 1987), and
improved methods of predicting hard and soft tissue changes
following surgery. Most surgeons have concluded that stability
following advancement is more problematic than for the noncleft population. However, the use of rigid internal fixation and
block bone grafts has reduced the instability of this procedure
(Waldrop and Wolford, 1989). The use of three-dimensional
cephalometrics and computed tomography scanned dental
models (Cutting et al., 1986; Grayson, 1990), video imaging
(Sarver et al., 1988; Sinclair et al., 1995), and computer-generated images (Gateno et al., 2000) have all improved the precision of surgical planning for hard tissue movements and the
predictability of soft tissue response. Although initially developed for noncleft orthognathic surgery, the application of these
methods to the cleft population has increased quickly.
Finally, with development of distraction osteogenesis and its
application to maxillary deficiency, described above, many
treatment-planning issues remain concerning the choice of orthognathic surgery versus distraction as the end-stage procedure of choice. While recent publications have demonstrated
the results possible with distraction (Figueroa and Polley,
1999) and a number of symposia have been held on the subject, there has been little attempt to develop differential diagnostic and treatment planning criteria, which would identify
those maxillary deficient patients that would be best treated
with routine orthognathic surgery versus distraction osteogenesis.
SUMMARY
The progress and evolution of orthodontic treatment for patients with complete clefts has taken a slow and sometimes
circuitous route to present-day standards. Undoubtedly, much
of our acquisition of new knowledge and establishment of a
sound scientific basis have been undermined and delayed because of the failure by care providers to submit to the rigors
of established procedures for clinical trials (Pocock, 1983;
Roberts et al., 1991; Shaw et al., 1996). As a result, we find

ourselves today still debating the relative merits of different
treatment approaches (e.g., infant orthopedics, timing and sequencing of alveolar repair and orthodontic intervention) that
have been in existence for more than enough years to have
reached concrete conclusions by now had the appropriate documentation and protocols been carried out. It is singularly representative of this problem that the debate over the relative
benefits of presurgical infant orthopedics, which was highlighted by Pruzansky in 1964, has only recently been addressed
using a randomized control trial approach (Kuijpers-Jagtman
and Prahl, 1996; Kuijpers-Jagtman and Prahl-Andersen, 1997).
Nonetheless, in spite this history of slow and inefficient scientific and clinical progress, review of the literature of the past
60 years does, in fact, reveal that progress has been made. The
preceding review has attempted to highlight the major milestones in that progress and the very clear improvements that
have been made in cleft management in general and cleft orthodontic treatment in particular. It is also noteworthy that our
progress has been not only in the area of improved surgical
and orthodontic techniques but also relative to the scientific
weaknesses of past decades. The investigations of more recent
years seem to be moving in a direction of better documentation, stricter methodologies, longer-term follow-up, larger sample sizes, etc.
The research shortcomings of our orthodontic predecessors
can be partially overlooked because of the purely clinical nature of their involvement at the time. However, it can no longer
be considered acceptable to engage in the orthodontic treatment of patients with clefts, without considering clinical practice to be part of an ongoing critical evaluation, the results
from which should allow us to accelerate our rate of progress
in the future. Only by adopting a more critical approach will
protocols be defined that can achieve optimal outcomes while
minimizing the burden of the orthodontic treatment through
elimination of superfluous intervention.
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STATE OF THE ART

Orthodontic Treatment of the Patient With Complete Clefts of Lip,

While orthodontics, as a specialty of dentistry, dates to the

Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6

the initial enthusiasm for this surgical and orthopedic approach

Long et al., ORTHODONTIC TREATMENT

age as the cleft maxilla fails to maintain normal growth rates.

Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6

Vertical Skeletal Dysplasias

manent canines adjacent to cleft sites has been shown to occur

Benefit Versus Burden

Long et al., ORTHODONTIC TREATMENT

agement of numerous cleft-related orthodontic problems

Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6

discrepancies and dental irregularities mentioned above are

This would seem to indicate that management of maxillary

Long et al., ORTHODONTIC TREATMENT

Maxillary Protraction Orthopedics

Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6

Long et al., ORTHODONTIC TREATMENT

Roberts et al., 1991; Shaw et al., 1996). As a result, we find

Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6

Multidisciplinary Management of Cleft Lip and Palate. Philadelphia: WB

Long et al., ORTHODONTIC TREATMENT

Cleft PalateCraniofacial Journal, November 2000, Vol. 37 No. 6

Pruzansky S, Aduss H. Arch form and the deciduous occlusion in complete

Clinical Standards Advisory Group (CSAG) Study: part 1background and

Long et al., ORTHODONTIC TREATMENT

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