An Uncommon Cleft Subtype of Unilateral Cleft Lip and Palate

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					                           Journal of Dental Research

                       An Uncommon Cleft Subtype of Unilateral Cleft Lip and Palate
T. Yamanishi, C. Kobayashi, I. Tsujimoto, H. Koizumi, S. Miya, Y. Yokota, R. Okamoto, S. Iida, T. Aikawa, H. Kohara, J.
                                                 Nishio and M. Kogo
                                              J DENT RES 2008 87: 164
                                         DOI: 10.1177/154405910808700212

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T. Yamanishi1*, C. Kobayashi1,2,
I. Tsujimoto1, H. Koizumi1, S. Miya1,                                    An Uncommon Cleft Subtype
Y. Yokota1, R. Okamoto1, S. Iida1,
T. Aikawa1, H. Kohara2, J. Nishio2,                                      of Unilateral Cleft Lip and Palate
and M. Kogo1
1 First
      Department of Oral and Maxillofacial Surgery,
Graduate School of Dentistry, Osaka University, 1-8
Yamadaoka, Suita, Osaka, Japan; and 2Department of Oral
and Maxillofacial Surgery, Osaka Medical Center and
Research Institute for Maternal and Child Health, 840
Murodocho, Izumi, Osaka, Japan; *corresponding author,
J Dent Res 87(2):164-168, 2008
                                                                         Among variance in the anatomical relationshipand the vomervomer we can
                                                                             see a
                                                                                   individuals with unilateral cleft lip    palate (UCLP),
                                                                                                                         between the
                                                                         secondary hard palate. In most persons with UCLP,
                                                                                                                                             and the
                                                                                                                                      is attached to
                                                                         the secondary palate, being indicative of unilateral cleft in the secondary
ABSTRACT                                                                 hard palate (u-UCLP). In some people with UCLP, the vomer is detached
The finding that the vomer plays a crucial role in                       from the secondary hard palate, that is, these individuals present with
maxillary growth suggests that the bilateral cleft                       bilateral clefts in the secondary hard palate (b-UCLP). Although cleft lip
configuration of unilateral cleft lip and palate                         and palate is one of the most common congenital diseases affecting oral
(UCLP), in which the vomer is detached from the                          functions and facial development, little attention has been paid to this
non-cleft-side secondary hard palate, negatively                         uncommon cleft subtype in UCLP.
influences palatal development, and this                                      The vomer, which exists inferior to the nasal septum, is known to play
hypothesis was tested. Sixty persons with                                an essential role in antero-posterior development of the palate (Friede,
complete UCLP, including those with the vomer                            1998). It has been reported that, in beagle pups, partial or entire resection of
detached from (n = 30, b-UCLP) and attached to                           the vomer significantly reduces antero-posterior maxillary growth (Squier et
(n = 30, u-UCLP) the secondary hard palate, were                         al., 1985). Several studies have revealed that surgical procedures for cleft
analyzed morphologically, with the use of cast                           palate with the use of a vomer flap have a worse influence on the maxillary
models taken at 10 days, 3 mos, and 12 mos of                            growth than those without a vomer flap (Delaire and Precious, 1985; Friede
age. The anterio-posterior palatal length at 12 mos                      and Lilya, 1994; Tanino et al., 1997). Based on these investigations, we
of age in those with b-UCLP was significantly                            hypothesized that the bilateral cleft configuration in the secondary palate of
shorter than that in those with u-UCLP, by 8.7%                          persons with UCLP negatively influences palatal development.
(p < 0.05). In addition, palatal width development                            The bilateral cleft subtype of UCLP has also been little considered in the
in the first year in those with b-UCLP was also                          classification of cleft lip and/or palate. Many studies have developed
significantly retarded. These results suggest that                       classifications and representative methods for cleft lip and/or palate
the uncommon bilateral cleft subtype in UCLP                             (Kernahan and Stark, 1958; Friedman et al., 1991; Schwartz et al., 1993;
should be included in the cleft classification.                          Mortier et al., 1997), but most have omitted this cleft subtype. Recently,
                                                                         Ortiz-Posadas et al. (2001) proposed a classification for clefts that includes
KEY WORDS: UCLP, cleft subtype, palatal                                  a representation of bilateral cleft in the secondary hard palate. However,
development.                                                             they intended to apply the category to persons with cleft palate alone, and
                                                                         did not mention the UCLP category including the bilateral cleft subtype.
                                                                              The aim of this study was to evaluate the palatal morphology of persons
                                                                         with b-UCLP and compare it with that of persons with u-UCLP. If those
                                                                         with b-UCLP possess characteristic features in the palatal developmental
                                                                         pattern, it is necessary for clinicians and researchers to clearly recognize this
                                                                         bilateral cleft subtype of UCLP.

                                                                         MATERIALS & METHODS
                                                                         Sixty persons with complete UCLP were enrolled into this retrospective study.
                                                                         They were non-syndromic Japanese who had no known anomaly other than
                                                                         UCLP, and consecutive persons who were referred to two facilities—the First
                                                                         Department of Oral and Maxillofacial Surgery, Osaka University Graduate
Received December 25, 2006; Last revision July 16, 2007;                 School of Dentistry (Facility A), and the Department of Oral and Maxillofacial
Accepted October 17, 2007                                                Surgery, Osaka Medical Center and Research Institute for Maternal and Child

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                                                             International and American Associations for Dental Research
J Dent Res 87(2) 2008                                   An Uncommon Cleft Subtype of UCLP                                                                        165

Health (Facility B)—from March, 1996, to July, 2000. We divided
them into two groups according to the morphological
characteristics between the vomer and the secondary hard palate
(Fig. 1): whether the vomer was detached from (b-UCLP group) or
attached to (u-UCLP group) the non-cleft-side secondary hard
palate. The b-UCLP group consisted of 30 children (15 from
Facility A, 15 from Facility B; 18 boys, 12 girls; (cleft side) 9
right, 21 left). The u-UCLP group was comprised of 30 children
(15 from Facility A, 15 from Facility B; 17 boys, 13 girls; (cleft
side) 11 right, 19 left). Although the treatments were carried out in
the two facilities independently, all persons were treated under the
same protocol as described below. Briefly, they underwent early
pre-surgical orthopedic treatment (Hotz and Gnoinski, 1979). The
cleft lip was closed at 3 mos of age, according to the modified
Millard method, by two oral and maxillofacial surgeons (MK from
Facility A; JN from Facility B). The birthweight, the age at which
early orthopedic treatment commenced, and the age at which the
participants received cheiloplasty showed no statistical differences                         Figure 1. Oral photographs and schematic drawings of cleft subtypes in
between the b-UCLP and u-UCLP groups (p = 0.39, p = 0.35, and                                UCLP. The pictures show palatal configurations of persons with left-side
p = 0.31, respectively) and between the two facilities (p = 0.25, p =                        complete UCLP. A difference exists in the relationship between the vomer
                                                                                             and secondary hard palate (black and white arrows). In the left picture
0.34, and p = 0.19, respectively). This study was approved by the                            and drawing, the vomer (shaded portion) attaches to the non-cleft-side
Ethical Committee of Osaka University Graduate School of                                     (right side) secondary hard palate (black arrow) (unilateral cleft in the
Dentistry and Osaka Medical Center and Research Institute for                                secondary palate; u-UCLP). In the person on the right, we can recognize
Maternal and Child Health.                                                                   a cleft between the vomer and right-side secondary hard palate, namely,
                                                                                             that there are bilateral clefts in the secondary hard palate (white arrow)
Measurements                                                                                 (b-UCLP). The vomer in persons with b-UCLP attaches only to the primary
                                                                                             hard palate. UCLP = unilateral cleft lip and palate.
We measured palatal dimensions using a consecutive series of
dental cast models taken of study participants at 10 days (Stage 1),
3 mos (Stage 2), and 12 mos (Stage 3) of age. The ages at which
the cast models were taken showed no statistical difference
                                                                                             t test. P values < 0.05 were considered statistically significant. All
between the two groups (Stage 1, p = 0.36; Stage 2, p = 0.31; Stage
                                                                                             results are expressed as means ± SD.
3, p = 0.32) and between the two facilities (Stage 1, p = 0.34; Stage
2, p = 0.19; Stage 3, p = 0.21). We set the reproducible reference
mucosal points on the cast models according to previous studies                              RESULTS
(Kramer et al., 1996; Schliephake et al., 2006) and measured                                 Of the 315 persons with UCLP referred to the two facilities
according to the following parameters (Fig. 2): antero-posterior                             from March, 1996, to July, 2000, we found 30 individuals with
length and transverse widths of the alveolar arch, segment and cleft                         bilateral cleft subtype. The occurrence of the cleft subtype was
widths, palatal heights (the distance between the levels of the cleft                        9.5%.
edges and the alveolar ridges), and slopes of palatal shelves. All of                            In Stage 3, the b-UCLP group showed less antero-posterior
the measurements at stage 1 showed no significant differences                                alveolar arch length (23.1 ± 1.6 mm) than did the u-UCLP
between individuals from the two facilities. Since comparison of                             group (25.4 ± 1.4 mm), by 8.7% with statistical significance (p
treatment outcomes between the two facilities was not the purpose                            < 0.05) (Table). Changes in arch length for 9 mos after
of this study, we did not draw statistical comparisons of                                    cheiloplasty (from Stage 2 to Stage 3) were 0.2 ± 1.4 mm
measurements at stages 2 and 3 between
the facilities.
     All measurements were performed
by two observers with a three-
dimensional measurement system (QM
Measure, MITSUTOYO Co., Kasugai,
Japan). In each model, series were
measured twice by each observer, with
a two-week interval between
measurements. All models of one
participant were measured in one
session. No statistical differences were
found in variable measurements
between the two measurement times by
each observer.
Mean values from different ages of the             Figure 2. Mucosal points and measurements on a dental cast and measurements.
two groups were compared by Student's
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                                                            International and American Associations for Dental Research
166                                                                        Yamanishi et al.                                                           J Dent Res 87(2) 2008

                                                                                                                                            however, no significant differences
                                                                                                                                            were seen in the changes in slope
                                                                                                                                            of both side palatal shelves from
                                                                                                                                            Stage 1 to Stage 3 (major side; p =
                                                                                                                                            0.09, minor side; p = 0.37) (Table).

                                                                                                           This is the first study to analyze
                                                                                                           the palatal morphology of an
                                                                                                           uncommon bilateral cleft subtype
                                                                                                           in persons with UCLP (b-UCLP).
                                                                                                           We demonstrated that persons with
                                                                                                           the bilateral cleft subtype of UCLP
                                                                                                           show more retarded palatal
                                                                                                           development, particularly in
                                                                                                           antero-posterior palatal develop-
                                                                                                           ment and the width of the major
                                                                                                           segment, than did UCLP persons
                                                                                                           with unilateral cleft in the
                                                                                                           secondary hard palate (u-UCLP)
                                                                                                           during the first 12 mos of age.
                                                                                                           Furthermore, the occurrence of the
                                                                                                           bilateral cleft subtype was 9.5% in
                                                                                                           our study, indicating that persons
Figure 3. Comparisons of palatal development in antero-posterior length and transverse width. Note that    with the bilateral cleft subtype are
development in the arch length from Stage 2 to Stage 3 (for 9 mos after cheiloplasty) in b-UCLP (n = 30)
was significantly less than in u-UCLP (n = 30). Although there was no significant difference in TT´
                                                                                                           not uncommon. Dental clinicians
between the two groups at all ages, the development in TT´ of b-UCLP (n = 30) from Stage 1 to Stage 3      should clearly recognize this cleft
(12 mos after birth) was significantly less than that in u-UCLP (n = 30) [u-UCLP, 4.0 ± 2.8 mm (12.9%);    subtype of UCLP in both clinical
b-UCLP, 1.7 ± 2.5 mm (5.2%), p < 0.05].                                                                    practice and research.
                                                                                                               The antero-posterior length in
                                                                                                           persons with b-UCLP at the age of
                                                                                                           12 mos was smaller than that in
(0.9%) in b-UCLP and 1.6 ± 1.4 mm (6.7%) in u-UCLP (p <                       persons with u-UCLP by 8.7%. Moreover, compared with
0.05) (Fig. 3). The alveolar arch lengths in stages 1 and 2                   persons with u-UCLP, the antero-posterior development of the
showed no significant differences between the two groups.                     alveolar arch 9 mos after cheiloplasty was significantly
    There were no significant differences between the two                     impeded in persons with b-UCLP. These results suggest that
groups in all measurements involved in maxillary arch width                   persons with b-UCLP possess less potential in antero-posterior
throughout the ages measured, although the increment in TT´                   palatal development and/or less resistibility against the
from Stage 1 to Stage 3 was 1.7 ± 2.5 mm (5.2%) in b-UCLP                     backward force of oral muscles reconstructed by cheiloplasty
and 4.0 ± 2.8 mm (12.9%) in u-UCLP, indicating significant                    than do persons with u-UCLP. Many studies have reported that
difference between the two groups (p < 0.05) (Fig. 3). In stages              the vomer plays an important role in antero-posterior maxillary
2 and 3, the width of the major segment in b-UCLP (stage 2;                   growth (Delaire and Precious, 1986; Friede, 1998). Vomer
10.6 ± 1.8 mm, stage 3; 12.1 ± 1.4 mm) was significantly                      resection resulted in severe developmental deterioration in the
smaller than that in u-UCLP (stage 2; 12.8 ± 1.5 mm, stage 3;                 antero-posterior length of the maxilla in dogs (Wada et al.,
14.8 ± 1.2 mm) (P < 0.05), whereas the widths in the minor                    1980, 1990). It is well-known that palatal surgical procedures
segment showed no significant differences between the two                     with involvement of the vomer negatively influence maxillary
groups in all stages (Table). Increments in major side-segment                development (Delaire and Precious, 1985; Friede and Lilja,
width from Stage 1 to Stage 3 were 2.6 ± 2.6 mm (27.4%) in b-                 1994; Tanino et al., 1997). These investigations have supported
UCLP and 3.6 ± 2.2 mm (33.0%) in u-UCLP, indicating no                        the notion that the vomer-premaxilla suture is a developmental
significant difference (p = 0.11) (Table).                                    center of forward development of the maxilla. Meanwhile, our
    In stage 3, u-UCLP showed significantly narrower cleft                    study demonstrated that the anatomical morphology of the
width (8.5 ± 2.1 mm) than b-UCLP (11.2 ± 2.6 mm) (P < 0.05)                   posterior part of the vomer influences antero-posterior
(Table). There were no significant differences in cleft width                 development of the palate. We believe that the posterior part of
between the two groups in stages 1 and 2. Palatal heights                     the vomer also plays an important role in palatal development,
increased gradually with age in both groups, but no significant               as an anchor that mechanically supports growth at the vomer-
differences were seen between the two groups in all stages.                   premaxilla suture. To convey endochondral bone growth at the
Progressive decline in the slopes of major- and minor-side                    vomer-premaxilla suture to the maxilla, the posterior part of the
palatal shelves was recognized in both groups. In stage 3,                    vomer would need to be fixed to the surrounding tissue.
persons with u-UCLP showed a slope on the major side of 35.1                  According to the septal-traction model that explains the
± 5.3°, and those with b-UCLP showed a slope of 41.4 ± 4.5°,                  mechanism of maxillary forward growth at an early
with a significant difference between them (P < 0.05);                        age, the forward and downward growth of the nasal septum
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                                                            International and American Associations for Dental Research
J Dent Res 87(2) 2008                                    An Uncommon Cleft Subtype of UCLP                                                             167

pulls the mid-face             Table. Summary of Results
forward via the septo-
premaxillary ligament                                     u-UCLP            b-UCLP       Segment Width                 u-UCLP          b-UCLP
(Mooney and Siegel,                                   mean ± SD, mm mean ± SD, mm                                  mean ± SD, mm mean ± SD, mm
1986; Siegel et al.,
1990). It also seems           A-TT´         Stage 1 23.4 ± 1.6           22.4 ± 2.1               Major Stage 1 10.9 ± 1.6           9.5 ± 1.9
necessary for the vomer                      Stage 2 23.9 ± 1.8           22.8 ± 2.4                       Stage 2 12.8 ± 1.5        10.6 ± 1.8*
to be fixed to the                           Stage 3 25.4 ± 1.4           23.1 ± 1.6*                      Stage 3 14.8 ± 1.2        12.1 ± 1.4*
secondary hard palate to                                                                           Minor Stage 1 10.5 ± 1.6           8.9 ± 1.5
convey the forward and         C-C´          Stage 1 30.1 ± 2.4           30.8 ± 2.8                       Stage 2 12.6 ± 1.8        11.7 ± 1.8
downward growth of                           Stage 2 30.8 ± 2.4           31.2 ± 2.5                       Stage 3 13.1 ± 2.0        12.4 ± 1.3
the nasal septum                             Stage 3 30.9 ± 2.3           31.3 ± 3.5
cartilage to the maxilla.                                                                  Palatal Height
Detachment of the              M-M´          Stage 1 35.3 ± 2.3           34.3 ± 3.2               Major Stage 1 8.1 ± 1.1            8.5 ± 2.3
vomer       from       the                   Stage 2 36.4 ± 1.4           35.9 ± 3.9                       Stage 2 9.2 ± 1.6          9.3 ± 1.8
secondary hard palate                        Stage 3 37.2 ± 1.9           36.8 ± 2.7                       Stage 3 10.4 ± 2.0        10.9 ± 1.8
possibly disturbs the                                                                              Minor Stage 1 8.9 ± 1.5            9.0 ± 2.0
sliding growth between         T-T´          Stage 1 31.1 ± 2.2           32.9 ± 3.1                       Stage 2 9.8 ± 1.2         10.5 ± 1.9
the vomer and the                            Stage 2 33.7 ± 1.9           33.8 ± 3.3                       Stage 3 10.8 ± 1.6        11.6 ± 1.4
maxilla that is observed                     Stage 3 35.3 ± 1.9           34.4 ± 3.4
in early childhood                                                                  Slope of Palatal Shelf
(Friede,           1998).      Cleft width Stage 1 12.9 ± 2.4             13.6 ± 2.5               Major Stage 1 39.1 ± 4.3          43.2 ± 5.3
Nevertheless, we must                        Stage 2 10.3 ± 2.3           11.9 ± 3.0                       Stage 2 38.1 ± 4.5        42.0 ± 6.1
closely follow the                           Stage 3     8.5 ± 2.1        11.2 ± 2.6*                      Stage 3 35.1 ± 5.3        41.4 ± 4.5*
palatal development of                                                                             Minor Stage 1 45.2 ± 6.1          44.6 ± 5.1
UCLP persons with                                                                                          Stage 2 42.8 ± 5.9        43.2 ± 5.5
bilateral cleft in the                                                                                     Stage 3 39.5 ± 5.4        42.5 ± 5.1
secondary hard palate,
because they have the          The Table shows the measurement results (u-UCLP, n = 30; b-UCLP, n = 30). Asterisks indicate statistical differences
                               between the two groups (p < 0.05). Note that antero-posteror length at Stage 3 in persons with b-UCLP was
potential to develop           significantly shorter than that in persons with u-UCLP, by 8.7%. In addition, the less major segment width in b-UCLP
severe crossbite in the        (Stages 2, 3) caused a wider cleft width in b-UCLP (Stage 3), indicating that transverse development in the major
permanent dentition.           segment of b-UCLP is also more impaired than that of u-UCLP. Major = non-cleft side, minor = cleft side, A-TT´ =
    Individuals with b-        antero-posterior length of the major segment, CC´ = primary inter-canine width (without teeth, width between
UCLP        showed        a    intersections of lateral sulcus and the alveolar ridge line on both sides), MM´ = primary inter-second molar width
                               (without teeth, the maximum arch width), TT´ = inter-tuberosity width.
narrower width in the
major segment at the
ages of 3 and 12 mos,
and larger cleft width at
12 mos of age, than did those with u-UCLP. In addition, the                   UCLP. Further study is necessary to explain the phenomenon
slope in the major side palatal shelf of those with b-UCLP was                observed in our study.
steeper than that of those with u-UCLP at the age of 12 mos.                      Many studies have attempted to classify and represent cleft
Our results also demonstrated that the development of inter-                  lip and palate since the embryological classification by
maxillary tuberosity width for the first 12 mos is larger in                  Kernahan and Stark (1958). Many clinicians now use a
individuals with u-UCLP than in those with b-UCLP. These                      composite schematic representation based on the 'striped Y'
results suggest that the vomer is involved in transverse palatal              approach. Recently, classifications that include a scoring
development. The configuration in which the vomer attaches to                 concept in which the severity of the anatomical deformity is
the secondary hard palate would enhance inward growth of the                  graded to predict the results of surgical treatments have been
cleft margin on the major segment. The connection between the                 developed (Friedman et al., 1991; Schwartz et al., 1993;
vomer and the osseous palate is made by the footplate of the                  Mortier et al., 1997). Few classifications of cleft lip and palate,
vomer, which supports the lower part of the vomer and initiates               however, are able to represent an anatomical configuration of
ossification at late gestational age (Sandikcioglu et al., 1994). It          the secondary palate of persons with UCLP. To the best of our
has been reported that the development of the footplate is                    knowledge, only one representation method can represent
dependent on the coalescence of the soft-tissue palatal shelves               bilateral cleft configuration in the secondary hard palate of
with the nasal septum (Hansen et al., 2004). Moreover, since                  persons with UCLP (Ortiz-Posadas et al., 2001). In the
the lower part of the vomer presents a marked deviation toward                secondary palate section of their classification, those authors
the side at which articulation occurs between the vomer and                   described bilateral and unilateral clefts, although they made no
osseous palate in persons with UCLP (Kimes et al., 1992), a                   mention of the bilateral cleft subtype in UCLP. Based on the
possible development at the footplate should push and widen                   results described here, we believe that it is crucial for a
the major side osseous plate laterally. Development at the                    classification of clefts to possess the ability to represent
vomeral footplate may explain the better transverse palatal                   configurations in the secondary hard palate.
development in persons with u-UCLP than in those with b-                          In our study, we restricted participants to those with UCLP,

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                                                             International and American Associations for Dental Research
168                                                                             Yamanishi et al.                                                           J Dent Res 87(2) 2008

but we can also see such a cleft subtype in the secondary hard                                        in coordination with delayed surgery for cleft lip and palate. J
palate in persons with cleft palate only or submucous cleft                                           Maxillofac Surg 7:201-210.
                                                                                                  Kernahan DA, Stark RB (1958). A new classification for cleft lip and cleft
palate. It has been reported that, in 53% of persons with
                                                                                                      palate. Plast Reconstr Surg Transplant Bull 22:435-441.
submucous cleft palate, the vomer did not fuse with the palatal                                   Kimes KR, Mooney MP, Siegel MI, Todhunter JS (1992). Growth rate of
shelves up to the incisive foramen (Grzonka et al., 2001).                                            the vomer in normal and cleft lip and palate human fetal specimens.
Further study is necessary to reveal the pattern of palatal                                           Cleft Palate Craniofac J 29:38-42.
development in persons with cleft palate alone or with                                            Kramer GJ, Hoeksma JB, Prahl-Andersen B (1996). Early palatal changes
submucous cleft palate.                                                                               after initial palatal surgery in children with cleft lip and palate. Cleft
                                                                                                      Palate Craniofac J 33:104-111.
                                                                                                  Mooney MP, Siegel MI (1986). Developmental relationship between
ACKNOWLEDGMENTS                                                                                       premaxillary-maxillary suture patency and anterior nasal spine
The authors thank Dr. Takeshi Wada and Dr. Kanji Nohara for                                           morphology. Cleft Palate J 23:101-107.
valuable advice. This research was supported by the Ministry                                      Mortier PB, Martinot VL, Anastassov Y, Kulik JF, Duhamel A, Pellerin
                                                                                                      PN (1997). Evaluation of the results of cleft lip and palate surgical
of Education, Science, Sports and Culture, Grant-in-Aid for
                                                                                                      treatment: preliminary report. Cleft Palate Craniofac J 34:247-255.
Scientific Research (B) (17390535, 2005), and the 21st Century                                    Ortiz-Posadas MR, Vega-Alvarado L, Maya-Behar J (2001). A new
COE entitled "Origination of Frontier BioDentistry" at Osaka                                          approach to classify cleft lip and palate. Cleft Palate Craniofac J
University Graduate School of Dentistry, supported by the                                             38:545-550.
Ministry of Education, Culture, Sports, Science and                                               Sandikcioglu M, Molsted K, Kjaer I (1994). The prenatal development of
Technology.                                                                                           the human nasal and vomeral bones. J Craniofac Genet Dev Biol
                                                                                                  Schliephake H, Donnerstag F, Berten JL, Lonquist N (2006). Palate
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                                                                 International and American Associations for Dental Research