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J Oral Maxillofac Surg67:477-484, 2009

Does Congenital Cleft Palate IntrinsicallyInfluence Craniofacial Morphology?:Craniofacial Features in Unoperated

Submucous Cleft Palate Childrenin Prepuberty

Hiroshi Iwasaki, DDS, PhD,* Motonori Kudo, DDS, PhD,† and

Yuko Yamamoto, SLHTs‡

Purpose: The aberrations in facial structure with concave midface are a well-recognized featurecharacterizing cleft palate patients. Several explanations have been given as to the aberration causes,including surgical interference or the primary tissue deficiency in the congenital cleft. The aim of thisstudy was to determine intrinsic effects of congenital cleft palate on craniofacial morphology byretrospectively comparing craniofacial features between children with unoperated submucous cleftpalate and noncleft children with normal occlusion in prepuberty.

Patients and Methods: Twelve Japanese children (7 girls and 5 boys) with unoperated submucouscleft palate at age 9 were examined cephalometrically. None of them had undergone any orthodontictreatments. Their craniofacial morphologies were compared with those in 60 Japanese noncleft children(30 girls and 30 boys) with normal occlusion at age 9.

Results: In cleft children, anteroposterior length of the maxilla was significantly short, and theposterior part of the maxilla was more in the anterior position compared with noncleft children. Also,the anterior parts of the maxilla tended to be slightly retruded in cleft children.

Conclusion: In the current study, we recognized characteristic differences in the craniofacial morphol-ogy between the unoperated submucous cleft palate children and the noncleft children in prepuberty,and showed that the craniofacial deviation in the cleft children can be defined as the intrinsic effects ofcongenital cleft palate itself.© 2009 American Association of Oral and Maxillofacial Surgeons

J Oral Maxillofac Surg 67:477-484, 2009

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he aberrations in facial structure with concave mid-ace, which Graber1 described for the first time in949, are a well-recognized feature characterizingleft palate patients. It is said that the characteristicsf the aberrations depend on the cleft types,2-4 andeveral explanations have been given as to the aber-ation causes, including surgical interference or therimary tissue deficiency in the congenital cleft.5

hus, there seem to be a number of determinant

eceived from Hokkaido University Hospital, Hokkaido University,

apporo, Japan.

*Assistant Professor, Department of Orthodontics, Dental Clinical

ivision.

†Lecturer, Division for General Dentistry.

‡Assistant Professor, Speech Clinic, Dental Clinical Division.

Address correspondence and reprint requests to Dr Iwasaki:

epartment of Orthodontics, Dental Clinical Division, Hokkaido

477

actors for the aberrations, namely, the characteristicsf the craniofacial morphology of patients with cleftalate. The main problem in the analysis of those

actors is that in many cases time lapses after comple-ion of cleft repair are so long because the repair isone in infancy as a rule.6 Therefore, the characteris-ics of craniofacial morphology to be observed areombined results of intrinsic effects of cleft and iat-ogenic effects, which confuses our inference of the 2

niversity Hospital, Hokkaido University, Kita 14-jo Nishi 5-chome,

ita-ku, Sapporo, 060-8648, Japan; e-mail: hiro-iwa@den.hokudai.

c.jp

2009 American Association of Oral and Maxillofacial Surgeons

278-2391/09/6703-0003$36.00/0

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478 DOES CLEFT PALATE INFLUENCE MORPHOLOGY?

ffects, respectively, on craniofacial morphology. Onhe other hand, there remains a controversy in theeparate evaluation of the 2 effects, intrinsic and iat-ogenic, on craniofacial morphology in patients withleft palate. Hence, it would be desirable if we couldevelop a morphogenetic discussion based on thenalysis data of craniofacial morphology of cleft pa-ients where there is no variable brought about byurgical repair.

Aiming at such a discussion, we chose patientsith unoperated submucous cleft palate. All pa-

ients with submucous cleft palate do not haveifficulties in speech, and do not require surgicalreatment.7 Some of them have no difficulties inroducing speech sounds, indicating adequate velo-haryngeal function.8,9 We assumed, therefore, that

t would be possible to determine the intrinsicffects of cleft palate on craniofacial morphologyy comparing patients with cleft palate and non-left control subjects.We evaluated possible relationships between the

resence of cleft palate and craniofacial morphologyy retrospectively comparing the craniofacial featuresf 12 unoperated submucous cleft children in prepu-erty with those in 60 noncleft chlidren with normalcclusion in prepuberty. Our essential aim was toetermine intrinsic effects of congenital cleft palaten craniofacial morphology. We also described aver-ge characteristics of craniofacial morphology of theubmucous cleft palate children.

FIGURE 1. Landmarks and

wasaki, Kudo, and Yamamoto. Does Cleft Palate Influence Morpholog

atients and Methods

Nonsyndromic 12 Japanese children (7 girls and 5oys) with submucous cleft palate were subjected tohe study. All of them had been diagnosed as havingubmucous cleft palate based on the anatomical triadf signs originally proposed by Calnan, namely a bifidvula, a translucent zone in the midline of the softalate, and absence of posterior border of the bonyalate. Articulation and velopharyngeal function werevaluated by a speech therapist (Y.Y.) and an oralurgeon (M.K.). They were regularly followed andiven training for soft blowing and articulation whenecessary, but did not require operation. They hadever undergone any orthodontic treatment, andere in the mixed dentition stage (Hellman’s dental

ge IIIA); their average age was 9.5 years, with a rangerom 9.1 to 9.9 years in both girls and boys. Lateralephalograms were used for the analysis of theirraniofacial morphologies.Data of 60 Japanese noncleft children (30 girls and

0 boys) with normal occlusion at 9 years of age werextracted as a control from the serial records assem-led in the Longitudinal Craniofacial Growth andevelopment Study of Japanese Children (available

t Hokkaido University School of Dentistry), whichad been obtained from children living in Nanporo-ho, Hokkaido prefecture in Japan.10 Lateral cephalo-rams were used in the analysis of noncleft childrens well. Figure 1 shows the cephalometric measure-ents used in the current study. Tracing of all the

es for lateral cephalograms.

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IWASAKI, KUDO, AND YAMAMOTO 479

ephalograms, determining all landmarks and digi-ization of the tracing measurements were done byinvestigator (H.I.). Nine angular and 6 linear vari-

bles were measured, and in addition, 5 of theinear variables were evaluated by standardizing theength of anterior cranial base (SN). An error in the

ethod was assessed by the same investigator, byuplicate tracing of 20 randomly selected lateralephalograms, giving landmarks and digitizinghem at intervals of at least 1 month. Measurementrrors were evaluated by using the Method Errortatistic, which averaged 0.3 mm (SD � 0.1 mm) forinear variables, and 0.4 degrees (SD � 0.1 degree)or angular variables.

Mann-Whitney’s U-test was applied to test the dif-erences in mean values. Probability was evaluated by2-sided test, and values less than .05 were consid-

red statistically significant. For statistical analysis, thePSS statistical program v14.0 (SPSS Inc, Chicago, IL)as used.

esults

Figure 2 shows the mean profilograms of girls andoys, respectively. By superimposing the mean plotsf unoperated submucous cleft plate children andhose of noncleft children on the anterior cranialase, differences in visual impression were examined.n girls, the maxilla was shorter in the unoperatedubmucous cleft palate girls, the anterior parts (ANSnd A) being retruded and the posterior part (PNS=)

IGURE 2. Mean profilograms illustrating the craniofacial morpholhildren (broken line). These profilograms were superimposed oespectively.

wasaki, Kudo, and Yamamoto. Does Cleft Palate Influence Morpholog

eing more in the anterior position; also, the positionf the mandible at the alveolar level (B), the inclina-ion of the mandibular plane, and the gonial angleere same as those of the noncleft control girls. The

ength of cranial base was the same between the 2roups of girls. In boys, the maxilla was shorter, thenterior parts (ANS and A) being retruded and theosterior part (PNS=) being more in the anterior po-ition in the cleft boys; while the mandible retrudedlightly at the alveolar level (B), the inclination of theandibular plane and the gonial angle were the same

s those in the noncleft control boys. Anterior cranialase was short in the cleft boys.Tables 1 and 2 show a cephalometrical comparison

f craniofacial morphology between the unoperatedubmucous cleft palate children and the noncleft chil-ren. Statistical analysis showed that in both the girlsnd the boys with unoperated submucous cleft palatehe posterior part of the maxilla was more in thenterior position (SN-HOL-PNS=, P � .01 in girls; SN-OL-PNS=/SN, P � .01 in girls; NSPNS=, P � 0.05 in

otal of boys and girls), and that in both the cleft girlsnd the cleft boys the maxilla bodies were smallerANS-PNS=, P � .001 in total of girls and boys; ANS-NS=/SN, P � .001 in girls and P � .01 in boys); and

n the cleft girls, the anterior part of the maxilla wasn the posterior position (NS-HOL-ANS/SN, P � .05),nd the value of the anteroposterior jaw relation wasmaller at the alveolar level (ANB, P � .01) in the cleftirls.

noperated submucous cleft palate children (solid line) and noncleftella(S)-nasion(N) line and registered at sella in girls and boys,

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Table 1. CEPHALOMETRIC MEASUREMENTS AT THE AGE OF 9 YEARS IN THE 7 UNOPERATED SUBMUCOUS CLEFT PALATE GIRLS AND THE 30 NONCLEFT GIRLS

VariablesPatient 1

(Girl)Patient 2

(Girl)Patient 3

(Girl)Patient 4

(Girl)Patient 5

(Girl)Patient 6

(Girl)Patient 7

(Girl)

Patients(Girls)

(n � 7)

NoncleftChildren(Girls)

(n � 30)

P ValueMean � SD Mean � SD

SNA (degrees) 83.72 75.04 78.64 82.83 79.30 84.28 78.64 80.35 � 3.12 83.25 � 3.60 0.0525SNB (degrees) 83.54 74.68 76.26 84.50 76.26 79.11 74.70 78.44 � 3.79 78.61 � 2.61 0.7271ANB (degrees) 0.18 0.36 2.38 �1.67 3.04 5.17 3.94 1.91 � 2.22 4.64 � 2.43 0.0052*SNANS (degrees) 87.31 80.91 83.53 87.72 85.82 88.42 84.10 85.40 � 2.50 87.92 � 3.78 0.0549NSPNS= (degrees) 74.39 77.59 77.32 69.61 77.59 76.48 79.15 76.02 � 2.94 79.57 � 3.05 0.0146*SNPP (degrees) 4.26 7.93 6.06 4.05 6.35 6.74 6.31 5.96 � 1.27 7.24 � 2.91 0.1873GZN (degrees) 78.06 89.50 88.48 76.36 88.39 87.85 91.39 85.72 � 5.50 86.08 � 3.20 0.4852SN-Mp (degrees) 33.69 43.42 37.42 31.41 40.80 38.10 40.40 37.89 � 3.88 36.69 � 3.21 0.4380Go angle (degrees) 135.64 133.92 128.94 135.05 132.41 130.25 129.01 132.17 � 2.61 130.61 � 4.24 0.3227SN (mm) 64.75 61.43 61.50 60.76 56.50 61.91 58.28 60.73 � 2.47 60.68 � 2.48 0.9382ANS-PNS= (mm) 48.88 44.17 46.57 44.35 43.74 49.44 43.93 45.87 � 2.26 51.19 � 2.15 0.0002*ANS-PNS=/SN 0.75 0.72 0.76 0.73 0.77 0.80 0.75 0.76 � 0.02 0.84 � 0.04 0.0001*NS-HOL-ANS (mm) 2.45 8.01 5.46 1.71 3.51 1.46 5.45 4.01 � 2.22 1.86 � 3.27 0.0684NS-HOL-ANS/SN 0.04 0.13 0.09 0.03 0.06 0.02 0.09 0.07 � 0.04 0.03 � 0.05 0.0480*SN-HOL-PNS= (mm) 13.56 9.68 9.73 14.80 9.51 11.35 9.18 11.12 � 2.07 8.10 � 2.44 0.0094*SN-HOL-PNS=/SN 0.21 0.16 0.16 0.24 0.17 0.18 0.16 0.18 � 0.03 0.13 � 0.04 0.0066*AUFH (mm) 52.15 50.06 48.17 42.96 48.08 53.02 52.71 49.60 � 3.31 50.25 � 2.29 0.9382AUFH/SN 0.81 0.82 0.78 0.71 0.85 0.86 0.90 0.82 � 0.06 0.83 � 0.04 0.8463PUFH (mm) 48.53 43.97 43.26 39.83 43.24 47.22 47.89 44.85 � 2.91 43.81 � 2.03 0.2951PUFH/SN 0.75 0.72 0.70 0.66 0.77 0.76 0.82 0.74 � 0.05 0.72 � 0.04 0.3323

*Significant differences (P � .05).

Iwasaki, Kudo, and Yamamoto. Does Cleft Palate Influence Morphology? J Oral Maxillofac Surg 2009.

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Table 2. CEPHALOMETRIC MEASUREMENTS AT THE AGE OF 9 YEARS IN THE 5 UNOPERATED SUBMUCOUS CLEFT PALATE BOYS AND THE 30 NONCLEFT BOYS

VariablesPatient 8

(Boy)Patient 9

(Boy)Patient 10

(Boy)Patient 11

(Boy)Patient 12

(Boy)

Patients(Boys)

(n � 5)

NoncleftChildren(Boys)

(n � 30)

P ValueMean � SD Mean � SD

SNA (degrees) 78.42 82.77 81.11 81.83 75.80 79.99 � 2.55 82.87 � 3.87 0.1868SNB (degrees) 76.62 76.36 73.65 82.36 81.85 78.17 � 3.38 78.54 � 3.77 0.7773ANB (degrees) 1.80 6.41 7.46 �0.53 �6.05 1.82 � 4.91 4.33 � 2.28 0.3457SNANS (degrees) 83.11 85.35 84.27 85.38 80.42 83.71 � 1.84 87.50 � 4.14 0.0593NSPNS= (degrees) 75.45 75.84 76.44 78.00 76.70 76.49 � 0.88 79.05 � 2.80 0.0339*SNPP (degrees) 9.69 8.90 7.72 5.25 11.94 8.70 � 2.21 7.01 � 3.38 0.2997GZN (degrees) 82.84 83.96 87.14 83.23 86.25 84.68 � 1.70 84.36 � 3.76 0.9624SN-Mp (degrees) 35.27 39.25 46.28 31.83 29.55 36.44 � 5.91 36.45 � 3.51 0.8504Go angle (degrees) 132.44 135.28 139.14 128.60 123.30 131.75 � 5.46 132.10 � 4.68 0.9249SN (mm) 63.53 58.42 58.12 58.66 58.68 59.48 � 2.03 61.85 � 2.71 0.0533ANS-PNS= (mm) 46.54 45.28 43.31 44.29 40.63 44.01 � 2.00 51.27 � 2.27 0.0005*ANS-PNS=/SN 0.73 0.78 0.75 0.76 0.69 0.74 � 0.03 0.83 � 0.04 0.0011*NS-HOL-ANS (mm) 6.26 3.77 4.88 4.24 8.71 5.57 � 1.78 2.26 � 3.74 0.0593NS-HOL-ANS/SN 0.10 0.06 0.08 0.07 0.15 0.09 � 0.03 0.04 � 0.06 0.0533SN-HOL-PNS= (mm) 11.40 9.92 10.33 10.31 10.22 10.43 � 0.51 8.79 � 2.56 0.1314SN-HOL-PNS=/SN 0.18 0.17 0.18 0.18 0.17 0.18 � 0.00 0.14 � 0.04 0.0593AUFH (mm) 51.74 46.31 48.66 52.53 51.61 50.17 � 2.34 51.33 � 2.20 0.2997AUFH/SN 0.81 0.79 0.84 0.90 0.88 0.84 � 0.04 0.83 � 0.05 0.6374PUFH (mm) 43.91 39.31 42.84 48.48 43.21 43.55 � 2.93 45.08 � 3.17 0.3458PUFH/SN 0.69 0.67 0.74 0.83 0.74 0.73 � 0.05 0.73 � 0.06 0.8875

*Significant differences (P � .05).

Iwasaki, Kudo, and Yamamoto. Does Cleft Palate Influence Morphology? J Oral Maxillofac Surg 2009.

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482 DOES CLEFT PALATE INFLUENCE MORPHOLOGY?

iscussion

It has been reported that the incidence of submu-ous cleft palate is lower than the other cleft types. Inhe current study, none of the examined submucousleft palate children required operation, as mentionedbove, and they were selected to match the age of 9ears. Furthermore, their complete lateral cephalo-rams at the age were available. Taken all these to-ether, we are confident that our criteria to select theaterials were solid enough for this study. Although

he sample size was small (7 girls and 5 boys), thealidity of the present study therefore lies in the facthat we had valuable materials that had been managedy a single multidisciplinary team. Thus, we couldonduct the study to obtain the intriguing findings.It has generally been pointed out that there areethodological limitations to retrospective studies

f craniofacial morphology such as extensive ageifferences in control subjects11 and difficulties inomparing craniofacial morphology between ethnicroups.12 It is also suggested that even within Japa-ese people, geographical differences are recognized

n facial morphology.13 Hence, we must carefullyhoose study subjects to collect valid information. Inhe present study, the noncleft children as well as thenoperated submucous cleft palate children were allt age 9, and from 1 geographical area. In this regard,ur comparison of the data of craniofacial morphol-gy was valid.Among the cephalometric variables, we analyzed

ll the linear variables except SN by standardizing theength of the anterior cranial base (SN), consideringndividual differences in the linear measurements. Inhe current study, no significant difference was statis-ically found in SN between the unoperated submu-ous cleft children and the noncleft children, namely,he 2 groups were found to be statistically equal in SNength. Characteristics of the length of anterior cranialase in patients with cleft palate has been equivo-al.14 Horswell et al15 and Shetye et al16 reported thatn unilateral cleft lip and palate patients and adultnoperated complete unilateral cleft lip and palateatients, the anterior cranial base was shortened in

ength; however, Horswell et al15 alluded that theength of the anterior cranial base in patients withleft palate could not be characterized by their find-ngs. Our finding is supported by the previous stud-es17-20 that, because of the distance from the cleftalate, the defect had no intrinsic effects on the sizer the shape of the cranial base in a patient with cleftalate. Based on this, we suggest that congenitalnomaly in the anteroposterior length of the cranialase is minimal; accordingly it is valid to evaluate the

inear measurements by standardizing the anterior

ranial base. o

Haseda et al21 showed that hypoplasia occurs in theaxilla of a number of submucous cleft palate pa-

ients due to the primary tissue deficiency in theongenital defect. In accordance with Kaplan22 andark et al,23 we observed that the anteroposterior

ength of the maxilla (ANS-PNS=) was significantlyhort in the unoperated submucous cleft palate chil-ren compared with the noncleft children in eitherirls or boys; this finding suggests that the presence ofleft palate itself restricts the anteroposterior size ofhe maxilla in the cleft children. From the finding thathe posterior part of the maxilla was more in thenterior position (NSPNS=) in the unoperated submu-ous cleft children, either girls or boys, we also rec-gnized the effects of cleft palate there.Van Limborph24 showed from his observation of

kulls that the pterygoid processes of the sphenoidone, which are posteriorly approximated to theaxilla, were slightly in the anterior direction in someatients with cleft palate. Furthermore, it was shownhat cleft palate could cause deviation of the muscletructures in the palatopharyngeal area and disturbheir balance.25 More interestingly, Subtelny et al26

xplained that the imbalance between the tensor velialatine muscle and the pterygoid muscles was 1 ofhe causes of deviation of the pterygoid process inleft palate patients, agreeing with the opinions ofoss and Johnston.27 Considering these views, wessume that the effects of muscular function aremong the most important factors involved in ourndings, that is, the restricted size of the maxilla, andhe posterior part of the maxilla being in the anteriorosition. Furthermore, the shortness in the anteropos-erior maxillary length during the growth period, aseen in the current study, is apprehended to causerch length discrepancies in the stage of permanententition, which is another subject we must deal with

n the future.Diah et al28 noted that in adult patients with unop-

rated cleft, tissue deficiency mostly occurs in thenterior part. In the present analysis of the anteropos-erior position of the anterior part of the maxilla, weoticed the part was significantly more in the poste-ior direction (NS-HOL-ANS/SN) in the unoperatedubmucous cleft girls than in the noncleft girls. Noignificant difference in the position of the part wasound in the comparison between the unoperatedubmucous cleft boys and the noncleft boys; how-ver, in comparison with the mean of the noncleftoys, a tendency toward retrusion of the anterior partf the maxilla was recognized: for SNA and SNANS, allf the submucous cleft boys individually showed amall but definite reduction, and for NS-HOL-ANS andS-HOL-ANS/SN, all 5 showed a small but definite

ncrease. These findings indicate that the anterior part

f the maxilla tended to be slightly retruded relative

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IWASAKI, KUDO, AND YAMAMOTO 483

o the anterior cranial base in the unoperated submu-ous cleft children. We speculate that the tendency isttributable to the congenital restriction to forwardrowth of the maxilla in submucous cleft palate chil-ren. Sarnat29 pointed out the importance of endo-hondral growth in the septovomeral region for theorward/downward growth of the maxilla as well asor the growth of the craniofacial complex. In theubmucous cleft palate children in the current study,bsence of the posterior border of the bony palate, aomponent of the septovomeral region, had beenonfirmed. The defect was likely to affect the endo-hondral growth in the septovomeral region. Wepeculate that the tendency of the anterior part of theaxilla being relatively in the posterior direction was

scribed to inadequate growth of the maxilla due tohe adverse effects of the defect on the septovomeralegion.

According to Ross,30 presurgical orthopedics, in-luding Hotz plate, in the neonatal does not bringbout long-term effects for growth and that differ-nces in the method or treatment time of palatalurgery; for example, 1-stage or 2-stage hard or softalate repair, cannot cause variations in the craniofa-ial growth. Instead, he strongly suggests possiblether factors, more influential than those iatrogenicffects, on the craniofacial growth in cleft lip andalate patients. Sarnat31 concluded in his experimentxtirpation of the Median and Transverse Palatineutures in Young Monkeys that extirpation of theedian and transverse palatine sutures did not pro-

uce a grossly apparent growth arrest in either thealate or the face.In the current study, we recognized characteristic

ifferences in the craniofacial morphology betweenhe unoperated submucous cleft palate childrennd the noncleft children in prepuberty, and showedhat the craniofacial deviation in the cleft children cane defined as the natural morphogenetic tendencyffected by the congenital cleft palate itself. Accord-ngly, the larger the defect in the tissue of the palater the lip is, the more conspicuous the tendency is. Itould be reasonable to think that the iatrogenic ef-

ects of palatoplasty are not as strong as we have beenoncerned with, and that the operative methods torevent growth inhibition of palate are not particu-

arly effective. The purpose of palatoplasty is for aleft patient to gain veropharyngeal function for nor-al articulation, and thus the important point is to

hoose the most appropriate operative method andime for that purpose. It is recognized as the congen-tal maxillary shape that the anterior part of the max-lla tend to be in the posterior direction in unoperatedubmucous cleft palate children, because the ten-ency lasts at least unil 9 years of age, it is unreason-

ble to expect to solve the problems of anteroposte-

ior jaw disharmonies by presurgical orthopedicsncluding Hotz plate treatment in the neonatal. Forhe problems, we recommend our method of promot-ng maxillary growth by maxillary protracting asentofacial Orthopedics,32 which we perform posi-

ively for cleft palate patients in our institute.Bishara5 and Liao6 suggested that deficiency in the

rowth potential of the maxilla could be one of theactors for its morphological deviations they actuallybserved in adults with unoperated cleft palates. Al-hough we could not confirm their view in the cur-ent study, we expect that our further studies of therowth potential and morphology would enable us tolucidate craniofacial morphology of cleft palate pa-ients more comprehensively.

We hope that our findings of the intrinsic effects onhe craniofacial structure of unoperated submucousleft palate children would be useful information forhose specializing in treating cleft palate patients, instablishing a guideline on treatment planning forptimal orthodontics or surgical treatment and itsppropriate time, and in qualitatively improving thereatments.

cknowledgments

The authors gratefully acknowledge the statistical suggestion ofr Masahiro Mizuta, DEng, who is the Professor of Theoretical

nformation Science Laboratory, Center for Information and Multi-edia Studies, Hokkaido University.

eferences1. Graber TM: Craniofacial morphology in cleft palate and cleft lip

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484 DOES CLEFT PALATE INFLUENCE MORPHOLOGY?

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3. Kamegai T, Naohiko I: Geographical difference of discrepancyfactor and ecology. J Dent Health 34:476, 1984

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7. Shibasaki Y, Ross RB: Facial growth in children with isolatedcleft palate. Cleft Plate J 6:290, 1969

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