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Incidence of cleft lip or palate in 303738 Japanese babies born between 1994 and 1995
British Journal of Oral and Maxillofacial Surgery (2000) 38, 605–607 © 2000 The British Association of Oral and Maxillofacial Surgeons doi: 10.1054/bjom.2000.0539
BRITISH JOURNAL OF ORAL
& M A X I L L O FAC I A L S U R G E RY
Incidence of cleft lip or palate in 303 738 Japanese babies born between 1994 and 1995 N. Natsume,* T. Kawai,* G. Kohama,† T. Teshima,‡ S. Kochi,‡ Y. Ohashi,§ S. Enomoto,¶ M. Ishii,¶ T. Shigematsu (deceased), Y. Nakano,** T. Matsuya,†† M. Kogo,†† Y. Yoshimura,‡‡ M. Ohishi,§ N. Nakamura,§ T. Katsuki,¶¶ M. Goto,¶¶ M. Shimizu,*** S. Yanagisawa,*** T. Mimura,††† H. Sunakawa‡‡‡ *Aichi-Gakuin University; †Sapporo Medical University; ‡Tohoku University; §Niigata University; ¶Tokyo Medical and Dental University; **Tokyo Dental College; ††Osaka University; ‡‡Shimane Medical University; §§Kyushu University; ¶¶Saga Medical University; ***Oita Medical University; †††Kagoshima University; ‡‡‡University of the Ryukyus, Japan SUMMARY. To investigate the incidence of cleft lip or palate or both (CLP) in Japan, 303 738 babies born in 1532 institutions between 1994 and 1995 were examined and 437 (0.14%) were found to have abnormalities. Of these babies, 32.1% had cleft lip, 43.3% had cleft lip and palate, and 24.8% had cleft palate (Table 2). These results show that the incidence of cleft lip and palate has declined compared with the period from 1981 to 1982. © 2000 The British Association of Oral and Maxillofacial Surgeons
INTRODUCTION
Extrapolating from these data, the total number of babies born with cleft abnormalities in Japan in 1994–1995 was estimated to be 1021 and the incidence 0.14%. In 1981, the number was 2770. Among 218 male infants, 81 (37%) had cleft lip, 99 (46%) had cleft lip and palate, and 38 (17%) had cleft palate, whereas of the 206 girls, 55 (27%) had cleft lip, 84 (41%) had cleft lip and palate and 67 (32%) had cleft palate. Data on the remaining 13 patients were incomplete. Data about gestation period were available for 395 births, 363 (92%) of whom were born between weeks 37 and 42 (normal term) and 32 (8%) between weeks 29 and 36 (preterm). The method of delivery was recorded for 398 births; 325 were spontaneous and 73 by vacuum extraction or cesarean section. Data on the pattern of clefts were available for only 310 babies (Table 1). Ninety-two babies had other deformities: 22 with cleft lips, 42 with cleft lip and palate, and 28 with cleft palate. The names of the other deformities are shown in Table 2.
Recently, the birth rate in Japan has fallen. Since 1982, the number of children with cleft lip and palate has decreased remarkably. However, we are not sure whether it resulted from a real reduction in incidence or a reduction in the number of children delivered. We therefore planned a nationwide survey. From 1993 to 1995, we sent out questionnaires to maternity institutions throughout Japan to ask about the incidence of CLP. We now report the results of the investigation and we compare our results with those of an investigation conducted by Miyazaki et al. during the period from 1981 to 1982.2 SUBJECTS AND METHODS The investigation was conducted between 1 January 1994 and 31 December 1995. We used a questionnaire survey, and aimed to find out the following details: total number of liveborn babies of each sex in each institution; the presence of babies with cleft lip or palate or both; type of cleft; sex of child; month of birth; gestation period; delivery process; and cleft patterns. We also recorded the presence of other disease (named) and the location of the institutions.
DISCUSSION The investigation of cleft lip or palate on such a nationwide scale is rare. This investigation was carried out to compare with the one conducted by Miyazaki et al. from
RESULTS
Table 1 – Pattern of cleft, figures and numbers (%)
The total number of babies born in the institutions that returned the questionnaire was 303 738, which was 42.8% of the total number of birth registered by the Ministry of Health and Welfare in the same area and period.9 Of these babies, 437 had some form of cleft lip or palate – an incidence of 0.14% (one in 695 babies).
Side Right Left Bilateral Central 605
Cleft lip
Cleft lip and palate
Cleft palate
Total
37 44 19 0
33 30 37 0
9 12 51 28
30 32 33 5
606
British Journal of Oral and Maxillofacial Surgery Table 2 – Associated deformities Cleft lip
Cleft lip and palate
Cleft palate
Hyperbilirubinaemia E-trisomy Oesophageal atresia Hydrocephalus Down syndrome Hypertrichosis Meningocele Microtia Non-insulin-dependent diabetes mellitus Otic imperforation Patent ductus arteriosus Polydactyly Syndactyly Tetralogy of Fallot Treacher–Collins syndrome 13-trisomy Ventricular septal defect
Persistent ductus arteriosus Polydactyly 18-trisomy Congenital anomalies of ear Congenital anomalies of multiple Hydrocephalus Inguinal hernia Micrognathia Neonatal jaundice Pes varus Sebaceous naevus Ventricular septal defect Congenital diaphragmatic hernia Dislocation of hip joint DiGorge syndrome Down syndrome Holoprosencephaly Hydrops fetalis Hyperbilirubinaemia Pierre–Robin syndrome Others
Pierre–Robin syndrome Chromosomal aberration Congenital anomalies of ear Congenital anomalies of lower jaw Congenital diaphragmatic hernia Tetralogy of Fallot Ventricular septal defect Blepharocoloboma Congenital anomalies of multiple Hydrocele of testis Hyperbilirubinaemia Microphthalmia Microtia Neonatal jaundice Polydactyly 13-trisomy 21-trisomy 22-trisomy
Age 30
No. of birth (ⴛ1000)
29
2000
28 27
1500
26 25
1000 24 23
500 22 21
0
20 1980
1993
1975
1980
1985
1990
1993
Fig. 2 – Total live births in Japan. Fig. 1 – Mean age of marriage (hatched) and first delivery (black).
1981 to 1982. We compared the women’s age of marriage and age at first delivery in 1993 with those in 1980 when the former investigation was made (Fig. 1). On the other hand, the total number of births has decreased (Fig. 2). The incidence of cleft lip or palate or both was 0.14% in 1994, while it was 0.18% in the 1981–1982 period. The estimated number of births with cleft lip or palate or both was about 2770 in 1981–1982 and about 1021 in 1994, so the incidence of clefts has decreased remarkably (P:0.01). We presume that women who know that their babies have malformations in the early stage of pregnancy may nowadays choose abortion. It is also speculated that a woman who delivers a baby with a cleft as a first child tends not to have another child.
Acknowledgements We are indebted to the obstetricians for their participation in this study, and to Ms Nariko Sumida for providing technical assistance in collecting and analysing the data. This study was supported by the grant-in-aid for scientific research of 1994 and 1995 from the Ministry of Education (Subject No. 06304042).
References 1. Bronshtein M, Blumenfeld I, Blumenfeld Z. Early prenatal diagnosis of cleft lip and its potential impact on the number of babies with cleft lip. Br J Oral Maxillofac Surg 1996; 34: 486–487. 2. Miyazaki T, Kohama G. The incidence of cleft lip and/or palate in Japanese: a record of 1981 and 1982. Jpn J Cleft Pal 1985; 10: 191–195.
Incidence of cleft lip or palate in 303 738 Japanese babies 3. Natsume N. Incidence of cleft lip and palate among Japanese newborns, 1982 to 1984. Plast Reconstr Surg 1987; 79: 499–501. 4. Natsume N, Suzuki T, Kawai T. The prevalence of cleft lip and palate in the Japanese: Their birth prevalence in 40,304 infants born during 1982. Oral Surg Oral Med Oral Pathol 1987; 63–64: 421–423. 5. Natsume N, Suzuki T, Kawai T. The prevalence of cleft lip and palate in Japanese. Br J Oral Maxillofac Surg 1988; 26: 232–236. 6. Natsume N, Suzuki T, Koie M et al. Incidence of cleft lip and/or palate in Aichi prefecture. Aichi-Gakuin J Dent Sci 1989; 2: 66–73. 7. Annual Report on the Labour Force Survey. Statistics Bureau Management and Coordination Agency, Japan, 1980. 8. Annual Report on the Labour Force Survey. Statistics Bureau Management and Coordination Agency, Japan, 1994. 9. Vital Statistics of Japan 1993: 1. Tokyo: Ministry of Health and Welfare.
The Authors N. Natsume T. Kawai Aichi-Gakuin University G. Kohama Sapporo Medical University T. Teshima S. Kochi Tohoku University Y. Ohashi Niigata University S. Enomoto M. Ishii T. Shigematsu (deceased) Tokyo Medical and Dental University
607
Y. Nakano Tokyo Dental College T. Matsuya M. Kogo Osaka University Y. Yoshimura Shimane Medical University M. Ohishi N. Nakamura Kyushu University T. Katsuki M. Goto Saga Medical University M. Shimizu S. Yanagisawa Oita Medical University T. Mimura Kagoshima University H. Sunakawa University of the Ryukyus Japan Correspondence and requests for offprints to: Nagato Natsume DDS, DMedSc, PhD, The Second Department of Oral and Maxillofacial Surgery, School of Dentistry, Aichi-Gakuin University, 2-11 Suemoridori, Chikusa-ku, Nagoya, 464-8651, Japan. Tel: ;81 52 751 7181 (ext. 293); Fax: ;81 52 752 5990 Paper received 27 September 1999 Accepted 15 August 2000
BRITISH JOURNAL OF ORAL
& M A X I L L O FAC I A L S U R G E RY
Incidence of cleft lip or palate in 303 738 Japanese babies born between 1994 and 1995 N. Natsume,* T. Kawai,* G. Kohama,† T. Teshima,‡ S. Kochi,‡ Y. Ohashi,§ S. Enomoto,¶ M. Ishii,¶ T. Shigematsu (deceased), Y. Nakano,** T. Matsuya,†† M. Kogo,†† Y. Yoshimura,‡‡ M. Ohishi,§ N. Nakamura,§ T. Katsuki,¶¶ M. Goto,¶¶ M. Shimizu,*** S. Yanagisawa,*** T. Mimura,††† H. Sunakawa‡‡‡ *Aichi-Gakuin University; †Sapporo Medical University; ‡Tohoku University; §Niigata University; ¶Tokyo Medical and Dental University; **Tokyo Dental College; ††Osaka University; ‡‡Shimane Medical University; §§Kyushu University; ¶¶Saga Medical University; ***Oita Medical University; †††Kagoshima University; ‡‡‡University of the Ryukyus, Japan SUMMARY. To investigate the incidence of cleft lip or palate or both (CLP) in Japan, 303 738 babies born in 1532 institutions between 1994 and 1995 were examined and 437 (0.14%) were found to have abnormalities. Of these babies, 32.1% had cleft lip, 43.3% had cleft lip and palate, and 24.8% had cleft palate (Table 2). These results show that the incidence of cleft lip and palate has declined compared with the period from 1981 to 1982. © 2000 The British Association of Oral and Maxillofacial Surgeons
INTRODUCTION
Extrapolating from these data, the total number of babies born with cleft abnormalities in Japan in 1994–1995 was estimated to be 1021 and the incidence 0.14%. In 1981, the number was 2770. Among 218 male infants, 81 (37%) had cleft lip, 99 (46%) had cleft lip and palate, and 38 (17%) had cleft palate, whereas of the 206 girls, 55 (27%) had cleft lip, 84 (41%) had cleft lip and palate and 67 (32%) had cleft palate. Data on the remaining 13 patients were incomplete. Data about gestation period were available for 395 births, 363 (92%) of whom were born between weeks 37 and 42 (normal term) and 32 (8%) between weeks 29 and 36 (preterm). The method of delivery was recorded for 398 births; 325 were spontaneous and 73 by vacuum extraction or cesarean section. Data on the pattern of clefts were available for only 310 babies (Table 1). Ninety-two babies had other deformities: 22 with cleft lips, 42 with cleft lip and palate, and 28 with cleft palate. The names of the other deformities are shown in Table 2.
Recently, the birth rate in Japan has fallen. Since 1982, the number of children with cleft lip and palate has decreased remarkably. However, we are not sure whether it resulted from a real reduction in incidence or a reduction in the number of children delivered. We therefore planned a nationwide survey. From 1993 to 1995, we sent out questionnaires to maternity institutions throughout Japan to ask about the incidence of CLP. We now report the results of the investigation and we compare our results with those of an investigation conducted by Miyazaki et al. during the period from 1981 to 1982.2 SUBJECTS AND METHODS The investigation was conducted between 1 January 1994 and 31 December 1995. We used a questionnaire survey, and aimed to find out the following details: total number of liveborn babies of each sex in each institution; the presence of babies with cleft lip or palate or both; type of cleft; sex of child; month of birth; gestation period; delivery process; and cleft patterns. We also recorded the presence of other disease (named) and the location of the institutions.
DISCUSSION The investigation of cleft lip or palate on such a nationwide scale is rare. This investigation was carried out to compare with the one conducted by Miyazaki et al. from
RESULTS
Table 1 – Pattern of cleft, figures and numbers (%)
The total number of babies born in the institutions that returned the questionnaire was 303 738, which was 42.8% of the total number of birth registered by the Ministry of Health and Welfare in the same area and period.9 Of these babies, 437 had some form of cleft lip or palate – an incidence of 0.14% (one in 695 babies).
Side Right Left Bilateral Central 605
Cleft lip
Cleft lip and palate
Cleft palate
Total
37 44 19 0
33 30 37 0
9 12 51 28
30 32 33 5
606
British Journal of Oral and Maxillofacial Surgery Table 2 – Associated deformities Cleft lip
Cleft lip and palate
Cleft palate
Hyperbilirubinaemia E-trisomy Oesophageal atresia Hydrocephalus Down syndrome Hypertrichosis Meningocele Microtia Non-insulin-dependent diabetes mellitus Otic imperforation Patent ductus arteriosus Polydactyly Syndactyly Tetralogy of Fallot Treacher–Collins syndrome 13-trisomy Ventricular septal defect
Persistent ductus arteriosus Polydactyly 18-trisomy Congenital anomalies of ear Congenital anomalies of multiple Hydrocephalus Inguinal hernia Micrognathia Neonatal jaundice Pes varus Sebaceous naevus Ventricular septal defect Congenital diaphragmatic hernia Dislocation of hip joint DiGorge syndrome Down syndrome Holoprosencephaly Hydrops fetalis Hyperbilirubinaemia Pierre–Robin syndrome Others
Pierre–Robin syndrome Chromosomal aberration Congenital anomalies of ear Congenital anomalies of lower jaw Congenital diaphragmatic hernia Tetralogy of Fallot Ventricular septal defect Blepharocoloboma Congenital anomalies of multiple Hydrocele of testis Hyperbilirubinaemia Microphthalmia Microtia Neonatal jaundice Polydactyly 13-trisomy 21-trisomy 22-trisomy
Age 30
No. of birth (ⴛ1000)
29
2000
28 27
1500
26 25
1000 24 23
500 22 21
0
20 1980
1993
1975
1980
1985
1990
1993
Fig. 2 – Total live births in Japan. Fig. 1 – Mean age of marriage (hatched) and first delivery (black).
1981 to 1982. We compared the women’s age of marriage and age at first delivery in 1993 with those in 1980 when the former investigation was made (Fig. 1). On the other hand, the total number of births has decreased (Fig. 2). The incidence of cleft lip or palate or both was 0.14% in 1994, while it was 0.18% in the 1981–1982 period. The estimated number of births with cleft lip or palate or both was about 2770 in 1981–1982 and about 1021 in 1994, so the incidence of clefts has decreased remarkably (P:0.01). We presume that women who know that their babies have malformations in the early stage of pregnancy may nowadays choose abortion. It is also speculated that a woman who delivers a baby with a cleft as a first child tends not to have another child.
Acknowledgements We are indebted to the obstetricians for their participation in this study, and to Ms Nariko Sumida for providing technical assistance in collecting and analysing the data. This study was supported by the grant-in-aid for scientific research of 1994 and 1995 from the Ministry of Education (Subject No. 06304042).
References 1. Bronshtein M, Blumenfeld I, Blumenfeld Z. Early prenatal diagnosis of cleft lip and its potential impact on the number of babies with cleft lip. Br J Oral Maxillofac Surg 1996; 34: 486–487. 2. Miyazaki T, Kohama G. The incidence of cleft lip and/or palate in Japanese: a record of 1981 and 1982. Jpn J Cleft Pal 1985; 10: 191–195.
Incidence of cleft lip or palate in 303 738 Japanese babies 3. Natsume N. Incidence of cleft lip and palate among Japanese newborns, 1982 to 1984. Plast Reconstr Surg 1987; 79: 499–501. 4. Natsume N, Suzuki T, Kawai T. The prevalence of cleft lip and palate in the Japanese: Their birth prevalence in 40,304 infants born during 1982. Oral Surg Oral Med Oral Pathol 1987; 63–64: 421–423. 5. Natsume N, Suzuki T, Kawai T. The prevalence of cleft lip and palate in Japanese. Br J Oral Maxillofac Surg 1988; 26: 232–236. 6. Natsume N, Suzuki T, Koie M et al. Incidence of cleft lip and/or palate in Aichi prefecture. Aichi-Gakuin J Dent Sci 1989; 2: 66–73. 7. Annual Report on the Labour Force Survey. Statistics Bureau Management and Coordination Agency, Japan, 1980. 8. Annual Report on the Labour Force Survey. Statistics Bureau Management and Coordination Agency, Japan, 1994. 9. Vital Statistics of Japan 1993: 1. Tokyo: Ministry of Health and Welfare.
The Authors N. Natsume T. Kawai Aichi-Gakuin University G. Kohama Sapporo Medical University T. Teshima S. Kochi Tohoku University Y. Ohashi Niigata University S. Enomoto M. Ishii T. Shigematsu (deceased) Tokyo Medical and Dental University
607
Y. Nakano Tokyo Dental College T. Matsuya M. Kogo Osaka University Y. Yoshimura Shimane Medical University M. Ohishi N. Nakamura Kyushu University T. Katsuki M. Goto Saga Medical University M. Shimizu S. Yanagisawa Oita Medical University T. Mimura Kagoshima University H. Sunakawa University of the Ryukyus Japan Correspondence and requests for offprints to: Nagato Natsume DDS, DMedSc, PhD, The Second Department of Oral and Maxillofacial Surgery, School of Dentistry, Aichi-Gakuin University, 2-11 Suemoridori, Chikusa-ku, Nagoya, 464-8651, Japan. Tel: ;81 52 751 7181 (ext. 293); Fax: ;81 52 752 5990 Paper received 27 September 1999 Accepted 15 August 2000