48例外胚层发育不良综合征患者颌面骨形态分析

doi:10.19439/j.sjos.2017.02.014

上海口腔医学 ›› 2017, Vol. 26 ›› Issue (2): 193-197.doi: 10.19439/j.sjos.2017.02.014

48例外胚层发育不良综合征患者颌面骨形态分析

王豪伟¹, 王凤¹, 黄伟¹, 周文洁², 王跃平², 吴轶群¹

1.上海交通大学医学院附属第九人民医院·口腔医学院口腔种植科,上海 200011;
2.上海交通大学医学院附属第九人民医院·口腔医学院口腔第二门诊部,上海 201900

收稿日期:2017-02-20 修回日期:2017-03-28 出版日期:2017-04-25 发布日期:2017-05-04
通讯作者: 吴轶群,E-mail：yiqunwu@hotmail.com
作者简介:王豪伟（1991-）,男,硕士研究生,E-mail: wanghwsmile@126.com
基金资助:
国家自然科学基金（81371190）; 上海交通大学医工交叉重点项目（YG2016ZD01）; 上海交通大学医学院附属第九人民医院融合课题

Morphometric analysis of maxillofacial bone in 48 patients with ectodermal dysplasia

WANG Hao-wei¹, WANG Feng¹, HUANG Wei¹, ZHOU Wen-jie², WANG Yue-ping², WU Yi-qun¹

1.Department of Oral Implantology, Shanghai Ninth People's Hospital, College of Stomatology, Shanghai Jiao Tong University School of Medicine. Shanghai 200011;
2.The Second Dental Center, Shanghai Ninth People's Hospital, College of Stomatology, Shanghai Jiao Tong University School of Medicine. Shanghai 201900, China

Received:2017-02-20 Revised:2017-03-28 Online:2017-04-25 Published:2017-05-04

摘要/Abstract

摘要： 目的: 了解外胚层发育不良综合征（ectodermal dysplasia,ED）患者的颌面部发育状况,为临床治疗提供参考。方法: 收集2013年6月—2016年10月收治的48例ED患者的临床信息和头颅侧位X线片。将患者分为2组：第1组23例,除缺牙外,累及≥2种外胚层组织;第2组25例,除缺牙外,仅累及1种外胚层组织。比较2组病例的缺牙数及颌面骨形态影像学参数的差异。采用SPSS 17.0软件包对数据进行单样本t检验和成组t检验。结果: 第1组缺牙数显著多于第2组（22.3±5.8:12±6.6,P<0.05）。在女性患者中,第1组滞留乳牙数显著多于第2组(8±4:2.4±2.7,P<0.05)。2组患者的上颌骨长度减少（ANS-Ptm减小）且位置后缩（S-Ptm、SNA、NA-PA减小）,下颌骨前突（NP-FH增大）、颏部前突（Y轴角减小）,下颌骨长度（Co-Po）及位置（S-Co）相对正常,骨性Ⅲ类错（ANB<0）,全面高降低（N-Me减小）。在男性患者中, 第1组较第2组的SNA、NA-PA、ANS-Ptm、S-Ptm、Y轴角显著减小（P<0.05）,NP-FH显著增大（P<0.05）。结论: ED患者颌面骨形态表现为上颌骨长度减少及位置后缩,下颌骨长度和位置相对正常,下颌骨前突、颏部前突、骨性Ⅲ类错和全面高降低。ED患者颌面骨形态异常的严重程度与缺失的恒牙数呈正相关,滞留的乳牙可能会促进颌面骨发育。

关键词: 外胚层发育不良, 缺牙, 上颌骨, 下颌骨

Abstract: PURPOSES: The study was performed to analyze the maxillofacial morphology of 48 patients with ectodermal dysplasia (ED) and to provide some reference for implant treatment of ED patients. METHODS: The study collected the clinical data and lateral cephalometric radiographs of 48 patients with ED treated between June 2013 and October 2016. The patients were grouped as follows: group 1, 23 patients, at least with two kinds of ectodermal disorder, besides hypodontia; group 2, 25 patients, only with one kind of ectodermal disorder, besides hypodontia. The number of missing teeth and maxillofacial cephalometric parameters were compared between 2 groups. SPSS 17.0 software package was performed for statistical analysis. RESULTS: The number of missing teeth was significantly larger in group 1(22.3±5.8) than group 2 (12± 6.6) (P<0.05). The number of retained deciduous teeth was significantly larger in group 1 (8±4) than in group 2 (2.4±2.7) in females (P<0.05). Cephalometric analysis showed a reduced maxilla length (ANS-Ptm), retrusive maxilla (S-Ptm, SNA, NA-PA), a normal size (S-Co) and length (Co-Po) of mandible, a protruding mandible (NP-FH) and chin (Y axis), skeletal Angle Ⅲ malocclusion(ANB<0) and a reduced facial height (N-Me) in two groups. SNA, NA-PA, ANS-Ptm, S-Ptm and Y axis were significantly decreased and NP-FH was significantly increased in group 1 compared to group 2 in males (P<0.05). CONCLUSIONS: Maxillofacial morphology of ED patients typically presented a reduced maxilla length and retrusive maxilla, a normal size and length of mandible, a protruding mandible and chin, skeletal Angle Ⅲ malocclusion and a reduced facial height. The severity of abnormalities in maxillofacial bone morphology was correlated positively with absence of permanent teeth. Furthermore, retained deciduous teeth might have positive effect on the development of maxillofacial bone.

Key words: Ectodermal dysplasia, Hypodontia, Maxilla, Mandible

中图分类号:

R782.2

王豪伟, 王凤, 黄伟, 周文洁, 王跃平, 吴轶群. 48例外胚层发育不良综合征患者颌面骨形态分析[J]. 上海口腔医学, 2017, 26(2): 193-197.

WANG Hao-wei, WANG Feng, HUANG Wei, ZHOU Wen-jie, WANG Yue-ping, WU Yi-qun. Morphometric analysis of maxillofacial bone in 48 patients with ectodermal dysplasia[J]. Shanghai Journal of Stomatology, 2017, 26(2): 193-197.

参考文献

[1] Freire-Maia N. Ectodermal dysplasias [J]. Hum Hered, 1971, 21(4): 309-312.
[2] Doan MS, Callea M, Yavuz I, et al. An evaluation of clinical, radiological and three-dimensional dental tomography findings in ectodermal dysplasia cases [J]. Med Oral Patol Oral Cir Bucal, 2015, 20(3): e340-e346.
[3] Henningsen E, Svendsen MT, Lildballe DL, et al. A novel mutation in the EDAR gene causes severe autosomal recessive hypohidrotic ectodermal dysplasia [J]. Am J Med Genet A, 2014, 164A(8): 2059-2061.
[4] García-Martín P, Hernández-Martín A, Torrelo A. Ectodermal dysplasias: a clinical and molecular review[J]. Actas Dermosifiliogr, 2013, 104(6): 451-470.
[5] Visinoni ÁF, Lisboa-Costa T, Pagnan NA, et al. Ectodermal dysplasias: clinical and molecular review [J]. Am J Med Genet A, 2009, 149A(9): 1980-2002.
[6] Sepulveda W, Sandoval R, Carstens E, et al. Hypohidrotic ectodermal dysplasia: prenatal diagnosis by three-dimensional ultrasonography [J]. J Ultrasound Med, 2003, 22(7): 731-735.
[7] Lexner MO, Bardow A, Bjorn-Jorgensen J, et al. Anthropometric and cephalometric measurements in X-linked hypohidrotic ectodermal dysplasia [J]. Orthod Craniofac Res, 2007, 10(4): 203-215.
[8] Bixler D, Saksena SS, Ward RE. Characterization of the face in hypohidrotic ectodermal dysplasia by cephalometric and anthropometric analysis [J]. Birth Defects Orig Artic Ser, 1988, 24(2): 197-203.
[9] Vierucci S, Baccetti T, Tollaro I. Dental and craniofacial findings in hypohidrotic ectodermal dysplasia during the primary dentition phase [J]. J Clin Pediatr Dent, 1994, 18(4): 291-297.
[10] Ruhin B, Martinot V, Lafforgue P, et al. Pure ectodermal dysplasia: retrospective study of 16 cases and literature review[J]. Cleft Palate Craniofac J, 2001, 38(5): 504-518.
[11] 傅民魁, 丁寅, 王林, 等. 口腔正畸学[M]. 第6版. 北京: 人民卫生出版社, 2012: 85-90.
[12] Arzoo PS, Klar J, Bergendal B, et al. WNT10A mutations account for 1/4 of population-based isolated oligodontia and show phenotypic correlations [J]. Am J Med Genet A, 2014, 164A(2): 353-359.
[13] Bergendal B, Norderyd J, Bagesund M, et al. Signs and symptoms from ectodermal organs in young Swedish individuals with oligodontia [J]. Int J Paediatr Dent, 2006, 16(5): 320-326.
[14] Singh GP, Saxena V. Hypohidrotic ectodermal dysplasia [J]. Med J Armed Forces India, 2015, 71(suppl 2): S530-553.
[15] Nieminen P. Genetic basis of tooth agenesis[J]. J Exp Zool B Mol Dev Evol, 2009, 312B(4): 320-342.
[16] Bondarets N, McDonald F. Analysis of the vertical facial form in patients with severe hypodontia [J]. Am J of Phys Anthropol, 2000, 111(2): 177-184.
[17] Sarnäs KV, Rune B. The facial profile in advanced hypodontia: a mixed longitudinal study of 141 children [J]. Eur J Orthod, 1983, 5(2): 133-143.
[18] Noal M, Kjaer I, Solow B. Craniofacial morphology in patients with multiple congenitally missing permanent teeth [J]. Eur J Orthod, 1994, 16(2): 104-109.
[19] Nomura S, Hasegawa S, Noda T, et al. Longitudinal study of jaw growth and prosthetic management in a patient with ectodermal dysplasia and anodontia [J]. Int J Paediatr Dent, 1993, 3(1): 29-38.
[20] Sarnat BG, Brodie AG, Kubacki WH. Fourteen-year report of facial growth in case of complete anodontia with ectodermal dysplasia [J]. AMA Am J Dis Child, 1953, 86(2): 162-169.

48例外胚层发育不良综合征患者颌面骨形态分析

Morphometric analysis of maxillofacial bone in 48 patients with ectodermal dysplasia

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