A rare case of complete unilateral cleft lip complicated with maxillary duplication：Apropos of a case report and systematic literature review

doi:10.3877/cma.j.issn.1674-1366.2025.02.006

Abstract

Abstract:

Craniofacial duplication is a kind of extremely rare birth defect.This congenital deformity demonstrates significantly various phenotypes and must be treated with personalized protocol based on the extent of soft and osseous tissue involvement and affected physiologic functions.Here we report a case of complete unilateral cleft lip complicated with maxillary duplication.Its clinical manifestation and surgical procedures are described.The literature covering its etiology and management is also systematically reviewed.

Key words: Jaw, Duplication anomaly, Embryogenesis, Birth defect, Prenatal diagnosis

Chenxi Li, Zhongcheng Gong, Xinhua Zhang, Lu Zhang. A rare case of complete unilateral cleft lip complicated with maxillary duplication：Apropos of a case report and systematic literature review[J]. Chinese Journal of Stomatological Research(Electronic Edition), 2025, 19(02): 119-123.

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References 18

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参考文献	具体分型特征
Barr^［8］（1982）	Ⅰ型：眼鼻复制畸形，伴或不伴上颌或下颌复制畸形
	Ⅱ型：鼻复制畸形，伴或不伴上颌复制畸形
	Ⅲ型：下颌复制畸形，伴或不伴下颌或垂体复制畸形
Gorlin等^［11］（1990）	Ⅰ型：单一口腔伴上颌牙弓复制
	Ⅱ型：下颌旁出现附口
	Ⅲ型：单一口腔伴下颌部分复制
Sun等^［10］（2013）^a	Ⅰ型：含有乳牙或牙胚的对称复制的下颌牙弓，可能伴舌复制畸形及唇腭裂（可能表现为部分复制的口腔）
	Ⅱ型：单侧下颌体和升支复制畸形，复制的下颌骨可以从下颌联合延伸至颞下颌关节作为独立的下颌结构
	Ⅲ型：为牙槽型，其特征是多生牙区的复制的牙槽骨与正常下颌骨相连
	Ⅳ型：双侧下颌升支复制畸形，并通常与Klippel-Feil综合征相关
Hamberis等^［1］（2020）^b	Ⅰ型：正常功能的口腔和位置不定的附口，可能包含上唇和下唇大部分口腔结构
	Ⅱ型：复制的口腔和正常口腔结构相连，带有上颌骨的上唇，或带有下颌骨的下唇
	Ⅲ型：正常口腔位于中心，复制的口腔和正常口腔结构相连，由口腔的软组织和骨组织结构构成

参考文献	具体分型特征
Barr^［8］（1982）	Ⅰ型：眼鼻复制畸形，伴或不伴上颌或下颌复制畸形
	Ⅱ型：鼻复制畸形，伴或不伴上颌复制畸形
	Ⅲ型：下颌复制畸形，伴或不伴下颌或垂体复制畸形
Gorlin等^［11］（1990）	Ⅰ型：单一口腔伴上颌牙弓复制
	Ⅱ型：下颌旁出现附口
	Ⅲ型：单一口腔伴下颌部分复制
Sun等^［10］（2013）^a	Ⅰ型：含有乳牙或牙胚的对称复制的下颌牙弓，可能伴舌复制畸形及唇腭裂（可能表现为部分复制的口腔）
	Ⅱ型：单侧下颌体和升支复制畸形，复制的下颌骨可以从下颌联合延伸至颞下颌关节作为独立的下颌结构
	Ⅲ型：为牙槽型，其特征是多生牙区的复制的牙槽骨与正常下颌骨相连
	Ⅳ型：双侧下颌升支复制畸形，并通常与Klippel-Feil综合征相关
Hamberis等^［1］（2020）^b	Ⅰ型：正常功能的口腔和位置不定的附口，可能包含上唇和下唇大部分口腔结构
	Ⅱ型：复制的口腔和正常口腔结构相连，带有上颌骨的上唇，或带有下颌骨的下唇
	Ⅲ型：正常口腔位于中心，复制的口腔和正常口腔结构相连，由口腔的软组织和骨组织结构构成

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