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重复拇指伴巨大软组织水肿——安抚奶嘴型拇指重复畸形

Duplicated thumb with enormous soft-tissue oedema - pacifier type of thumb duplication.

作者信息

Ochi Kensuke, Horiuchi Yukio, Takayama Shinichiro, Saito Harukazu

机构信息

Department of Orthopaedic Surgery, Kawasaki Municipal Kawasaki Hospital, Kanagawa, 210-0013, Japan.

出版信息

Hand Surg. 2011;16(1):91-3. doi: 10.1142/S0218810411005138.

DOI:10.1142/S0218810411005138
PMID:21348039
Abstract

Here we presented the first case of pacifier type thumb duplication. A newborn Japanese girl with no family history had a duplicated thumb on her left hand. The duplicated thumb showed a very large, oedematous soft-tissue nubbin in its appearance and was resected on the fifth day after birth. X-ray showed hypoplastic phalanx bone, suggesting type II polydactyly. Histology of the resected thumb showed enormous oedema in its connective tissue with cartilaginous and neural elements. This case was quite similar to literary reported cases of pacifier polydactyly in post-axial polydactyly, and its pathological condition seemed to be distinctly different from floating type or rudimentary type thumb duplication. We considered this type of thumb duplication as pacifier type thumb duplication, rather than floating or rudimentary type, in order to understand its underlying pathophysiology and to avoid confusion in further discussions.

摘要

在此,我们展示了首例安抚奶嘴型拇指重复畸形病例。一名无家族病史的日本新生儿女孩左手有一个重复拇指。该重复拇指外观呈现为一个非常大的、水肿的软组织小结节,并在出生后第5天进行了切除。X线显示指骨发育不全,提示为Ⅱ型多指畸形。切除拇指的组织学检查显示其结缔组织中有巨大水肿,并伴有软骨和神经成分。该病例与文献报道的轴后多指畸形中安抚奶嘴多指畸形的病例非常相似,其病理状况似乎与漂浮型或残迹型拇指重复畸形明显不同。为了理解其潜在的病理生理学并避免在进一步讨论中产生混淆,我们将这种类型的拇指重复畸形视为安抚奶嘴型拇指重复畸形,而非漂浮型或残迹型。

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引用本文的文献

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An unusual presentation of pacifier thumb duplication with VACTERL association: Case report and review of literature.伴有VACTERL综合征的安抚奶嘴拇指重复畸形的罕见表现:病例报告及文献综述
Int J Surg Case Rep. 2024 Sep;122:110090. doi: 10.1016/j.ijscr.2024.110090. Epub 2024 Jul 27.
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Excision of Rudimentary Preaxial Polydactyly of the Hand with Use of Electrocautery.手的初级桡侧多指畸形的切除用电烙术。
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Large preaxial polydactyly with intraepidermal bulla: an unusual form of polydactyly.
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BMJ Case Rep. 2019 Jan 3;12(1):e228183. doi: 10.1136/bcr-2018-228183.