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肢端持久性丘疹性黏蛋白沉积症:1例儿童罕见病例报告及文献复习

Acral persistent papular mucinosis: a rare child case report and literature review.

作者信息

Ding Mei-Yun, Zuo Hai-Liang, Sun Yan, Zhang Jian-Cheng, Yang Yong, Ma Fu-Jiang, Hu Xiao-Li, Zhan Jiang-Hua

机构信息

Department of Plastic Surgery, Tianjin Children's Hospital (Children's Hospital, Tianjin University), Tianjin, China.

Department of General Surgery, Tianjin Children's Hospital (Children's Hospital, Tianjin University), Tianjin, China.

出版信息

Transl Pediatr. 2024 Jun 30;13(6):987-993. doi: 10.21037/tp-23-607. Epub 2024 Jun 27.

DOI:10.21037/tp-23-607
PMID:38984022
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11228902/
Abstract

BACKGROUND

Acral persistent papular mucinosis (APPM) is a rare idiopathic subtype of localized lichen myxedematosus. To date, there have been less than 41 APPM cases reported worldwide, however, almost all patients were older than 18 years of age. A 7-year-old child was first reported in this paper.

CASE DESCRIPTION

A 7-year-old boy was admitted to our hospital with a solitary skin-colored papule on the radial side of the middle segment of his right index finger. The patient wanted to know the exact diagnosis and remove it because the flexion movement of the middle segment had been affected. Thus, a surgery was performed. Histopathological examination of a biopsy specimen obtained from the papule on the radial side of the middle segment of his right index finger showed a focal and well-circumscribed deposit of mucin in the papillary and middermis. The deposit never extended deeply into the reticular dermis. Mucin spared a subepidermal area in the papillary dermis. Alcian blue stains can highlight the mucin. The papule was histologically diagnosed as an APPM and excised surgically. The wound gradually healed after the operation, and no obvious recurrence, scar or other discomfort was observed during follow-up so far.

CONCLUSIONS

To the best of our knowledge, this is the rare case of a child APPM presenting as a solitary papule affecting the flexion movement of the middle segment. Since it is a rare disease, we report this case to contribute to future research on the diagnosis and pathogenesis of APPM.

摘要

背景

肢端持久性丘疹性粘蛋白病(APPM)是局限性粘液性苔藓的一种罕见特发性亚型。迄今为止,全球报道的APPM病例少于41例,然而,几乎所有患者年龄均超过18岁。本文首次报道了一名7岁儿童的病例。

病例描述

一名7岁男孩因右示指中段桡侧出现单个肤色丘疹入院。由于该中段的屈曲运动受到影响,患者想知道确切诊断并将其去除。因此,进行了手术。对从右示指中段桡侧丘疹获取的活检标本进行组织病理学检查,结果显示在乳头层和真皮中层有局灶性且边界清楚的粘蛋白沉积。该沉积从未深入到网状真皮层。粘蛋白在乳头层真皮中保留了一个表皮下区域。阿尔辛蓝染色可使粘蛋白显色。该丘疹经组织学诊断为APPM并进行了手术切除。术后伤口逐渐愈合,随访至今未观察到明显复发、瘢痕或其他不适。

结论

据我们所知,这是罕见的儿童APPM病例,表现为单个丘疹并影响中段的屈曲运动。由于这是一种罕见疾病,我们报告此病例以促进未来对APPM诊断和发病机制的研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/f46155586baa/tp-13-06-987-f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/64912f074d75/tp-13-06-987-f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/ab53b555e80c/tp-13-06-987-f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/f46155586baa/tp-13-06-987-f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/64912f074d75/tp-13-06-987-f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/ab53b555e80c/tp-13-06-987-f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2f7a/11228902/f46155586baa/tp-13-06-987-f3.jpg

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本文引用的文献

1
Solitary acral persistent papular mucinosis nodule: A case report and summary of eight Korean cases.孤立性肢端持续性丘疹性黏蛋白沉积症结节:一例报告及八例韩国病例总结
World J Clin Cases. 2023 May 6;11(13):3086-3091. doi: 10.12998/wjcc.v11.i13.3086.
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Clin Dermatol. 2021 Mar-Apr;39(2):211-214. doi: 10.1016/j.clindermatol.2020.10.001. Epub 2020 Oct 16.
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Acral persistent papular mucinosis: A case report and summary of 24 Japanese cases.肢端持久性丘疹性粘蛋白沉积症:一例报告及 24 例日本病例总结。
J Dermatol. 2021 Oct;48(10):1574-1578. doi: 10.1111/1346-8138.16043. Epub 2021 Jun 25.
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Acral persistent papular mucinosis with pruritic skin lesions.伴有瘙痒性皮肤损害的肢端持久性丘疹性黏蛋白沉积症。
An Bras Dermatol. 2018 Sep-Oct;93(5):769-770. doi: 10.1590/abd1806-4841.20187878.
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Acral persistent papular mucinosis (APPM): Dermoscopy of an uncommon disease.
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