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食管节细胞神经瘤病;一种难治性食管狭窄的罕见病因:病例报告。

Esophageal ganglioneuromatosis; a rare cause of intractable esophageal stenosis: a case report.

机构信息

Department of Pediatric Surgery, Faculty of Medicine, Alexandria University, Alexandria, Egypt.

Nile of Hope Hospital for congenital anomalies, Alexandria, Egypt.

出版信息

BMC Pediatr. 2024 Jul 16;24(1):456. doi: 10.1186/s12887-024-04923-8.

DOI:10.1186/s12887-024-04923-8
PMID:39014304
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11251384/
Abstract

BACKGROUND

Ganglioneuromatosis is a rare type of benign neurogenic tumor that usually affects the sites of the major sympathetic ganglia in the retroperitoneum and the posterior mediastinum. Affection of the gastrointestinal tract is rare, and involvement of the esophagus is exceptional. To the best of our knowledge, only 4 cases of esophageal ganglioneuromatosis in adults were reported in the literature. No cases have been reported in the pediatric age group.

CASE PRESENTATION

An 11-year-old boy presented with dysphagia due to severe esophageal stenosis caused by esophageal ganglioneuromatosis.

CONCLUSIONS

Despite its rarity, the present case implies that ganglioneuromatosis should be considered in children with idiopathic esophageal stenosis.

摘要

背景

神经节细胞瘤是一种罕见的良性神经源性肿瘤,通常影响腹膜后和后纵隔的主要交感神经节部位。胃肠道受累很少见,食管受累则更为罕见。据我们所知,文献中仅报道了 4 例成人食管神经节细胞瘤。在儿科年龄组中没有病例报告。

病例介绍

一名 11 岁男孩因食管神经节细胞瘤导致严重的食管狭窄而出现吞咽困难。

结论

尽管罕见,但本例提示对于特发性食管狭窄的儿童,应考虑神经节细胞瘤的可能。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/b387621dcd66/12887_2024_4923_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/dd2e8ebadcab/12887_2024_4923_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/fe16a4eb3ad4/12887_2024_4923_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/6fba62db91de/12887_2024_4923_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/b387621dcd66/12887_2024_4923_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/dd2e8ebadcab/12887_2024_4923_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/fe16a4eb3ad4/12887_2024_4923_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/6fba62db91de/12887_2024_4923_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d390/11251384/b387621dcd66/12887_2024_4923_Fig4_HTML.jpg

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本文引用的文献

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A case of ganglioneuromatosis in a child and its outcome.一名儿童神经节神经瘤病病例及其转归。
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Isolated intestinal Ganglioneuromatosis: case report and literature review.孤立性肠神经节细胞瘤:病例报告及文献复习。
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Diffuse Intestinal Ganglioneuromatosis Causing Severe Intestinal Dysmotility in a Child With a PTEN Mutation.弥漫性肠道神经节瘤病导致一名携带PTEN突变儿童的严重肠道运动障碍。
J Pediatr Gastroenterol Nutr. 2019 Feb;68(2):e35-e37. doi: 10.1097/MPG.0000000000002072.
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International Survey on the Management of Anastomotic Leakage After Esophageal Resection.国际食管切除术后吻合口漏管理调查。
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Evaluation of Anastomotic Leak after Esophagectomy for Esophageal Cancer: Typical Time Point of Occurrence, Mode of Diagnosis, Value of Routine Radiocontrast Agent Studies and Therapeutic Options.食管癌食管切除术后吻合口漏的评估:典型发生时间点、诊断方式、常规造影剂检查的价值及治疗选择
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Pseudo-obstruction of the Gastric Outlet Caused by Combined Hyperganglionosis and Ganglioneuromatosis in an adult: Case Report and Literature Review.成人合并高神经节症和神经节瘤病导致胃出口假性梗阻:病例报告及文献复习
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Recent improvements in the management of esophageal anastomotic leak after surgery for cancer.癌症手术后食管吻合口漏管理的近期进展。
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Intestinal ganglioneuromatosis: an unusual aetiology for occult gastrointestinal bleeding.肠道神经节瘤病:隐匿性胃肠道出血的一种罕见病因。
BMJ Case Rep. 2015 Sep 30;2015:bcr2015211764. doi: 10.1136/bcr-2015-211764.
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