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一例具有支气管类癌的异位促肾上腺皮质激素综合征的疑难病例及文献综述

A Challenging Case of Ectopic Adrenocorticotropin Hormone Syndrome with Bronchial Carcinoid and Literature Review.

作者信息

Abseyi Sema Nilay, Şıklar Zeynep, Özsu Elif, Kayı Cangır Ayten, Cabi Ünal Emel, Taçyıldız Nurdan, Aycan Zehra, Berberoğlu Merih

机构信息

Ankara University Faculty of Medicine, Department of Pediatric Endocrinology, Ankara, Türkiye

Ankara University Faculty of Medicine, Department of Thoracic Surgery, Ankara, Türkiye

出版信息

J Clin Res Pediatr Endocrinol. 2025 Aug 22;17(3):352-358. doi: 10.4274/jcrpe.galenos.2023.2023-7-6. Epub 2023 Nov 9.

DOI:10.4274/jcrpe.galenos.2023.2023-7-6
PMID:37942887
Abstract

We report an adolescent boy diagnosed with ectopic adrenocorticotropin hormone syndrome (EAS) due to an atypical bronchial carcinoid. The patient was managed by a multidisciplinary team. He underwent surgery and subsequent chemotherapy and radiotherapy treatments. The patient is still under our follow-up. At the time of writing, eighteen pediatric and adolescent patients with EAS because of bronchial carcinoid tumors have been reported. EAS due to bronchial carcinoids is very rare in children and adolescents. Careful diagnostic evaluation and rapid treatment should be started immediately. Although complete remission is possible, atypical carcinoids have a more aggressive nature. A multidisciplinary approach and follow-up is recommended in terms of quality of life and survival.

摘要

我们报告了一名因非典型支气管类癌而被诊断为异位促肾上腺皮质激素综合征(EAS)的青春期男孩。该患者由多学科团队进行管理。他接受了手术以及后续的化疗和放疗。患者仍在我们的随访中。在撰写本文时,已有18例因支气管类癌肿瘤导致EAS的儿科和青少年患者被报道。支气管类癌导致的EAS在儿童和青少年中非常罕见。应立即开始仔细的诊断评估和快速治疗。尽管完全缓解是可能的,但非典型类癌具有更强的侵袭性。就生活质量和生存率而言,建议采用多学科方法并进行随访。

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本文引用的文献

1
Clinical profile and treatment outcomes of patients with ectopic ACTH syndrome compared to Cushing disease: a single-center experience.与库欣病相比,异位促肾上腺皮质激素(ACTH)综合征患者的临床特征和治疗结果:单中心经验
Endocrine. 2023 May;80(2):408-418. doi: 10.1007/s12020-022-03298-z. Epub 2023 Jan 7.
2
[Clinical and laboratory characteristics and results of treatment of patients with ACTH-producing neuroendocrine tumors of various localization].[不同部位产生促肾上腺皮质激素的神经内分泌肿瘤患者的临床和实验室特征及治疗结果]
Ter Arkh. 2021 Oct 15;93(10):1171-1178. doi: 10.26442/00403660.2021.10.201102.
3
Characterization of Outcomes by Surgical Management of Lung Neuroendocrine Tumors Associated With Cushing Syndrome.
手术治疗伴有库欣综合征的肺神经内分泌肿瘤的结局特征。
JAMA Netw Open. 2021 Sep 1;4(9):e2124739. doi: 10.1001/jamanetworkopen.2021.24739.
4
Clinical Heterogeneity of Ectopic ACTH Syndrome: A Long-Term Follow-Up Study.异位促肾上腺皮质激素综合征的临床异质性:一项长期随访研究。
Endocr Pract. 2020 Dec;26(12):1435-1441. doi: 10.4158/EP-2020-0368.
5
Bronchial Carcinoid Tumour as a Rare Cause of Cushing’s Syndrome in Children: A Case Report and Review of Literature.支气管类癌肿瘤致儿童库欣综合征罕见病因:病例报告及文献复习。
J Clin Res Pediatr Endocrinol. 2020 Nov 25;12(4):340-346. doi: 10.4274/jcrpe.galenos.2019.2019.0156. Epub 2020 Jan 3.
6
The Clinical Impact of [ Ga]-DOTATATE PET/CT for the Diagnosis and Management of Ectopic Adrenocorticotropic Hormone - Secreting Tumours.[Ga]-DOTATATE PET/CT 对异位促肾上腺皮质激素分泌肿瘤的诊断和治疗的临床影响。
Clin Endocrinol (Oxf). 2019 Aug;91(2):288-294. doi: 10.1111/cen.14008. Epub 2019 Jun 12.
7
The utility of 18F-FDG and 68Ga-DOTA-Peptide PET/CT in the evaluation of primary pulmonary carcinoid: A systematic review and meta-analysis.18F-FDG和68Ga-DOTA-肽PET/CT在原发性肺类癌评估中的应用:一项系统评价和荟萃分析
Medicine (Baltimore). 2019 Mar;98(10):e14769. doi: 10.1097/MD.0000000000014769.
8
Cushing's Syndrome in Pediatrics: An Update.儿童库欣综合征:最新进展。
Endocrinol Metab Clin North Am. 2018 Jun;47(2):451-462. doi: 10.1016/j.ecl.2018.02.008.
9
An update on Cushing syndrome in pediatrics.儿童库欣综合征的最新进展。
Ann Endocrinol (Paris). 2018 Jun;79(3):125-131. doi: 10.1016/j.ando.2018.03.010. Epub 2018 Apr 9.
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