Suppr超能文献

声门下异位胸腺酷似气道血管瘤。

Subglottic ectopic thymus mimicking airway haemangioma.

机构信息

Paediatric Otolaryngology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.

Stem Cell and Regenerative medicine, UCL Great Ormond Street Hospital Institue of Child Health, London, UK.

出版信息

BMJ Case Rep. 2021 May 19;14(5):e241074. doi: 10.1136/bcr-2020-241074.

Abstract

Subglottic haemangioma presents as progressive obstruction in the neonatal and infantile airway, with a soft lesion seen during endoscopy. Diagnosis is based on macroscopic findings, biopsy is not usually performed and propranolol is first-line treatment. In contrast, ectopic thymus is a rare differential diagnosis for subglottic mass made by histopathological examination after excision or autopsy. In this article, we present a case of an infant with a subglottic lesion with endoscopic features consistent with haemangioma. After initial clinical response to propranolol, the patient represented with progressive stridor no longer responding to therapy. Open excision of the lesion was performed, and histopathology revealed ectopic thymus tissue. In this case, ectopic thymus tissue mimicked the presentation of subglottic haemangioma, and confirmation bias persisted due to an apparent initial clinical response to treatment with propranolol. In cases of subglottic mass refractory to medical treatment, excision of the lesion should be considered.

摘要

声门下血管瘤表现为新生儿和婴儿气道进行性阻塞,内镜下可见到柔软的病变。诊断基于宏观发现,通常不进行活检,而是将普萘洛尔作为一线治疗。相比之下,异位胸腺是切除或尸检后组织病理学检查诊断声门下肿块的罕见鉴别诊断。本文报道了 1 例婴儿声门下病变,内镜特征符合血管瘤。最初对普萘洛尔有临床反应,但随后出现对治疗无反应的进行性喘鸣。对病变进行了开放性切除,组织病理学显示为异位胸腺组织。在这种情况下,异位胸腺组织模拟了声门下血管瘤的表现,由于对普萘洛尔治疗有明显的初始临床反应,因此存在确认偏倚。对于药物治疗无效的声门下肿块,应考虑切除病变。

相似文献

1
Subglottic ectopic thymus mimicking airway haemangioma.
BMJ Case Rep. 2021 May 19;14(5):e241074. doi: 10.1136/bcr-2020-241074.
2
Treatment failure with propranolol for subglottic haemangioma.
BMJ Case Rep. 2019 May 13;12(5):e227135. doi: 10.1136/bcr-2018-227135.
3
Great Ormond Street Hospital treatment guidelines for use of propranolol in infantile isolated subglottic haemangioma.
J Laryngol Otol. 2013 Mar;127(3):295-8. doi: 10.1017/S0022215112003192. Epub 2013 Feb 1.
4
Propranolol as first-line treatment of a severe subglottic haemangioma.
Eur J Cardiothorac Surg. 2013 Jan;43(1):187-9. doi: 10.1093/ejcts/ezs218. Epub 2012 Oct 8.
6
Ultrasound and colour Doppler in infantile subglottic haemangioma.
Pediatr Radiol. 2011 Nov;41(11):1421-8. doi: 10.1007/s00247-011-2213-1. Epub 2011 Sep 9.
7
[Subglottic haemangioma].
Ugeskr Laeger. 2014 Aug 4;176(32).
8
Propranolol for the treatment of subglottic hemangiomas.
Int J Pediatr Otorhinolaryngol. 2011 Dec;75(12):1510-4. doi: 10.1016/j.ijporl.2011.08.017. Epub 2011 Sep 23.
9
Successful treatment of isolated subglottic haemangioma with propranolol alone.
Int J Pediatr Otorhinolaryngol. 2009 Dec;73(12):1821-3. doi: 10.1016/j.ijporl.2009.08.020. Epub 2009 Sep 30.
10
Propranolol for the treatment of a life-threatening subglottic and mediastinal infantile hemangioma.
J Pediatr. 2010 Feb;156(2):335-8. doi: 10.1016/j.jpeds.2009.10.010.

本文引用的文献

1
Addressing a rare cause of paediatric stridor: subglottic cyst.
BMJ Case Rep. 2020 Jun 28;13(6):e236600. doi: 10.1136/bcr-2020-236600.
2
Ectopic Thymus: An Unusual Case of Subglottic Mass.
Ann Otol Rhinol Laryngol. 2019 Dec;128(12):1182-1188. doi: 10.1177/0003489419863828. Epub 2019 Jul 18.
3
Infantile hemangiomas: what have we learned from propranolol?
Curr Opin Pediatr. 2018 Aug;30(4):499-504. doi: 10.1097/MOP.0000000000000650.
4
Topical propranolol for infantile haemangiomas: a systematic review.
J Eur Acad Dermatol Venereol. 2018 Dec;32(12):2083-2089. doi: 10.1111/jdv.14963. Epub 2018 May 3.
5
Management of Infantile Hemangiomas of the Airway.
Otolaryngol Clin North Am. 2018 Feb;51(1):133-146. doi: 10.1016/j.otc.2017.09.001.
6
Treatment of infantile haemangiomas: recommendations of a European expert group.
Eur J Pediatr. 2015 Jul;174(7):855-65. doi: 10.1007/s00431-015-2570-0. Epub 2015 May 29.
7
Ectopic cervical thymus associated with infant death: 2 case reports and literature review.
Int J Pediatr Otorhinolaryngol. 2013 Sep;77(9):1609-12. doi: 10.1016/j.ijporl.2013.06.030. Epub 2013 Jul 23.
8
Propranolol and infantile hemangiomas four years later: a systematic review.
Pediatr Dermatol. 2013 Mar-Apr;30(2):182-91. doi: 10.1111/pde.12089. Epub 2013 Feb 14.
9
Propranolol for infantile haemangiomas: insights into the molecular mechanisms of action.
Br J Dermatol. 2010 Aug;163(2):269-74. doi: 10.1111/j.1365-2133.2010.09848.x. Epub 2010 May 8.
10
Propranolol for severe hemangiomas of infancy.
N Engl J Med. 2008 Jun 12;358(24):2649-51. doi: 10.1056/NEJMc0708819.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验