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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.

DOI:10.1136/bcr-2020-241074
PMID:34011669
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8137208/
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。

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1
Subglottic ectopic thymus mimicking airway haemangioma.声门下异位胸腺酷似气道血管瘤。
BMJ Case Rep. 2021 May 19;14(5):e241074. doi: 10.1136/bcr-2020-241074.
2
Treatment failure with propranolol for subglottic haemangioma.普萘洛尔治疗声门下血管瘤失败。
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Propranolol as first-line treatment of a severe subglottic haemangioma.普萘洛尔作为严重声门下型血管瘤的一线治疗药物。
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本文引用的文献

1
Addressing a rare cause of paediatric stridor: subglottic cyst.探讨小儿喘鸣的罕见病因:声门下囊肿。
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?婴幼儿血管瘤:普萘洛尔给我们带来了哪些启示?
Curr Opin Pediatr. 2018 Aug;30(4):499-504. doi: 10.1097/MOP.0000000000000650.
4
Topical propranolol for infantile haemangiomas: a systematic review.外用普萘洛尔治疗婴儿血管瘤:一项系统评价。
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.气道婴儿血管瘤的管理
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.婴儿血管瘤的治疗:欧洲专家组的建议
Eur J Pediatr. 2015 Jul;174(7):855-65. doi: 10.1007/s00431-015-2570-0. Epub 2015 May 29.
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Ectopic cervical thymus associated with infant death: 2 case reports and literature review.
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Propranolol and infantile hemangiomas four years later: a systematic review.四年后的普萘洛尔与婴儿血管瘤:一项系统综述
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10
Propranolol for severe hemangiomas of infancy.普萘洛尔治疗婴儿重症血管瘤。
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