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胸腺瘤样窦组织细胞增生症:一例报告。

Thymic Rosai-Dorfmann disease: a case report.

机构信息

Department of Cardiothoracic Surgery, The First People's Hospital of Neijiang, Sichuan, 641000, China.

Department of Pathology, The First People's Hospital of Neijiang, Sichuan, 641000, China.

出版信息

J Cardiothorac Surg. 2024 Jun 21;19(1):351. doi: 10.1186/s13019-024-02919-0.

DOI:10.1186/s13019-024-02919-0
PMID:38907282
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11191219/
Abstract

BACKGROUND

Rosai-Dorfman disease (RDD), known as sinus histiocytosis with massive lymphadenopathy, commonly involves lymph nodes in the neck or mediastinum, although extranodal involvement is observed in approximately 40% of RDD patients. RDD involving only the thymus has rarely been reported. Here, we report a case of RDD originating in the thymus. The lesion was surgically removed, and a cure was finally achieved. There was no recurrence after telephone follow-up for 3 years.

CASE PRESENTATION

A 52-year-old male was accidentally found to have a 7 × 6 cm anterior mediastinum lump by chest computed tomography (CT). The mediastinal lesion was resected by surgery, and postoperative pathology revealed RDD originating from the thymus. Regular telephone follow-up after surgery lasted 3 years and showed that the patient remained in good condition without any relevant symptoms.

CONCLUSIONS

RDD originating in the thymus cannot be characterized from CT images and is easily misdiagnosed as a traditional mediastinal tumor. This is mainly because there is so little disease in this area that physicians are not aware of it. We report this case with the hope that clinicians will have a better understanding of this disease. According to our follow-up results, surgery is an effective means of treatment.

摘要

背景

罗道尔夫-多夫曼病(RDD),又称窦组织细胞增生伴巨大淋巴结病,常累及颈部或纵隔淋巴结,但约 40%的 RDD 患者存在结外累及。仅累及胸腺的 RDD 很少见。在此,我们报告一例起源于胸腺的 RDD。病变通过手术切除,最终治愈。电话随访 3 年无复发。

病例介绍

一名 52 岁男性因胸部 CT 偶然发现前纵隔 7×6cm 肿块。纵隔病变通过手术切除,术后病理显示 RDD 起源于胸腺。手术后定期电话随访 3 年,患者状况良好,无相关症状。

结论

起源于胸腺的 RDD 不能从 CT 图像上进行特征描述,容易误诊为传统纵隔肿瘤。这主要是因为该区域的疾病非常少见,医生没有意识到。我们报告这个病例,希望临床医生能更好地了解这种疾病。根据我们的随访结果,手术是一种有效的治疗手段。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a198/11191219/9ae2f7aa11b8/13019_2024_2919_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a198/11191219/bca89bb2c8f4/13019_2024_2919_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a198/11191219/9ae2f7aa11b8/13019_2024_2919_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a198/11191219/bca89bb2c8f4/13019_2024_2919_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a198/11191219/9ae2f7aa11b8/13019_2024_2919_Fig2_HTML.jpg

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本文引用的文献

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An extremely rare case of Rosai-Dorfman disease in the thymus.胸腺 Rosai-Dorfman 病极罕见病例报告。
J Cardiothorac Surg. 2021 Jul 31;16(1):212. doi: 10.1186/s13019-021-01595-8.
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An extremely rare case of Rosai-Dorfman disease in the spleen.脾脏 Rosai-Dorfman 病极为罕见。
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Rosai Dorfman Disease -A rare case of cervical lymphadenopathy.罗萨伊-多夫曼病——一例罕见的颈部淋巴结病病例。
Clin Case Rep. 2020 Aug 26;8(12):2712-2715. doi: 10.1002/ccr3.3255. eCollection 2020 Dec.
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Consensus recommendations for the diagnosis and clinical management of Rosai-Dorfman-Destombes disease.罗萨-多夫曼-德斯东贝病的诊断和临床管理共识建议。
Blood. 2018 Jun 28;131(26):2877-2890. doi: 10.1182/blood-2018-03-839753. Epub 2018 May 2.
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Rosai-Dorfman disease: a report of eight cases in a tertiary care center and a review of the literature.罗萨伊-多夫曼病:一家三级医疗中心的8例报告及文献综述
Braz J Med Biol Res. 2015 Jan;48(1):6-12. doi: 10.1590/1414-431X20144110. Epub 2014 Oct 10.
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Rosai-Dorfman disease: tumor biology, clinical features, pathology, and treatment.罗萨伊-多夫曼病:肿瘤生物学、临床特征、病理学及治疗
Cancer Control. 2014 Oct;21(4):322-7. doi: 10.1177/107327481402100408.
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Steroid-resistant extranodal rosai-dorfman disease of cheek mass and ptosis treated with radiation therapy.采用放射治疗的类固醇抵抗性颊部肿块和上睑下垂的结外罗道病
Case Rep Hematol. 2013;2013:428297. doi: 10.1155/2013/428297. Epub 2013 May 2.
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Primary splenic diffuse large B-cell lymphoma in a patient with thymus Rosai-Dorfman disease.胸腺 Rosai-Dorfman 病患者的原发性脾脏弥漫性大 B 细胞淋巴瘤。
Am J Med Sci. 2012 Aug;344(2):155-9. doi: 10.1097/MAJ.0b013e31824e940d.
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Cutaneous Rosai-Dorfman disease with increased number of eosinophils: coincidence or histologic variant?伴有嗜酸性粒细胞增多的皮肤 Rosai-Dorfman 病:偶然还是组织学变异?
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Radiotherapy for steroid-resistant laryngeal Rosai-Dorfman disease.放疗治疗类固醇抵抗性喉 Rosai-Dorfman 病。
Curr Oncol. 2011 Jun;18(3):e158-62. doi: 10.3747/co.v18i3.761.