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罕见的皮下淀粉样瘤伴局限性淋巴浆细胞淋巴瘤病例:诊断挑战与治疗考虑。

A Rare Case of Subcutaneous Amyloidoma Associated with Localized Lymphoplasmacytic Lymphoma: Diagnostic Challenges and Treatment Considerations.

机构信息

Department of Pathology, ASST Santi Paolo e Carlo, Milan, Italy.

Department of Pathology, UZ Leuven, University Hospitals, Leuven, Belgium.

出版信息

Am J Case Rep. 2023 Oct 2;24:e940789. doi: 10.12659/AJCR.940789.

DOI:10.12659/AJCR.940789
PMID:37779307
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10556539/
Abstract

BACKGROUND AL amyloidomas are solitary, localized, tumor-like deposits of immunoglobulin light-chain-derived amyloid fibrils in the absence of systemic amyloidosis. A rare entity, they have been described in various anatomical sites, typically in spatial association with a sparse lymphoplasmacytic infiltrate, ultimately corresponding to a clonal, malignant, lymphomatous disorder accounting for the amyloidogenic activity. Most frequently, the amyloidoma-associated hematological disorder corresponds to either a solitary plasmacytoma or an extranodal marginal zone lymphoma of MALT. Much rarer is the association with lymphoplasmacytic lymphoma, which by itself is usually a bone marrow-bound disorder with systemic burden. The almost anecdotic combination of an amyloidoma and a localized lymphoplasmacytic lymphoma deserves attention, as it entails a thorough diagnostic workup to exclude systemic involvement and a proportionate therapeutic approach to avoid overtreatment. A review of the literature provides an insight on pathogenesis and prognosis, and can assist both pathologists and clinicians in establishing optimal patient management strategies. CASE REPORT We herein report the incidental finding of a subcutaneous amyloidoma caused by a spatially related, similarly localized lymphoplasmacytic lymphoma diagnosed in a 54-year-old female patient with no other disease localizations and a complete remission following 2 subsequent surgical excisions. CONCLUSIONS Whatever the specific combination of an amyloidoma and the related hematological neoplasm, a multidisciplinary collaboration and a comprehensive clinical-pathological staging are warranted to exclude systemic involvement and identify patients with localized diseases who would benefit from local active treatment and close follow-up.

摘要

背景

AL 淀粉样瘤是一种无系统性淀粉样变性的单发性、局限性、肿瘤样的免疫球蛋白轻链衍生淀粉样纤维沉积。这是一种罕见的实体,已在各种解剖部位被描述,通常与稀疏的淋巴浆细胞浸润有关,最终对应于一种克隆性、恶性、淋巴瘤性疾病,其导致淀粉样变性活性。最常见的是淀粉样瘤相关的血液学疾病对应于单一浆细胞瘤或结外黏膜相关边缘区淋巴瘤(MALT)。更罕见的是与淋巴浆细胞淋巴瘤相关,其本身通常是一种骨髓受累的疾病,伴有全身受累。淀粉样瘤和局部淋巴浆细胞淋巴瘤的几乎轶事性组合值得关注,因为需要进行彻底的诊断性检查以排除系统性受累,并采取适当的治疗方法以避免过度治疗。对文献的回顾提供了对发病机制和预后的深入了解,并可以帮助病理学家和临床医生制定最佳的患者管理策略。

病例报告

我们在此报告了一例 54 岁女性患者偶然发现的皮下淀粉样瘤,该患者无其他疾病局部定位,且在随后的 2 次手术切除后完全缓解。淀粉样瘤由空间上相关的、同样局限的淋巴浆细胞淋巴瘤引起。

结论

无论淀粉样瘤和相关血液学肿瘤的具体组合如何,都需要多学科合作和全面的临床病理分期,以排除系统性受累并识别出局部疾病患者,这些患者将受益于局部积极治疗和密切随访。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/2b2abce35a05/amjcaserep-24-e940789-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/40c3cf753fcd/amjcaserep-24-e940789-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/b670abbe7c43/amjcaserep-24-e940789-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/181e263904c5/amjcaserep-24-e940789-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/2b2abce35a05/amjcaserep-24-e940789-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/40c3cf753fcd/amjcaserep-24-e940789-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/b670abbe7c43/amjcaserep-24-e940789-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/181e263904c5/amjcaserep-24-e940789-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ae/10556539/2b2abce35a05/amjcaserep-24-e940789-g004.jpg

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本文引用的文献

1
Amyloid nomenclature 2022: update, novel proteins, and recommendations by the International Society of Amyloidosis (ISA) Nomenclature Committee.淀粉样变命名 2022:更新、新型蛋白质以及国际淀粉样变性学会(ISA)命名委员会的建议。
Amyloid. 2022 Dec;29(4):213-219. doi: 10.1080/13506129.2022.2147636. Epub 2022 Nov 24.
2
AL amyloidosis with a localized B cell neoplasia.伴有局部 B 细胞肿瘤的 AL 淀粉样变性。
Virchows Arch. 2019 Mar;474(3):353-363. doi: 10.1007/s00428-019-02527-7. Epub 2019 Jan 24.
3
Cerebral Amyloidoma Resulting from Central Nervous System Lymphoplasmacytic Lymphoma: A Case Report and Literature Review.
中枢神经系统淋巴浆细胞淋巴瘤导致的脑淀粉样瘤:一例报告及文献复习
Case Rep Pathol. 2018 Jun 26;2018:5083234. doi: 10.1155/2018/5083234. eCollection 2018.
4
A tale of the unexpected: Amyloidoma associated with intracerebral lymphoplasmacytic lymphoma.一则意外病例:与脑内淋巴浆细胞淋巴瘤相关的淀粉样瘤
J Neurol Sci. 2015 Dec 15;359(1-2):404-8. doi: 10.1016/j.jns.2015.09.372. Epub 2015 Oct 23.
5
Localized AL amyloidosis: a suicidal neoplasm?局部性 AL 淀粉样变性:自杀性肿瘤?
Ups J Med Sci. 2012 May;117(2):244-50. doi: 10.3109/03009734.2012.654861. Epub 2012 Feb 15.
6
Salivary gland lymphoplasmacytoid lymphoma with nodular cutaneous amyloid deposition and lambda chain paraproteinaemia.伴有结节性皮肤淀粉样沉积和λ链副蛋白血症的涎腺淋巴浆细胞样淋巴瘤。
Br J Dermatol. 1996 Aug;135(2):327-9.
7
Local amyloid deposits in a primary central nervous system lymphoma. Study of a stereotactic brain biopsy.原发性中枢神经系统淋巴瘤中的局部淀粉样蛋白沉积。立体定向脑活检研究。
Clin Neuropathol. 1990 Sep-Oct;9(5):231-3.