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伴有广泛骨骼受累及高钙血症的边缘区淋巴瘤：1例罕见病例并文献系统综述

Marginal Zone Lymphoma With Extensive Skeletal Involvement and Hypercalcemia: A Rare Case With a Systematic Review of the Literature.

作者信息

Mondal Soumayan, Mahajan Apoorav, Sengupta Piyali, Sahoo Krushna K, Misra Purusottam, Patro Shubhransu, Pattnaik Sidharth S

机构信息

Internal Medicine, Kalinga Institute of Medical Sciences, Bhubaneswar, IND.

Pathology, Kalinga Institute of Medical Sciences, Bhubaneswar, IND.

出版信息

Cureus. 2025 Apr 27;17(4):e83100. doi: 10.7759/cureus.83100. eCollection 2025 Apr.

DOI:10.7759/cureus.83100
PMID:40438858
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12119074/
Abstract

Marginal zone lymphoma (MZL) rarely presents with skeletal involvement or hypercalcemia, making diagnosis and management particularly challenging. We report a rare case of a 44-year-old woman with extensive lytic bone lesions, hypercalcemia, and no lymphadenopathy, initially suspected to have multiple myeloma. Imaging revealed widespread osteolytic involvement, and a bone marrow biopsy confirmed MZL. She was successfully treated with rituximab-bendamustine and bisphosphonates. To contextualize this presentation, we conducted a systematic review of case reports and series describing non-Hodgkin lymphoma with skeletal disease and hypercalcemia. Across 16 studies, diffuse large B-cell lymphoma was the most common subtype, but mechanisms of hypercalcemia, including cytokine-driven osteoclast activation, parathyroid hormone-related protein (PTHrP) secretion, and vitamin D dysregulation, were consistent across subtypes. Our case illustrates the importance of considering lymphoma in patients with unexplained lytic lesions and hypercalcemia. Early biopsy, positron emission tomography-computed tomography (PET-CT) imaging, and appropriate therapy can significantly improve outcomes. This case highlights the diagnostic complexity of bone-involved lymphoma and is supported by findings from a systematic review, emphasizing the need for increased recognition of this underreported presentation.

摘要

边缘区淋巴瘤（MZL）很少出现骨骼受累或高钙血症，这使得诊断和治疗极具挑战性。我们报告了一例罕见病例，一名44岁女性，有广泛的溶骨性骨病变、高钙血症，且无淋巴结病，最初怀疑患有多发性骨髓瘤。影像学检查显示广泛的溶骨性病变，骨髓活检确诊为MZL。她接受利妥昔单抗-苯达莫司汀和双膦酸盐治疗后获得成功。为了更好地理解这一病例表现，我们对描述伴有骨骼疾病和高钙血症的非霍奇金淋巴瘤的病例报告和系列研究进行了系统综述。在16项研究中，弥漫性大B细胞淋巴瘤是最常见的亚型，但高钙血症的机制，包括细胞因子驱动的破骨细胞活化、甲状旁腺激素相关蛋白（PTHrP）分泌和维生素D调节异常，在各亚型中是一致的。我们的病例说明了在不明原因的溶骨性病变和高钙血症患者中考虑淋巴瘤的重要性。早期活检、正电子发射断层扫描-计算机断层扫描（PET-CT）成像和适当的治疗可显著改善预后。该病例突出了骨受累淋巴瘤的诊断复杂性，并得到了系统综述结果的支持，强调需要提高对这种报告不足的病例表现的认识。

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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be33/12119074/03b257fced56/cureus-0017-00000083100-i10.jpg
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本文引用的文献

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Cureus. 2024 Jan 8;16(1):e51856. doi: 10.7759/cureus.51856. eCollection 2024 Jan.
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Multiple osteolytic primary peripheral T-cell bone lymphoma: the first case report.多发性溶骨性原发性周围 T 细胞骨淋巴瘤：首例报告。
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Hypercalcaemia, renal dysfunction, anaemia and bone lesions (CRAB) do not always represent multiple myeloma: diffuse large B cell lymphoma presenting with CRAB symptoms in a 69-year-old man.
高钙血症、肾功能不全、贫血和骨病变（CRAB）并不总是代表多发性骨髓瘤：一名69岁男性患者出现CRAB症状的弥漫性大B细胞淋巴瘤。
BMJ Case Rep. 2019 Aug 4;12(8):e229070. doi: 10.1136/bcr-2018-229070.
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FDG-PET/CT in the management of lymphomas: current status and future directions.FDG-PET/CT 在淋巴瘤治疗中的应用：现状与未来方向。
J Intern Med. 2018 Oct;284(4):358-376. doi: 10.1111/joim.12813. Epub 2018 Jul 24.
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