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移植物抗宿主病所致高钙血症一例罕见病例

An Unusual Case of Hypercalcemia Due to Graft-Versus-Host Disease.

作者信息

Fatima Paras, Czerwinski Vincent, Panthi Sujata, Choudhary Chitra

机构信息

Department of Internal Medicine, University of Kansas Medical Center, Kansas City, Kansas.

University of Kansas School of Medicine, Kansas City, Kansas.

出版信息

AACE Clin Case Rep. 2023 Dec 13;10(2):45-48. doi: 10.1016/j.aace.2023.12.002. eCollection 2024 Mar-Apr.

DOI:10.1016/j.aace.2023.12.002
PMID:38523852
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10958635/
Abstract

BACKGROUND/OBJECTIVE: Hypercalcemia is a common disorder with a wide differential and is most commonly related to malignancy and hyperparathyroidism. Hypercalcemia is a rarely reported consequence of graft-versus-host disease (GVHD) and may be related to a granulomatous manifestation of the common stem cell transplantation procedure.

CASE REPORT

A 67-year-old woman with a history of allogenic stem cell transplantation due to myelodysplastic syndrome presented to the bone marrow transplant clinic with dysphagia, muscle aches, and rash. She was found to have an extremely increased calcium and 1,25-dihydroxyvitamin D levels, which were ultimately corrected with administration of steroids and zoledronic acid.

DISCUSSION

While uncommon, granulomatous disease can lead to hypercalcemia via the activation of 1α-hydroxylase within macrophages, which, in turn, activates 1,25-dihydroxyvitamin D leading to an increased serum calcium level. GVHD is a common, variably presenting complication of bone marrow transplantation. Granulomatous processes related to GVHD may mediate hypercalcemia in patients with both increased calcium and 1,25-dihydroxyvitamin D levels.

CONCLUSION

This is a rare cause of calcitriol-mediated hypercalcemia associated with GVHD. There have been cases of granulomas associated with GVHD, and this could potentially lead to ectopic production of calcitriol. We deemed GVHD to be a likely cause of the patient's calcitriol-mediated hypercalcemia because we did not find another etiology that fit the clinical findings. Physician awareness of this complication and the appropriate workup will allow future researchers to properly elucidate the etiology of this rare complication.

摘要

背景/目的:高钙血症是一种常见疾病,鉴别诊断范围广泛,最常与恶性肿瘤和甲状旁腺功能亢进有关。高钙血症是移植物抗宿主病(GVHD)极少报道的后果,可能与常见干细胞移植程序的肉芽肿表现有关。

病例报告

一名67岁女性,有因骨髓增生异常综合征进行异基因干细胞移植的病史,因吞咽困难、肌肉疼痛和皮疹就诊于骨髓移植诊所。发现她的钙和1,25 - 二羟维生素D水平极度升高,最终通过给予类固醇和唑来膦酸得以纠正。

讨论

虽然罕见,但肉芽肿性疾病可通过巨噬细胞内1α - 羟化酶的激活导致高钙血症,这反过来又激活1,25 - 二羟维生素D,导致血清钙水平升高。GVHD是骨髓移植常见的、表现多样的并发症。与GVHD相关的肉芽肿过程可能在钙和1,25 - 二羟维生素D水平均升高的患者中介导高钙血症。

结论

这是与GVHD相关的维生素D介导的高钙血症的罕见原因。已有与GVHD相关的肉芽肿病例,这可能潜在地导致维生素D的异位产生。我们认为GVHD是患者维生素D介导的高钙血症的可能原因,因为我们未发现符合临床发现的其他病因。医生对这种并发症的认识以及适当的检查将使未来的研究人员能够正确阐明这种罕见并发症的病因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7b59/10958635/b13d9c6b975f/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7b59/10958635/b13d9c6b975f/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7b59/10958635/b13d9c6b975f/gr1.jpg

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本文引用的文献

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Granulomatous Myositis as a Manifestation of Chronic Graft-Versus-Host Disease: A Case Series and Review of the Literature.肉芽肿性肌炎作为慢性移植物抗宿主病的一种表现:病例系列及文献综述
Clin Lymphoma Myeloma Leuk. 2022 Jul;22(7):e459-e462. doi: 10.1016/j.clml.2022.01.004. Epub 2022 Jan 8.
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Sarcoidosis Following Hematopoietic Stem Cell Transplantation: Clinical Characteristics and HLA Associations.造血干细胞移植后发生的肉样瘤病:临床特征和 HLA 相关性。
Front Immunol. 2021 Oct 7;12:746996. doi: 10.3389/fimmu.2021.746996. eCollection 2021.
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The diagnostic and clinical significance of granulomas in gastrointestinal biopsies from haematopoietic transplant patients.
造血移植患者胃肠活检中肉芽肿的诊断和临床意义。
Histopathology. 2021 Apr;78(5):772-777. doi: 10.1111/his.14303. Epub 2021 Jan 28.
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Acute Graft-versus-Host Disease - Biologic Process, Prevention, and Therapy.急性移植物抗宿主病——生物学过程、预防与治疗
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Endocr Rev. 2016 Oct;37(5):521-547. doi: 10.1210/er.2016-1070. Epub 2016 Sep 2.
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