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酷似纤维性纵隔炎的B-急性淋巴细胞白血病/淋巴细胞淋巴瘤:病例报告及诊断见解

B-Acute Lymphoblastic Leukemia/Lymphoblastic Lymphoma Mimicking Fibrosing Mediastinitis: A Case Report and Diagnostic Insight.

作者信息

Kitamura Aya, Yanagi Shigehisa, Shide Kotaro, Sato Yuichiro, Kamiunten Ayako, Yamanari Yasuhiro, Kitamura Akiko, Sumiyoshi Makoto, Oda Yasuharu, Tsubouchi Hironobu, Shimoda Kazuya, Miyazaki Taiga

机构信息

Division of Respirology, Rheumatology, Infectious Diseases, and Neurology, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, Japan.

Division of Hematology, Diabetes, and Endocrinology, Department of Internal Medicine, Faulty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, Japan.

出版信息

Am J Case Rep. 2024 Dec 30;25:e945804. doi: 10.12659/AJCR.945804.

Abstract

BACKGROUND Fibrosing mediastinitis (FM) is a rare, fibroproliferative disorder within the mediastinum. It is extremely rare for hematologic malignancies to develop as FM. CASE REPORT A 32-year-old Japanese man with a 1-month history of headache and 2-week history of facial swelling underwent chest computed tomography (CT); a diffuse mass-like lesion was revealed in the anterior mediastinum with severe stenosis of vital mediastinal organs. After a surgical biopsy, an initial diagnosis of idiopathic FM was made. The FM lesions responded mildly to corticosteroids but recurred repeatedly. Sixteen months after the treatment initiation, blasts appeared in the peripheral blood (PB), and the patient was diagnosed with B-acute lymphoblastic leukemia/lymphoblastic lymphoma (B-ALL/LBL). Chemotherapy led to complete remission of the B-ALL/LBL and almost complete disappearance of FM-like lesions. Immunohistochemistry of the mediastinal biopsy specimen taken before the blasts' appearance in PB demonstrated a CD34/CD7/terminal deoxynucleotidyl transferase-positive population, an identical pattern of expression common to the blasts in the patient's PB and bone marrow. CONCLUSIONS This is the first case report of B-ALL/LBL presenting as FM. This case underscores the importance of considering the possibility of latent hematologic malignancy even in the absence of new symptoms other than those caused by FM lesions for a long period of time. This is the first demonstration that leukemia cells may be present in the FM lesions from the initial stage of disease onset. Even if a diagnosis of idiopathic FM is confirmed, continued suspicion of the presence of hematologic malignancy is vital for improving patient outcomes.

摘要

背景

纤维性纵隔炎(FM)是一种罕见的纵隔纤维增生性疾病。血液系统恶性肿瘤发展为FM极为罕见。

病例报告

一名32岁的日本男性,有1个月头痛病史和2周面部肿胀病史,接受了胸部计算机断层扫描(CT);前纵隔发现弥漫性肿块样病变,重要纵隔器官严重狭窄。手术活检后,初步诊断为特发性FM。FM病变对皮质类固醇反应轻微,但反复复发。治疗开始16个月后,外周血(PB)中出现原始细胞,患者被诊断为B淋巴细胞急性淋巴细胞白血病/淋巴瘤(B-ALL/LBL)。化疗导致B-ALL/LBL完全缓解,FM样病变几乎完全消失。在PB中出现原始细胞之前采集的纵隔活检标本的免疫组织化学显示CD34/CD7/末端脱氧核苷酸转移酶阳性群体,与患者PB和骨髓中的原始细胞表达模式相同。

结论

这是首例以FM形式出现的B-ALL/LBL病例报告。该病例强调,即使在很长一段时间内除FM病变引起的症状外没有新症状,也需考虑潜在血液系统恶性肿瘤的可能性。这是首次证明白血病细胞可能在疾病发病初期就存在于FM病变中。即使确诊为特发性FM,持续怀疑血液系统恶性肿瘤的存在对改善患者预后至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/08fc/11694771/bdd55b21bdde/amjcaserep-25-e945804-g001.jpg

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