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用促性腺激素释放激素治疗的酷似肌瘤的子宫炎性肌纤维母细胞瘤:一例报告

Uterine Inflammatory Myofibroblastic Tumor Mimicking Myoma Treated With Gonadotropin-Releasing Hormone Therapy: A Case Report.

作者信息

Yorita Kenji, Hirano Koki, Nakatani Kimiko

机构信息

Department of Diagnostic Pathology, Japanese Red Cross Kochi Hospital, Kochi, JPN.

Department of Obstetrics and Gynecology, Japanese Red Cross Kochi Hospital, Kochi, JPN.

出版信息

Cureus. 2025 May 24;17(5):e84722. doi: 10.7759/cureus.84722. eCollection 2025 May.

DOI:10.7759/cureus.84722
PMID:40557015
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12185251/
Abstract

Inflammatory myofibroblastic tumors (IMTs) of the uterus are a rare entity that can be challenging to distinguish from leiomyomas, both radiologically and pathologically. No previous reports have documented the response of uterine IMT to gonadotropin-releasing hormone (GnRH) therapy. A 47-year-old Japanese woman presented to our hospital with excessive menstruation and uterine prolapse. Magnetic resonance imaging (MRI) revealed four well-demarcated uterine masses, with the largest measuring 81 mm. Myomas were suspected. Oral GnRH therapy was initiated to relieve the symptoms and reduce the preoperative volume. After four months, follow-up MRI showed a reduction in the largest mass to 62 mm and increased hyperintense areas on T2-weighted images. At six months, the patient underwent laparoscopic hysterectomy and colporrhaphy. Histopathological examination revealed that the largest tumor was an IMT with abundant myxoid matrix, positive for anaplastic lymphoma kinase (ALK), and ALK gene rearrangement. The remaining three masses were diagnosed as usual-type leiomyomas. This is the first reported case of uterine IMT treated with GnRH therapy, which resulted in notable tumor shrinkage. This case highlights both the potential therapeutic approach for uterine IMT and the diagnostic difficulty in distinguishing it from leiomyomas preoperatively.

摘要

子宫炎性肌纤维母细胞瘤(IMT)是一种罕见的疾病,在影像学和病理学上都很难与平滑肌瘤区分开来。此前尚无关于子宫IMT对促性腺激素释放激素(GnRH)治疗反应的报道。一名47岁的日本女性因月经过多和子宫脱垂前来我院就诊。磁共振成像(MRI)显示有四个边界清晰的子宫肿块,最大的肿块为81毫米。怀疑为肌瘤。开始口服GnRH治疗以缓解症状并减少术前体积。四个月后,随访MRI显示最大肿块缩小至62毫米,T2加权图像上高信号区域增加。六个月时,患者接受了腹腔镜子宫切除术和阴道修补术。组织病理学检查显示,最大的肿瘤为IMT,伴有丰富的黏液样基质,间变性淋巴瘤激酶(ALK)呈阳性,且存在ALK基因重排。其余三个肿块被诊断为普通型平滑肌瘤。这是首例报道的用GnRH治疗的子宫IMT病例,该治疗导致肿瘤明显缩小。该病例既凸显了子宫IMT潜在的治疗方法,也凸显了术前将其与平滑肌瘤区分开来的诊断困难。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/45227a47a111/cureus-0017-00000084722-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/d661cba41043/cureus-0017-00000084722-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/9faee1869f9a/cureus-0017-00000084722-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/45227a47a111/cureus-0017-00000084722-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/d661cba41043/cureus-0017-00000084722-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/9faee1869f9a/cureus-0017-00000084722-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ad1d/12185251/45227a47a111/cureus-0017-00000084722-i03.jpg

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本文引用的文献

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The clinicopathologic features of uterine inflammatory myofibroblastic tumor: A case report.子宫炎性肌纤维母细胞瘤的临床病理特征:一例报告
Medicine (Baltimore). 2025 Jan 17;104(3):e41310. doi: 10.1097/MD.0000000000041310.
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Relugolix reduces leiomyoma extracellular matrix production via the transforming growth factor-beta pathway.
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Histopathologic and Molecular Characterization of Uterine Leiomyoma-like Inflammatory Myofibroblastic Tumor: Comparison to Molecular Subtypes of Uterine Leiomyoma.子宫平滑肌瘤样炎症性肌纤维母细胞瘤的组织病理学和分子特征:与子宫平滑肌瘤分子亚型的比较。
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World J Surg Oncol. 2021 Nov 20;19(1):331. doi: 10.1186/s12957-021-02438-5.
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MR imaging findings of unusual leiomyoma and malignant uterine myometrial tumors: what the radiologist should know.磁共振成像对罕见的平滑肌瘤和恶性子宫平滑肌肿瘤的表现:放射科医生应该知道的。
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Inflammatory Myofibroblastic Tumors of the Female Genital Tract Are Under-recognized: A Low Threshold for ALK Immunohistochemistry Is Required.女性生殖道炎性肌纤维母细胞瘤未得到充分认识:需要对ALK免疫组织化学进行低阈值检测。
Am J Surg Pathol. 2017 Oct;41(10):1433-1442. doi: 10.1097/PAS.0000000000000909.
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Inflammatory myofibroblastic tumor of the uterus: a clinicopathological, immunohistochemical, and molecular analysis of 13 cases highlighting their broad morphologic spectrum.子宫炎性肌纤维母细胞瘤:13 例的临床病理、免疫组化和分子分析,突出其广泛的形态谱。
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Myxoid Leiomyosarcoma of the Uterus: A Clinicopathologic Analysis of 30 Cases and Review of the Literature With Reappraisal of Its Distinction From Other Uterine Myxoid Mesenchymal Neoplasms.子宫黏液样平滑肌肉瘤:30例临床病理分析及文献复习,并重新评估其与其他子宫黏液样间叶性肿瘤的鉴别诊断
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STUMP un"stumped": anti-tumor response to anaplastic lymphoma kinase (ALK) inhibitor based targeted therapy in uterine inflammatory myofibroblastic tumor with myxoid features harboring DCTN1-ALK fusion.STUMP不再“棘手”:具有黏液样特征且携带DCTN1-ALK融合基因的子宫炎性肌纤维母细胞瘤对基于间变性淋巴瘤激酶(ALK)抑制剂的靶向治疗产生抗肿瘤反应。
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