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嗜铬细胞瘤初次手术后46年发生的迟发性骨转移:一例报告

Late-Onset Bone Metastasis 46 Years After Initial Surgery for Pheochromocytoma: A Case Report.

作者信息

Ishizaki Fumio, Hiruma Kaede, Tani Yusuke, Sugino Hideaki, Sanami Tatsuro, Anraku Tsutomu, Ikeda Masahiro, Tasaki Masayuki, Saito Kazuhide, Tomita Yoshihiko

机构信息

Department of Urology, Molecular Oncology, Graduate School of Medical and Dental Sciences Niigata University Niigata Japan.

Division of Molecular and Diagnostic Pathology, Niigata University Graduate School of Medical and Dental Sciences Niigata University Niigata Japan.

出版信息

IJU Case Rep. 2025 Aug 13;8(5):533-538. doi: 10.1002/iju5.70086. eCollection 2025 Sep.

Abstract

INTRODUCTION

Pheochromocytoma is a rare catecholamine-producing tumor with metastatic potential. Recurrence after more than 40 years is exceptionally rare.

CASE PRESENTATION

During evaluation for ischemic colitis, a 71-year-old woman was found to have multiple bone metastases, possibly linked to catecholamine excess. She had undergone left adrenalectomy for pheochromocytoma at age 25. Bone biopsy confirmed metastatic pheochromocytoma, and immunohistochemical findings were similar to the original tumor. Urinary metanephrine and normetanephrine were markedly elevated. She declined systemic therapy and has remained clinically stable for 6 years, with her blood pressure well controlled on doxazosin.

CONCLUSION

This case illustrates a recurrence 46 years after adrenalectomy, potentially representing the longest reported interval to date. It highlights the silent and indolent nature of some metastatic pheochromocytomas and underscores the necessity of lifelong follow-up. The patient's stable course also emphasizes the clinical heterogeneity of metastatic pheochromocytoma and supports the need for individualized follow-up and treatment strategies.

摘要

引言

嗜铬细胞瘤是一种罕见的具有转移潜能的儿茶酚胺分泌肿瘤。40多年后复发极为罕见。

病例介绍

在对一名71岁女性进行缺血性结肠炎评估期间,发现她有多处骨转移,可能与儿茶酚胺过量有关。她25岁时因嗜铬细胞瘤接受了左肾上腺切除术。骨活检证实为转移性嗜铬细胞瘤,免疫组化结果与原发肿瘤相似。尿间甲肾上腺素和去甲间甲肾上腺素明显升高。她拒绝了全身治疗,6年来一直保持临床稳定,服用多沙唑嗪后血压得到良好控制。

结论

本病例显示肾上腺切除术后46年复发,这可能是迄今为止报道的最长间隔时间。它凸显了一些转移性嗜铬细胞瘤的隐匿性和惰性,强调了终身随访的必要性。患者的稳定病程也强调了转移性嗜铬细胞瘤的临床异质性,并支持需要个性化的随访和治疗策略。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fea4/12408166/74ebf3cddb49/IJU5-8-533-g002.jpg

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