5258Mount Sinai Medical Center, Miami Beach, FL, USA.
158263Florida International University, Miami, FL, USA.
Int J Surg Pathol. 2021 May;29(3):321-326. doi: 10.1177/1066896920942891. Epub 2020 Jul 17.
Genetically driven tissue destruction followed by remodeling in adult polycystic kidney disease (APKD) raises the possibility of malignant transformation. Renal cell carcinoma (RCC) associated with APKD has been frequently reported in the literature; however, only a few cases of nonepithelial neoplasms arising in APKD have been described so far. Histiocytic sarcoma (HS) is a lymphohematopoietic malignant neoplasm that accounts for less than 1% of hematologic malignancies. In this article, we describe a case of primary HS occurring in a 61-year-old man with end-stage renal disease secondary to APKD. This is the first reported case of primary HS in the setting of APKD. The aberrant h-caldesmon expression seen in this case is another novel finding that has previously not been described. This case highlights the importance of morphology in guiding diagnostic workup and reiterates the necessity of maintaining a high index of suspicion for neoplastic entities in APKD.
在成人多囊肾病 (APKD) 中,遗传驱动的组织破坏随后进行重塑,这增加了恶性转化的可能性。与 APKD 相关的肾细胞癌 (RCC) 在文献中经常被报道;然而,迄今为止,仅描述了少数几种在 APKD 中发生的非上皮性肿瘤病例。组织细胞肉瘤 (HS) 是一种淋巴造血系统恶性肿瘤,占血液系统恶性肿瘤的不到 1%。本文描述了一例发生在一名 61 岁男性的原发性 HS 病例,该患者患有由 APKD 引起的终末期肾病。这是 APKD 背景下首例原发性 HS 病例报告。在这种情况下观察到的异常 h-caldesmon 表达是另一个以前未描述的新发现。该病例强调了形态学在指导诊断性检查中的重要性,并再次强调了在 APKD 中保持对肿瘤实体高度怀疑的必要性。