Department of Histopathology, Addenbrookes Hospital and Cambridge Breast Unit, Cambridge UK.
Am J Surg Pathol. 2010 Apr;34(4):584-8. doi: 10.1097/PAS.0b013e3181d39a3d.
Erdheim-Chester disease is a rare non-Langerhans cell histiocytosis of unknown etiology, the commonest sites of involvement being the long bones, skin, orbit, pituitary and retroperitoneum. Breast involvement is rare, with only four reported cases in the English literature. We present a case of a 78-year-old female presenting with bilateral clinically malignant breast masses, with mammographic and ultrasound findings suggestive of locally advanced bilateral breast cancer. Core biopsies from both breasts showed identical features, with a diffuse xanthomatous infiltrate with scattered Touton-type giant cells and a patchy lymphocytic infiltrate. The cells were CD68 positive, and negative for S100, CD1a and a broad panel of cytokeratins. The patient has a background history of cerebrovascular disease with carotid artery stenosis, and subsequently developed rapid restenosis after carotid endarterectomy. With the combined clinical history and classic histological findings in the breast, a diagnosis of Erdheim-Chester disease was made. This is the fifth case report of Erdheim-Chester disease involving the breast, and only the second case with breast lesions as the presenting symptom. Perivascular infiltration is also a rare but recognized presentation of Erdheim-Chester disease. Histiocytic proliferations including ECD can mimic breast carcinoma clinically, radiologically, and histologically, and should be considered in the differential diagnosis of breast mass lesions.
厄尔-当-切斯特病是一种罕见的非朗格汉斯细胞组织细胞增生症,其最常见的受累部位是长骨、皮肤、眼眶、垂体和腹膜后。乳腺受累罕见,英文文献中仅报道了 4 例。我们报告了一例 78 岁女性,表现为双侧临床上恶性乳腺肿块,乳腺 X 线摄影和超声检查提示双侧局部晚期乳腺癌。双侧乳腺的核心活检均显示相同的特征,弥漫性黄色瘤样浸润伴散在的托顿样巨细胞和局灶性淋巴细胞浸润。细胞 CD68 阳性,S100、CD1a 和广泛的细胞角蛋白阴性。患者有脑血管病伴颈动脉狭窄病史,颈动脉内膜切除术(carotid endarterectomy)后迅速再狭窄。结合患者的临床病史和乳腺的典型组织学发现,诊断为厄尔-当-切斯特病。这是第五例累及乳腺的厄尔-当-切斯特病病例报告,也是仅有的以乳腺病变为首发症状的第二例病例。血管周围浸润也是厄尔-当-切斯特病的一种罕见但已被认识到的表现。组织细胞增生症包括 ECD 在临床上、影像学上和组织学上均可模拟乳腺癌,在鉴别乳腺肿块病变时应考虑到这一点。