Department of Dermatology, Kansai Medical University Medical Center, Osaka, Japan.
Department of Dermatology, Aichi Medical University School of Medicine, Nagakute, Japan.
J Dermatol. 2018 Dec;45(12):1444-1447. doi: 10.1111/1346-8138.14673. Epub 2018 Oct 15.
A 34-year-old Japanese man presented with an indolent nodule on the right flank. Computed tomography of the chest and abdomen demonstrated a large nodule measuring 55 mm × 50 mm in the abdominal oblique muscle layer of the right flank, and several small nodules were seen in the muscle layer throughout the body and subcutaneous tissue of the lower abdomen. F-fluorodeoxyglucose-positron emission tomography/computed tomography revealed nodular lesions in the bilateral parotid glands, bilateral cervical lymph nodes and lower lobe of the right lung. Intermittently, ground-glass shadows developed in the bilateral lungs. Histologically, sheet-like nodules in the abdominal oblique muscle layer and parotid gland were composed of large polygonal cells with convoluted nuclei and ample eosinophilic cytoplasm. Several lymphocytes and considerable eosinophils were intermingled. Lung biopsy demonstrated an inflammatory infiltrate of lymphocytes and considerable eosinophils in the alveoli. Immunohistochemically, polygonal cells were positive for S100 protein and CD1a, but negative for langerin and BRAF . Some cells were positive for CD68. Electron microscopy demonstrated histiocytic cells with phagosomes and interdigitating processes. However, no Birbeck granules were observed. Eosinophilia was seen in the peripheral blood. Multifocal nodules and ground-glass shadows gradually diminished following systemic administration of oral prednisolone. We describe a case of indeterminate dendritic cell neoplasm with multifocal involvement of the muscle, subcutis, lymph node and parotid gland accompanied by chronic eosinophilic pneumonia that was successfully treated by systemic steroid therapy. Neither muscular nor parotid indeterminate dendritic cell neoplasms accompanied by eosinophilic pneumonia have been previously reported.
一位 34 岁的日本男性因右腰部惰性结节就诊。胸部和腹部的计算机断层扫描显示右腰部腹斜肌层有一个 55mm×50mm 的大结节，全身肌肉层和下腹皮下组织可见多个小结节。F-氟脱氧葡萄糖正电子发射断层扫描/计算机断层扫描显示双侧腮腺、双侧颈部淋巴结和右下肺叶有结节性病变。间断性地，双肺出现磨玻璃影。组织学上，腹斜肌层和腮腺中的片状结节由具有卷曲核和丰富嗜酸性细胞质的大多边形细胞组成。混杂有几个淋巴细胞和相当数量的嗜酸性粒细胞。肺活检显示肺泡中有淋巴细胞和相当数量的嗜酸性粒细胞浸润。免疫组织化学染色显示，多边形细胞 S100 蛋白和 CD1a 阳性，但朗格汉斯细胞和 BRAF 阴性。一些细胞 CD68 阳性。电子显微镜显示具有吞噬体和交错突起的组织细胞。然而，未观察到 Birbeck 颗粒。外周血嗜酸性粒细胞增多。全身给予口服泼尼松龙后，多灶性结节和磨玻璃影逐渐减少。我们描述了一例伴有多灶性肌肉、皮下组织、淋巴结和腮腺累及的未确定树突状细胞瘤，伴有慢性嗜酸性粒细胞性肺炎，经全身类固醇治疗成功。以前没有报道过伴有嗜酸性粒细胞肺炎的肌肉或腮腺未确定树突状细胞瘤。
