Bautista-Quach Marnelli A, Booth Cassie L, Kheradpour Albert, Zuppan Craig W, Rowsell Edward H, Weiss Lawrence, Wang Jun
Department of Pathology and Laboratory Medicine, Loma Linda, CA 92354, USA.
J Pediatr Hematol Oncol. 2013 May;35(4):315-20. doi: 10.1097/MPH.0b013e318279e392.
Mast cell diseases comprise a spectrum of disorders including cutaneous mastocytosis, indolent or aggressive systemic variants including leukemia, and unifocal tumor formations such as benign extracutaneous mastocytoma or aggressive mast cell sarcoma (MCS). Many mast cell diseases are associated with aberrancy of c-KIT proto-oncogene resulting in tyrosine kinase activity, typically exhibiting point mutation in codon 816. MCS is an exceedingly rare clinicopathologic entity characterized by a unifocal accumulation of neoplastic mast cells that grow in a locally destructive manner. We report a case in a 2-year-old boy who was initially diagnosed at 8 months of age with atypical cutaneous mastocytoma of the right ear with subsequent aggressive, destructive growth pattern; features that were most consistent with MCS. So far, MCS has been documented in the literature in at least 6 human cases. To the best of our knowledge, our case represents the first MCS in an infant. Thorough multimodal approach with strict follow-up is relevant in appropriately diagnosing this rare entity, particularly in differentiating this lesion from other neoplasms that are more likely to occur in infancy.
肥大细胞疾病包括一系列病症,包括皮肤肥大细胞增多症、惰性或侵袭性全身性变体(如白血病)以及单灶性肿瘤形成(如良性皮肤外肥大细胞瘤或侵袭性肥大细胞肉瘤(MCS))。许多肥大细胞疾病与c-KIT原癌基因异常有关,导致酪氨酸激酶活性,通常在密码子816处出现点突变。MCS是一种极其罕见的临床病理实体,其特征是肿瘤性肥大细胞单灶性聚集,呈局部破坏性生长方式。我们报告了一例2岁男孩的病例,该男孩最初在8个月大时被诊断为右耳非典型皮肤肥大细胞瘤,随后出现侵袭性、破坏性生长模式;这些特征与MCS最为一致。到目前为止,文献中至少记录了6例人类MCS病例。据我们所知,我们的病例是婴儿中的首例MCS。采用全面的多模式方法并进行严格随访,对于正确诊断这种罕见实体非常重要,特别是在将这种病变与更可能在婴儿期发生的其他肿瘤区分开来方面。