Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, USA.
Mod Pathol. 2012 Jul;25(7):983-92. doi: 10.1038/modpathol.2012.38. Epub 2012 Mar 2.
CD30-positive T-cell lymphoproliferative disorders are classified as cutaneous (primary cutaneous anaplastic large cell lymphoma and lymphomatoid papulosis) or systemic. As extent of disease dictates prognosis and treatment, patients with skin involvement need clinical staging to determine whether systemic lymphoma also is present. Similar processes may involve mucosal sites of the head and neck, constituting a spectrum that includes both neoplasms and reactive conditions (eg, traumatic ulcerative granuloma with stromal eosinophilia). However, no standard classification exists for mucosal CD30-positive T-cell lymphoproliferations. To improve our understanding of these processes, we identified 15 such patients and examined clinical presentation, treatment and outcome, morphology, phenotype using immunohistochemistry, and genetics using gene rearrangement studies and fluorescence in situ hybridization. The 15 patients (11 M, 4 F; mean age, 57 years) had disease involving the oral cavity/lip/tongue (9), orbit/conjunctiva (3) or nasal cavity/sinuses (3). Of 14 patients with staging data, 7 had mucosal disease only; 2 had mucocutaneous disease; and 5 had systemic anaplastic large cell lymphoma. Patients with mucosal or mucocutaneous disease only had a favorable prognosis and none developed systemic spread (follow-up, 4-93 months). Three of five patients with systemic disease died of lymphoma after 1-48 months. Morphologic and phenotypic features were similar regardless of extent of disease. One anaplastic lymphoma kinase-positive case was associated with systemic disease. Two cases had rearrangements of the DUSP22-IRF4 locus on chromosome 6p25.3, seen most frequently in primary cutaneous anaplastic large cell lymphoma. Our findings suggest mucosal CD30-positive T-cell lymphoproliferations share features with cutaneous CD30-positive T-cell lymphoproliferative disorders, and require clinical staging for stratification into primary and secondary types. Primary cases have clinicopathologic features closer to primary cutaneous disease than to systemic anaplastic large cell lymphoma, including indolent clinical behavior. Understanding the spectrum of mucosal CD30-positive T-cell lymphoproliferations is important to avoid possible overtreatment resulting from a diagnosis of overt T-cell lymphoma.
CD30 阳性 T 细胞淋巴增生性疾病分为皮肤型(原发性皮肤间变性大细胞淋巴瘤和淋巴母细胞性丘疹病)或系统性。由于疾病的范围决定了预后和治疗,因此皮肤受累的患者需要进行临床分期以确定是否存在系统性淋巴瘤。类似的过程可能涉及头颈部的黏膜部位,构成一个包括肿瘤和反应性疾病的谱(例如,伴有间质嗜酸性粒细胞增多的创伤性溃疡性肉芽肿)。然而,目前尚不存在用于黏膜 CD30 阳性 T 细胞淋巴增生的标准分类。为了更好地了解这些过程,我们鉴定了 15 例此类患者,并研究了临床表现、治疗和转归、形态学、免疫组织化学表型以及使用基因重排研究和荧光原位杂交的遗传学。15 例患者(11 例男性,4 例女性;平均年龄 57 岁)的疾病累及口腔/唇/舌(9 例)、眼眶/结膜(3 例)或鼻腔/鼻窦(3 例)。在有分期数据的 14 例患者中,7 例仅有黏膜疾病;2 例有黏膜皮肤疾病;5 例有系统性间变性大细胞淋巴瘤。仅有黏膜或黏膜皮肤疾病的患者预后良好,均未发生系统性播散(随访时间 4-93 个月)。5 例系统性疾病患者中有 3 例在 1-48 个月后死于淋巴瘤。无论疾病范围如何,形态学和表型特征均相似。1 例间变性淋巴瘤激酶阳性病例与系统性疾病有关。2 例患者存在染色体 6p25.3 上 DUSP22-IRF4 基因座的重排,这种重排最常见于原发性皮肤间变性大细胞淋巴瘤。我们的研究结果表明,黏膜 CD30 阳性 T 细胞淋巴增生与皮肤 CD30 阳性 T 细胞淋巴增生性疾病具有共同特征,需要进行临床分期以分层为原发性和继发性类型。原发性病例的临床病理特征与原发性皮肤疾病更接近,而与系统性间变性大细胞淋巴瘤不同,包括惰性的临床行为。了解黏膜 CD30 阳性 T 细胞淋巴增生的谱对于避免因诊断为显性 T 细胞淋巴瘤而可能导致的过度治疗非常重要。