Sayar Beyza S, Rüegg Simon, Schmidt Enno, Sibilia Maria, Siffert Myriam, Suter Maja M, Galichet Arnaud, Müller Eliane J
Molecular Dermatology, Institute of Animal Pathology, University of Bern, Bern, Switzerland; DermFocus, University of Bern, Bern, Switzerland.
Exp Dermatol. 2014 Jan;23(1):33-8. doi: 10.1111/exd.12290.
Novel insights into intra-cellular signalling involved in pemphigus vulgaris (PV), an autoimmune blistering disease of skin and mucous membranes, are now revealing new therapeutic approaches such as the chemical inhibition of PV-associated signals in conjunction with standard immunosuppressive therapy. However, extensive inhibition of signalling molecules that are required for normal tissue function and integrity may hamper this approach. Using a neonatal PV mouse model, we demonstrate that epidermal blistering can be prevented in a dose-dependent manner by clinically approved EGFR inhibitors erlotinib and lapatinib, but only up to approximately 50% of normal EGFR activity. At lower EGFR activity, blisters again aggravated and were highly exacerbated in mice with a conditional deletion of EGFR. Statistical analysis of the relation between EGFR activity and the extent of skin blistering revealed the best fit with a non-linear, V-shaped curve with a median break point at 52% EGFR activity (P = 0.0005). Moreover, lapatinib (a dual EGFR/ErbB2 inhibitor) but not erlotinib significantly reduced blistering in the oral cavity, suggesting that signalling mechanisms differ between PV predilection sites. Our results demonstrate that future clinical trials evaluating EGFR/ErbB2 inhibitors in PV patients must select treatment doses that retain a specific level of signal molecule activity. These findings may also be of relevance for cancer patients treated with EGFR inhibitors, for whom skin lesions due to extensive EGFR inhibition represent a major threat.
寻常型天疱疮(PV)是一种皮肤和黏膜的自身免疫性水疱病,目前对其细胞内信号传导的新见解揭示了新的治疗方法,例如在标准免疫抑制治疗的同时对PV相关信号进行化学抑制。然而,对正常组织功能和完整性所需的信号分子进行广泛抑制可能会阻碍这种方法。使用新生PV小鼠模型,我们证明临床批准的表皮生长因子受体(EGFR)抑制剂厄洛替尼和拉帕替尼可以以剂量依赖的方式预防表皮水疱形成,但EGFR活性只能维持在正常水平的约50%。在较低的EGFR活性水平下,水疱再次加重,并且在条件性缺失EGFR的小鼠中高度恶化。对EGFR活性与皮肤水疱程度之间关系的统计分析显示,最佳拟合为非线性V形曲线,中位断点在EGFR活性为52%处(P = 0.0005)。此外,拉帕替尼(一种双重EGFR/ErbB2抑制剂)而非厄洛替尼能显著减少口腔水疱,这表明PV好发部位的信号传导机制有所不同。我们的结果表明,未来评估PV患者使用EGFR/ErbB2抑制剂的临床试验必须选择能保留特定水平信号分子活性的治疗剂量。这些发现可能也与接受EGFR抑制剂治疗的癌症患者相关对于他们而言,由于广泛抑制EGFR导致的皮肤病变是一个重大威胁。