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Generalized pustular psoriasis patient with a heterozygous hypomorphic MPO variant refractory to intravenous spesolimab. 泛发性脓疱型银屑病患者伴有对静脉注射斯贝单抗无效的杂合子低表型 MPO 变异。
Pub Date : 2024-09-20 DOI: 10.1111/1346-8138.17464
Youichi Ogawa, Eri Maejima, Takuya Takeichi, Takashi Okamoto, Hiroshi Mitsui, Shinji Shimada, Masashi Akiyama, Tatsuyoshi Kawamura

Generalized pustular psoriasis (GPP) is a recurrent and sometimes life-threatening sterile pustular disease. Because interleukin (IL)-36 is the central cytokine in disease formation, spesolimab, which interferes with IL-36 receptor signaling, is highly effective. Here, we report a patient with GPP with a heterozygous hypomorphic MPO variant refractory to intravenous spesolimab. Although spesolimab showed excellent clinical efficacy in resolving pre-existing pustules and erythema, it did not suppress the emergence of new pustules and erythema, which did not decrease the peripheral blood neutrophil count, therefore bimekizumab, an anti-IL-17A/IL-17F antibody, was administered after the second spesolimab infusion, which resolved the pustules and erythema. We discuss the possible reasons for the resistance mechanism to spesolimab.

泛发性脓疱型银屑病(GPP)是一种反复发作、有时可危及生命的无菌性脓疱病。由于白细胞介素(IL)-36 是疾病形成的核心细胞因子,干扰 IL-36 受体信号转导的斯派索利单抗非常有效。在此,我们报告了一名对静脉注射司来索利单抗无效的GPP患者,该患者患有杂合子低形态MPO变异。虽然斯派索利单抗在消退原有脓疱和红斑方面显示出了极佳的临床疗效,但它并没有抑制新脓疱和红斑的出现,而这些新脓疱和红斑并没有降低外周血中性粒细胞计数,因此,在第二次输注斯派索利单抗后,又输注了一种抗IL-17A/IL-17F抗体--bimekizumab,从而消退了脓疱和红斑。我们讨论了对斯来索利单抗产生耐药机制的可能原因。
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引用次数: 0
Recurrent eosinophilic cellulitis (Wells syndrome) after vaccination in a child. 一名儿童接种疫苗后复发嗜酸性细胞性蜂窝织炎(韦尔斯综合征)。
Pub Date : 2024-09-20 DOI: 10.1111/1346-8138.17417
Ilaria Praturlon, Lisa Sant, Lorenzo D'Alì, Stefanny Andrade, Enzo Errichetti, Paola Cogo, Giorgia Martini
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引用次数: 0
A case of Stevens-Johnson syndrome after influenza a virus infection. 一例感染甲型流感病毒后出现史蒂文斯-约翰逊综合征的病例。
Pub Date : 2024-09-20 DOI: 10.1111/1346-8138.17467
Chiaki Sakashita, Kento Mizutani, Mai Nishimura, Hiroshi Kitagawa, Koji Habe, Keiichi Yamanaka
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引用次数: 0
What is "eczema"? 什么是 "湿疹"?
Pub Date : 2024-09-20 DOI: 10.1111/1346-8138.17439
Yoshiki Tokura, Marina Yunoki, Shumpei Kondo, Masaki Otsuka

Eczema is the most common category of inflammatory skin disorders as dermatologists see many patients with eczematous diseases in daily practice. It is characterized by the three major morphological features: multiple-pinpoint condition, polymorphism, and itch. To describe polymorphism, "eczema triangle" has been used in German/Japanese dermatology. The multiple pinpoints correspond to numerous tiny foci from which individual papules/vesicles arise. The polymorphism betrays composition of erythema, papule, seropapule, vesicle, pustule, scale, and crust, which are seen in acute eczema. Meanwhile, chronic eczema is represented by lichenification and hyperpigmentation, and possibly by hypopigmentation. In acute eczema, spongiosis is associated with overproduction of hyaluronic acid, secretion of self-protective galectin-7, and decreased expression of E-cadherin. In the upper dermis, Th1/Tc1 or Th2/Tc2, and additional Th17, Th22, and/or Tc22 infiltrate, depending on each eczematous disease. Innate lymphoid cells are also involved in the formation of eczema. In chronic eczema, periostin contributes to remodeling of inflammatory skin with dermal fibrosis, and epidermal melanogenesis and dermal pigment deposition result in hyperpigmentation. Finally, eczematous diseases are potentially associated with increased risk of comorbidities, including not only other allergic diseases but also coronary heart disease and mental problems such as depression. Although the original word for eczema is derived from old Greek "ekzein," eczema remains a major target of modern science and novel therapies.

湿疹是炎症性皮肤病中最常见的一类,因为皮肤科医生在日常工作中会遇到很多湿疹患者。湿疹有三大形态特征:多针尖状态、多形性和瘙痒。为了描述多形性,德国/日本皮肤病学界使用了 "湿疹三角 "一词。多针点相当于许多微小的病灶,单个丘疹/疱疹就是从这些病灶中产生的。多形性反映了急性湿疹中红斑、丘疹、血清丘疹、水疱、脓疱、鳞屑和结痂的构成。而慢性湿疹则表现为苔藓化和色素沉着,也可能表现为色素减退。在急性湿疹中,海绵状增生与透明质酸过度分泌、自我保护的 galectin-7 分泌和 E-cadherin 表达减少有关。在真皮上层,Th1/Tc1 或 Th2/Tc2 以及额外的 Th17、Th22 和/或 Tc22 细胞浸润,这取决于每种湿疹疾病。先天性淋巴细胞也参与了湿疹的形成。在慢性湿疹中,表皮生长因子(periostin)有助于炎症皮肤的重塑和真皮纤维化,表皮黑色素生成和真皮色素沉积导致色素沉着。最后,湿疹疾病可能会增加合并症的风险,其中不仅包括其他过敏性疾病,还包括冠心病和抑郁症等精神问题。虽然湿疹的原词源自古希腊语 "ekzein",但湿疹仍然是现代科学和新型疗法的主要目标。
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引用次数: 0
Upadacitinib shows efficacy in Netherton syndrome with poor response to Dupilumab. 乌达帕替尼对尼瑟顿综合征中对杜匹单抗反应不佳的患者有疗效。
Pub Date : 2024-09-20 DOI: 10.1111/1346-8138.17461
Hui Li, Jianbo Wang
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引用次数: 0
Grade 3 acute kidney injury caused anti‐glomerular basement membrane nephritis during immune checkpoint inhibitor treatment for melanoma 黑色素瘤免疫检查点抑制剂治疗期间抗肾小球基底膜肾炎引起的三级急性肾损伤
Pub Date : 2024-09-13 DOI: 10.1111/1346-8138.17460
Akemi Fukuda, Dai Ogata, Yoichi Oshima, Eiji Nakano, Kenjiro Namikawa, Motoaki Komatsu, Akira Nakamura, Kohei Sugiura, Naoya Yamazaki
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引用次数: 0
Case of tinea corporis caused by a terbinafine‐sensitive Trichophyton indotineae strain in a Vietnamese worker in Japan 在日本工作的越南工人对特比萘芬敏感的吲哚癣菌株引起的体癣病例
Pub Date : 2024-09-13 DOI: 10.1111/1346-8138.17463
Takashi Mochizuki, Kazushi Anzawa, Andrea Marie Bernales‐Mendoza, Akira Shimizu
A 42‐year‐old Vietnamese egg factory worker in Ishikawa prefecture, Japan, presented with itchy concentric erythema on the trunk and left calf. The lesions tested positive by direct potassium hydroxide examination, and two fungal strains were isolated. The isolates produced conidia abundantly and were morphologically indistinguishable from Trichophyton mentagrophytes/interdigitale, but were identified as Trichophyton indotineae by internal transcribed spacer sequence of ribosomal DNA. The lesions were refractory to treatment with topical luliconazole (LLCZ) cream for 4 weeks but subsided with oral itraconazole (ITCZ) 100 mg/day for 4 weeks in combination with topical lanoconazole (LCZ) cream. The lesions recurred 6 weeks after discontinuation of oral ITCZ, and an additional isolate was cultured. The minimum inhibitory concentrations of antimycotics for the isolate cultured at the first visit were: terbinafine (TBF) 0.03 μg/mL, ITCZ 0.015 μg/mL, LLCZ 0.0005 μg/mL, and LCZ 0.002 μg/mL. No TBF‐resistant mutation in the amino acid sequence of squalene epoxidase, i.e., Leu 393 Ser/Phe or Phe 397 Leu, was detected in the isolate. The reason for recalcitrance in this case, despite the isolate's sensitivity to antimycotics, was unclear. Possible factors include insufficient use of the antimycotics, incomplete removal of abundantly produced conidia from the lesions, the patient's environment, and a language gap between the patient and physician hindering communication.
日本石川县一名 42 岁的越南蛋厂工人因躯干和左小腿出现同心圆红斑而感到瘙痒。经氢氧化钾直接检测,病变呈阳性,并分离出两种真菌菌株。分离出的菌株产生大量分生孢子,从形态上看与门冬癣菌(Trichophyton mentagrophytes/interdigitale)没有区别,但通过核糖体 DNA 内部转录间隔序列被鉴定为 indotineae 毛癣菌。外用卢立康唑(LLCZ)乳膏治疗 4 周后,皮损仍难治,但口服伊曲康唑(ITCZ)100 毫克/天,联合外用兰诺康唑(LCZ)乳膏治疗 4 周后,皮损有所缓解。停用口服伊曲康唑 6 周后,皮损复发,又培养出了一个分离株。首次就诊时培养出的分离株的抗霉菌药物最低抑菌浓度为:特比萘芬(TBF)0.03 μg/mL,ITCZ 0.015 μg/mL,LLCZ 0.0005 μg/mL,LCZ 0.002 μg/mL。在该分离株中,角鲨烯环氧化物酶的氨基酸序列(即 Leu 393 Ser/Phe 或 Phe 397 Leu)中没有检测到抗 TBF 的突变。尽管该分离物对抗霉剂很敏感,但其抗药性的原因尚不清楚。可能的因素包括抗霉菌药物使用不足、未完全清除病灶中大量产生的分生孢子、患者所处的环境以及患者与医生之间的语言障碍阻碍了沟通。
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引用次数: 0
Nevus comedonicus with lesional growth of terminal hair: An unusual case 伴有末端毛发病变性生长的网状痣:一个不寻常的病例
Pub Date : 2024-09-13 DOI: 10.1111/1346-8138.17457
Hazem A. Juratli, Matiar Madanchi, Riccardo Curatolo, Dario Didona, Rudolph Happle
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引用次数: 0
A case of IgA vasculitis with Koebner phenomenon of the nose 一例 IgA 血管炎合并鼻腔柯布纳现象的病例
Pub Date : 2024-09-13 DOI: 10.1111/1346-8138.17458
Taiki Nakai, Chikane Maeda, Issei Kido, Kimiko Nakajima, Yuki Osakabe, Tatsuki Matsumoto, Yuki Ogasawara, Yusuke Oki, Kozo Nakai
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引用次数: 0
Psoriasis treatment and biologic switching: The association with clinical characteristics and laboratory biomarkers over a 13‐year retrospective study 银屑病治疗与生物制剂转换:13 年回顾性研究与临床特征和实验室生物标志物的关系
Pub Date : 2024-09-13 DOI: 10.1111/1346-8138.17465
Asumi Koyama, Lixin Li, Toyoki Yamamoto, Haruka Taira, Eiki Sugimoto, Yukiko Ito, Yuka Mizuno, Kentaro Awaji, Shoko Tateishi, Hiroko Kanda, Shinichi Sato, Sayaka Shibata
The advent of biologics has greatly improved patient outcomes, yet some patients are compelled to switch therapies. Predicting these therapeutic failures is important; however, the factors associated with switching biologics have not been fully explored. This study examined patterns and determinants of biologics switching in psoriasis treatment retrospectively over 13 years. We focused on the association between clinical characteristics, basal laboratory data, and frequency of biologics switching. The findings revealed that elevated Psoriasis Area Severity Index scores and the presence of arthritis were observed in patients who experienced two or more treatment switches compared with those without treatment switches. Moreover, neutrophil to lymphocyte ratio was associated with higher biologics switching rates, indicating that systemic inflammation significantly impacts treatment adherence. A treatment approach, taking into account both the clinical presentation and inflammatory biomarkers, may be important for optimizing patient management in psoriasis.
生物制剂的出现大大改善了患者的治疗效果,但有些患者不得不更换疗法。预测这些治疗失败非常重要;然而,与转换生物制剂相关的因素尚未得到充分探讨。本研究回顾性研究了 13 年来银屑病治疗中生物制剂转换的模式和决定因素。我们重点研究了临床特征、基础实验室数据和生物制剂转换频率之间的关联。研究结果表明,与未更换治疗方案的患者相比,更换过两次或两次以上治疗方案的患者的银屑病面积严重性指数评分升高,并伴有关节炎。此外,中性粒细胞与淋巴细胞比率与较高的生物制剂转换率相关,表明全身炎症对治疗依从性有重大影响。同时考虑临床表现和炎症生物标志物的治疗方法可能对优化银屑病患者的管理非常重要。
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The Journal of dermatology
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