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Diagnostic Utility of TUNEL Staining for Degenerative Keratoacanthoma Requiring Pathologic Differentiation from Seborrheic Keratosis. TUNEL染色对需要与脂溢性角化病病理鉴别的退行性角棘瘤的诊断价值。
IF 0.9 Q3 Medicine Pub Date : 2023-03-16 eCollection Date: 2023-01-01 DOI: 10.1159/000529728
Mari Nakanishi, Makoto Kondo, Koji Habe, Akinobu Hayashi, Keiichi Yamanaka

Tumors developed in 2 old women presented with pathological findings similar to seborrheic keratosis, although the clinical feature of tumor showed typical keratoacanthoma. In addition to these two cases, we compared the pathological findings of a total of four cases, one case each of keratoacanthoma and seborrheic keratosis, which were clinically and histopathological typical. These two cases and the typical keratoacanthoma showed cell apoptosis by terminal deoxynucleotidyl transferase dUTP nick end labeling (TUNEL) staining and infiltration of cytotoxic T cells. The keratoacanthoma in the decompensated stage may be histologically similar to seborrheic keratosis. TUNEL staining can help in the diagnosis of fading keratoacanthoma.

2例老年妇女发生的肿瘤病理表现与脂溢性角化病相似,但临床表现为典型的角棘瘤。除这2例外,我们还比较了4例的病理表现,角棘瘤和脂溢性角化病各1例,均具有临床和组织病理学的典型。末端脱氧核苷酸转移酶dUTP缺口末端标记(TUNEL)染色及细胞毒性T细胞浸润显示细胞凋亡,与典型的角棘瘤相似。失代偿期的角棘瘤在组织学上可能与脂溢性角化病相似。TUNEL染色有助于消退性角棘瘤的诊断。
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引用次数: 0
Idiopathic Acroosteolysis: A Novel Cutaneous Sign Can Help Identify the Condition Early. 特发性骨溶解症:一种新的皮肤体征有助于早期识别这种疾病。
IF 0.9 Q3 Medicine Pub Date : 2023-03-15 eCollection Date: 2023-01-01 DOI: 10.1159/000529727
Samir Shrestha, Bashant Regmi, Raksha Pathak, George Kroumpouzos

Acroosteolysis (AO) is a rare condition characterized by resorption of the distal phalanges of the fingers and/or toes. It can be familial, idiopathic (IAO), occupational, or secondary. Other authors suggest a classification into primary (genetic disorders, lysosomal storage disorders) or secondary AO. Various skin and nail changes have been reported in this condition. However, the cutaneous change on the affected digit(s)/toe(s) during the natural course of AO has been poorly documented. A 5-year-old girl presented with a 3-month history of a distinct transverse boundary between normal skin proximally and affected crusted skin overlying osteolysis distally ("split" sign) on the plantar surface of the third toe. This boundary gradually elongated circumferentially to involve the dorsal surface. The mother gave a similar history of a delimitation line on the 2nd, 4th, and 5th toes of the right foot with durations of 3 months, 1 year, and 2 years, respectively, that disappeared before she noticed a shortening of those toes. X-rays revealed partial resorption of the terminal phalanx of the third toe and several lytic changes in the middle and terminal phalanx of the second, fourth, and fifth toes. The clinical features, radiology findings, and a workup that helped rule out conditions associated with AO (secondary AO) helped establish the diagnosis of IAO in our patient. This case study highlights that the natural course of IAO includes distinct skin findings, such as the "split" sign that we describe. This sign can help identify the condition early.

骨松症(AO)是一种罕见的疾病,其特征是手指和/或脚趾的远端指骨吸收。它可以是家族性的、特发性的(IAO)、职业性的或继发性的。其他作者建议将其分为原发性(遗传性疾病、溶酶体贮积症)或继发性 AO。据报道,这种疾病会引起各种皮肤和指甲变化。然而,关于受影响的手指/脚趾在AO自然病程中的皮肤变化却鲜有记载。一名 5 岁的女孩在 3 个月前出现了这样的病史:在第三脚趾的跖面,近端正常皮肤和远端覆盖骨溶解的受累结痂皮肤之间有一条明显的横向边界("分裂 "征)。该边界逐渐向周缘延伸,并涉及足背表面。母亲也有类似的病史,右脚的第2、4和5个脚趾上分别有一条持续3个月、1年和2年的分界线,在她发现这些脚趾变短之前,分界线就已经消失了。X 射线检查显示,第三趾的末节指骨部分吸收,第二、第四和第五趾的中节和末节指骨有多处溶解性病变。临床特征、放射学检查结果以及有助于排除与AO相关的疾病(继发性AO)的检查结果帮助确定了患者IAO的诊断。本病例研究强调,IAO的自然病程包括明显的皮肤症状,如我们描述的 "分裂 "征。这种体征有助于早期发现病情。
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引用次数: 0
Cutaneous Manifestations of Sarcoidosis Seen in a Patient with a History of Tuberous Sclerosis. 一名有结节性硬化症病史的患者身上出现的肉样瘤病皮肤表现。
IF 0.9 Q3 Medicine Pub Date : 2023-02-15 eCollection Date: 2023-01-01 DOI: 10.1159/000529159
Seth Bernacki, Paarth Dodia, Mikél Elizabeth Muse, Luke Maxfield

This case report details a patient with a history of tuberous sclerosis presenting with new-onset cutaneous lesions that turn out to be sarcoidosis. There may be a shared dysfunction of mTOR present in sarcoidosis and tuberous sclerosis. As a dermatologist, it is worth understanding the cutaneous manifestations of both diseases and maintaining a wide differential when new lesions arise in a patient with a history of either disorder.

本病例报告详细描述了一名有结节性硬化症病史的患者出现的新发皮肤病变,结果发现是肉样瘤病。肉样瘤病和结节性硬化症可能存在共同的 mTOR 功能障碍。作为一名皮肤科医生,值得了解这两种疾病的皮肤表现,并在有这两种疾病病史的患者出现新的皮损时保持广泛的鉴别。
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引用次数: 0
A Diagnostically Challenging Case of De Novo Febrile Ulceronecrotic Mucha-Habermann Disease with Fatal Pulmonary Involvement: A Case Report. 一例诊断困难的新发热性溃疡性穆查-哈伯曼病伴致命肺部受累病例:病例报告。
IF 0.9 Q3 Medicine Pub Date : 2023-01-18 eCollection Date: 2023-01-01 DOI: 10.1159/000528500
Waseem Alhawsawi, Khalid Al Hawsawi, Bashaer Almahdi, Shahad Alkidaiwi, Khlood Alzubaidy, Reema Alhuthayli, Abdulmohsin Algethami, Alhusain Alshareef

The febrile ulceronecrotic Mucha-Habermann disease is a rare and potentially lethal variant of pityriasis lichenoides et varioliformis acuta (PLEVA). It is characterized by a sudden onset of ulceronecrotic skin lesions associated with high fever and systemic symptoms. Herein, we report a 23-year-old male, not known to have any medical illnesses, presented with a month-long history of persistent fever of unknown origin associated with a sudden onset of progressive diffuse necrotic ulcers and widespread papulosquamous lesions. Pan CT showed enlarged lymph nodes in the cervix, chest, and abdomen. Unfortunately, a skin biopsy was done late, showing features consistent with PLEVA. Few days after admission, despite being on intravenous methylprednisolone, our patient rapidly deteriorated by showing severe acute respiratory symptoms and consequently died. In spite of the continuous addition of new case reports to the literature, no definite diagnostic criteria have been established, leading to late or missed cases, and an optimum treatment is still waiting.

发热性溃疡坏死型穆查-哈伯曼病(Mucha-Habermann disease)是一种罕见的、可能致命的脓疱型苔癣和变异型斑秃(PLEVA)变种。该病的特点是突然出现溃疡坏死性皮损,并伴有高烧和全身症状。在此,我们报告了一名 23 岁男性患者的病例,该患者无任何内科疾病,有一个月不明原因的持续发热史,伴有突然出现的进行性弥漫性坏死溃疡和广泛的丘疹鳞屑性病变。平扫 CT 显示宫颈、胸部和腹部淋巴结肿大。不幸的是,皮肤活检做得很晚,显示的特征与白塞病一致。入院几天后,尽管患者静脉注射了甲基强的松龙,但病情迅速恶化,出现了严重的急性呼吸道症状,最终死亡。尽管不断有新的病例报告出现在文献中,但由于尚未建立明确的诊断标准,导致病例迟发或漏诊,最佳治疗方法仍在等待中。
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引用次数: 0
A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate. 用英夫利西单抗和甲氨蝶呤成功治疗肾细胞癌相关多中心网状组织胞增多症的新病例
IF 0.9 Q3 Medicine Pub Date : 2023-01-09 eCollection Date: 2023-01-01 DOI: 10.1159/000528254
Suraj Patel, Mandy Alhajj, Conrad Brimhall

Multicentric reticulohistiocytosis (MRH) is categorized as a rare non-Langerhans cell histiocytosis most commonly seen in women in the fourth to fifth decade of life. This systemic inflammatory condition affects multiple organ systems and can result in severe joint destruction which can progress to arthritis mutilans. To date, various underlying malignancies have been discovered in patients with MRH including breast, gastric, thymic, hepatic, and melanoma. There has been 1 case of underlying renal cell carcinoma reported in a patient diagnosed with MRH. Additionally, there is no consistently recognized treatment for MRH described in the literature. The rarity of the disease contributes to the difficulty in defining a standardized treatment. We present the case of a patient with extensive joint and skin involvement who was successfully treated with infliximab and methotrexate, experienced clinical improvement, and was later diagnosed with clear cell renal cell carcinoma. The synergistic effects of infliximab and methotrexate, in combination with the low side-effect profile, appear to be promising in the setting of MRH and in our patient resulted in the resolution of symptoms and cutaneous manifestations. We suggest this regimen as an effective combination therapy. We emphasize thorough and continuous screening for underlying malignancy associated with MRH, despite clinical improvement or negative malignancy work-up upon initial diagnosis.

多中心网状组织细胞增生症(MRH)是一种罕见的非朗格汉斯细胞组织细胞增生症,最常见于四五十岁的女性。这种全身性炎症会影响多个器官系统,并可导致严重的关节破坏,进而发展为关节炎。迄今为止,已在 MRH 患者中发现多种潜在恶性肿瘤,包括乳腺癌、胃癌、胸腺癌、肝癌和黑色素瘤。据报道,确诊为 MRH 的患者中有 1 例患有潜在的肾细胞癌。此外,文献中也没有一致公认的 MRH 治疗方法。这种疾病的罕见性导致难以确定标准化的治疗方法。我们介绍了一例关节和皮肤广泛受累的患者,该患者成功接受了英夫利昔单抗和甲氨蝶呤的治疗,临床症状有所改善,后来被诊断为透明细胞肾细胞癌。英夫利昔单抗和甲氨蝶呤的协同作用与低副作用相结合,在 MRH 的治疗中似乎很有前景,我们的患者的症状和皮肤表现也得到了缓解。我们建议将该方案作为一种有效的联合疗法。我们强调,尽管初诊时临床症状有所改善或恶性肿瘤检查结果呈阴性,但仍要对与 MRH 相关的潜在恶性肿瘤进行彻底、持续的筛查。
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引用次数: 1
A Rare Case of Prurigo Pigmentosa in a Danish Sibling Couple. 丹麦一对兄弟姐妹患罕见色斑疹1例。
IF 0.9 Q3 Medicine Pub Date : 2023-01-01 DOI: 10.1159/000528422
Maria Danielsen, Kristine Pallesen, Rikke Riber-Hansen, Anne Bregnhøj

Prurigo pigmentosa (PP) is probably underdiagnosed due to lack of awareness. Previously, it was assumed that PP primarily affected Japanese females; however, more cases are reported worldwide, and the pathogenesis is still not completely understood. In this case report, we present two healthy Danish siblings, who developed PP approximately 2 weeks after starting a ketogenic diet, suggesting that both increased levels of ketone bodies in the blood together with a genetic predisposition might play a role in the development of PP.

由于缺乏认识,色素性痒疹(PP)可能被误诊。以前,人们认为PP主要影响日本女性;然而,世界范围内报告的病例更多,发病机制仍未完全了解。在本病例报告中,我们介绍了两个健康的丹麦兄弟姐妹,他们在开始生酮饮食大约2周后患上了PP,这表明血液中酮体水平的增加以及遗传易感性可能在PP的发展中起作用。
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引用次数: 0
Localized Bullous Pemphigoid in a Patient with Acquired Reactive Perforating Collagenosis. 获得性反应性穿孔性胶原沉积患者的局限性大疱性类天疱疮。
IF 0.9 Q3 Medicine Pub Date : 2023-01-01 DOI: 10.1159/000528140
Masahiro Oka

A 61-year-old man presented with 6-month and 5-day histories of multiple, pruritic nodular eruptions on the trunk and extremities and bullous eruptions on the left foot, respectively. The nodular eruptions had been treated with topical corticosteroids without improvement. He had been diagnosed with diabetes mellitus at the age of 42 years and had been suffering from end-stage renal disease for 1 year. Physical examination revealed scattered violet-brown papules and nodules on the trunk and extremities, many of which had central umbilicated necrosis or keratin plugs. Additionally, two tense bullae and five erosions were noted on the dorsal aspect of the left foot. Laboratory tests showed elevated levels of serum anti-bullous pemphigoid (BP)180 antibody. Histopathological findings of a nodule and a bulla were compatible with those of acquired reactive perforating collagenosis (ARPC) and BP, respectively. The papular and nodular lesions were diagnosed as ARPC, while bullous and erosive lesions were diagnosed as localized BP. The present case, together with previously reported cases of coexisting generalized BP and ARPC, suggests that coexistence of BP, regardless of whether generalized or localized, is significantly associated with ARPC.

61岁男性,6个月和5天,躯干和四肢多发瘙痒性结节性皮疹,左脚大疱性皮疹。结节性爆发曾用局部皮质类固醇治疗,但未见改善。他在42岁时被诊断出患有糖尿病,并患有终末期肾脏疾病1年。体格检查发现躯干和四肢散在紫棕色丘疹和结节,其中许多有中央脐状坏死或角蛋白塞。此外,在左脚背侧发现2个紧绷大泡和5个糜烂。实验室检查显示血清抗大疱性类天疱疮(BP)180抗体水平升高。结节和大疱的组织病理学结果分别与获得性反应性穿孔性胶原症(ARPC)和BP的组织病理学结果一致。丘疹和结节性病变诊断为ARPC,大疱性和糜烂性病变诊断为局限性BP。本病例,连同先前报道的广泛性BP和ARPC共存的病例,表明BP的共存,无论是广泛性BP还是局部性BP,都与ARPC显著相关。
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引用次数: 1
Possible Efficacy of Vedolizumab, an Anti-α4β7 Integrin Antibody, in Palmoplantar Pustulosis. 抗-α4β7整合素抗体Vedolizumab治疗掌跖脓疱病的可能疗效
IF 0.9 Q3 Medicine Pub Date : 2023-01-01 DOI: 10.1159/000529080
Hitoshi Terui, Rintaro Moroi, Atsushi Masamune, Setsuya Aiba, Kenshi Yamasaki

Palmoplantar pustulosis (PPP) is a chronic skin inflammatory disease in which blisters and pustules repeatedly develop on palms and soles. PPP is often refractory to topical therapy, oral therapy, phototherapy, and biologics that are usually applied for PPP. We report a patient with PPP improved by vedolizumab (anti-α4β7 integrin antibody) treatment for ulcerative colitis, suggesting the possibility of a new molecular target for PPP therapy.

掌跖脓疱病(PPP)是一种慢性皮肤炎症性疾病,在手掌和脚底反复出现水泡和脓疱。局部治疗、口服治疗、光疗和生物制剂通常对PPP无效。我们报道了一例通过vedolizumab(抗α4β7整合素抗体)治疗溃疡性结肠炎改善PPP的患者,提示PPP治疗的新分子靶点的可能性。
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引用次数: 2
Generalized Pustular Psoriasis Flare-Up after Both Doses of BBIBP-CorV Vaccination in a Patient under Adalimumab Treatment: A Case Report. 在阿达木单抗治疗的患者中,两剂BBIBP-CorV疫苗接种后全身性脓疱性银屑病发作:一例报告
IF 0.9 Q3 Medicine Pub Date : 2023-01-01 DOI: 10.1159/000530074
Dorsa Dayani, Hatam Rokhafrouz, Kamran Balighi

Generalized pustular psoriasis is a possibly serious condition that can be triggered by various factors. Previous studies show a slight likelihood of disease exacerbation subsequent to COVID-19 vaccination. Here, we present the first (to the best of our knowledge) case of pustular psoriasis flare after each one of the two shots of the BBIBP-CorV (Sinopharm) vaccine despite adalimumab treatment.

广泛性脓疱性牛皮癣是一种可能由多种因素引发的严重疾病。先前的研究表明,接种COVID-19疫苗后疾病恶化的可能性很小。在这里,我们报告了第一例(据我们所知)在阿达木单抗治疗的情况下,两次注射BBIBP-CorV(国药控股)疫苗后每次都出现脓疱性牛皮癣爆发的病例。
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引用次数: 1
Use of Talimogene Laherparepvec to Treat Cutaneous Squamous Cell Carcinoma in a Renal Transplant Patient. 利莫gene Laherparepvec治疗肾移植患者皮肤鳞状细胞癌。
IF 0.9 Q3 Medicine Pub Date : 2023-01-01 DOI: 10.1159/000530851
Max Miller, Nancy H Kim, Maya K Thosani, Justin C Moser

A 66-year-old female with a history of two renal transplants due to recurrent thrombotic thrombocytopenic purpura presented to clinic with multiple lesions identified to be non-metastatic cutaneous squamous cell carcinoma (CSCC). The patient previously underwent multiple Mohs procedures and radiation therapy treatment but continued to develop CSCC lesions with increasing frequency. After discussing multiple treatment options, it was elected to pursue treatment with Talimogene laherparepvec (T-VEC) given the systemic immune responses it can cause, with low theoretical risk of graft rejection. After starting intratumoral T-VEC injections, treated lesions began to decrease in size, and a reduction in the rate of new CSCC lesions was observed. Treatment was held due to unrelated renal complications during which time new CSCCs developed. Patient was restarted on T-VEC therapy with no recurrent renal issues. Upon reinitiating treatment, injected and non-injected lesions showed reduction in size, and the development of new lesions again ceased. One injected lesion was resected via Mohs micrographic surgery due to its size and discomfort. On sectioning, this demonstrated an exuberant lymphocytic perivascular infiltrate which was consistent with treatment response to T-VEC, with little active tumor. With high rates of non-melanoma skin cancer in renal transplant patients, their transplant status significantly limits treatment options, specifically with regards to anti-PD-1 therapy. This case suggests T-VEC can generate local and systemic immune responses in the setting of immunosuppression and that T-VEC may be a beneficial therapeutic option for transplant patients with CSCC.

一位66岁女性,因复发性血栓性血小板减少性紫癜而进行过两次肾移植,并伴有多处非转移性皮肤鳞状细胞癌(CSCC)。该患者先前接受了多次Mohs手术和放射治疗,但持续发生CSCC病变的频率越来越高。在讨论了多种治疗方案后,考虑到T-VEC可引起全身免疫反应,移植排斥的理论风险较低,最终选择了T-VEC治疗。在开始瘤内注射T-VEC后,治疗后的病变开始缩小,并且观察到新的CSCC病变发生率降低。由于不相关的肾脏并发症,在此期间出现了新的CSCCs。患者重新开始T-VEC治疗,无复发性肾脏问题。在重新开始治疗后,注射和非注射病变的大小都缩小了,新病变的发展再次停止。一个注射性病灶由于其大小和不适,通过Mohs显微摄影手术切除。在切片上,显示了旺盛的淋巴细胞浸润血管周围,这与T-VEC治疗反应一致,几乎没有活动性肿瘤。肾移植患者的非黑色素瘤皮肤癌发病率很高,他们的移植状况显著限制了治疗选择,特别是抗pd -1治疗。该病例提示,在免疫抑制的情况下,T-VEC可以产生局部和全身免疫反应,并且T-VEC可能是移植CSCC患者的有益治疗选择。
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引用次数: 0
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Case Reports in Dermatology
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