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Intralesional Kenalog Injections for Recurrent Cutaneous Rosai-Dorfman Disease (RDD) in the Lower Extremity 治疗下肢复发性皮肤罗赛-多夫曼病(RDD)的 KENALOG 局内注射疗法
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.15
Victoria Jiminez, Lawangeen Zeb, Lauren Graham, Anna Gray
Rosai-Dorfman disease (RDD) is a rare disorder of histiocytic origin with nodal, extranodal, and cutaneous classifications often presenting as eruptive rashes, most commonly affecting the face, trunk, and thigh. Due to its rarity, no consensus exists regarding standard of care for these lesions, as most knowledge regarding treatment is provided by case reports. To our knowledge, we report the first case of a patient with a recurrent lower extremity mass consistent with cutaneous RDD, despite surgical excision and topical steroids treatment. A 51-year-old female presented with recurrent RDD after failed treatment with clobetasol ointment and surgical excision at an outside practice. Examination revealed violaceous plaques with overlying scale, without lymphadenopathy. With the failure of the previous excision, the patient was prescribed 5 mg/ml intralesional Kenalog (ILK) injections to suppress inflammation of the lesion. She experienced improved clinical response to treatment and remission of active disease with residual post-inflammatory hyperpigmentation at 10 months after two rounds of injections, although the patient reported vast improvement at 5 months as well. Treatment of RDD is difficult due to its rarity, hindering clinical trial development. Therapeutics reported to be efficacious include topical, intralesional, and systemic steroids, retinoids, methotrexate, chemotherapy, cryotherapy, phototherapy, and radiation. Clinical management is variable and often requires multiple forms of treatment on a case-to-case basis, highlighting the need for larger clinical studies to establish a standard of care. Our case poses interest in the use of ILK for RDD and should be considered in recurrent cases after prior surgical excision.
罗赛-多夫曼病(RDD)是一种罕见的组织细胞源性疾病,有结节型、结节外型和皮肤型三种类型,通常表现为爆发性皮疹,最常累及面部、躯干和大腿。由于其罕见性,目前还没有就这些病变的治疗标准达成共识,因为有关治疗的大多数知识都是由病例报告提供的。据我们所知,我们报告了第一例复发性下肢肿块患者,尽管患者接受了手术切除和外用类固醇治疗,但其症状与皮肤 RDD 一致。一名 51 岁的女性患者在一家外院接受氯倍他索软膏和手术切除治疗失败后,复发了 RDD。检查发现该患者身上出现粟粒大小的斑块,上覆鳞屑,无淋巴结肿大。由于之前的切除手术失败,患者被处方注射 5 毫克/毫升的凯纳洛(ILK)以抑制皮损的炎症。经过两轮注射后,她的临床治疗反应有所改善,10 个月时活动性疾病得到缓解,但仍有炎症后色素沉着残留,不过患者称 5 个月时情况也大有好转。RDD 的治疗因其罕见性而困难重重,阻碍了临床试验的发展。据报道,有效的治疗方法包括局部、鞘内和全身类固醇、维甲酸、甲氨蝶呤、化疗、冷冻疗法、光疗和放射治疗。临床治疗方法多种多样,通常需要根据具体病例采取多种治疗方法,因此需要进行更大规模的临床研究,以确定治疗标准。我们的病例引起了人们对使用 ILK 治疗 RDD 的兴趣,在手术切除后复发的病例中应考虑使用 ILK。
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引用次数: 0
Merkel cell carcinoma primary sites and overall survival: The prognostic indications of head and neck tumors 梅克尔细胞癌原发部位和总生存率:头颈部肿瘤的预后指标
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.6
Marcus L Elias, Joshua Burshtein, Victoria R Sharon
Merkel cell carcinoma (MCC) is a rare cutaneous neuroendocrine tumor. Primarily affecting the elderly, the incidence of MCC is 0.10 to 1.6 cases per 100,000 people per year. 1 MCC has a low overall survival (OS), ranging between 37 and 74% at 5 years. 1–5 The majority of lesions occur on the head and neck followed by the trunk and extremities
梅克尔细胞癌(MCC)是一种罕见的皮肤神经内分泌肿瘤。MCC 主要影响老年人,发病率为每年每 10 万人中有 0.10 到 1.6 例。1 MCC 的总生存率(OS)较低,5 年生存率在 37% 到 74% 之间。1-5 大多数病变发生在头颈部,其次是躯干和四肢。
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引用次数: 0
Epithelioid Angiosarcoma: A Diagnostic Dilemma with Clinically and Histopathologically Overlapping Features 上皮样血管肉瘤:临床和组织病理学特征重叠的诊断难题
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.14
Jessica Forbes Kaprive, Romsin Zaya, Alexandra Loperfito, Craig J Garofola
Cutaneous angiosarcoma is a rare malignancy of endothelial cell origin that commonly presents in the head and neck region of elderly individuals and is known for high rates of lymph node metastasis and mortality. There is significant variability in the histopathologic features, and immunohistochemistry is necessary to help differentiate epithelioid angiosarcoma from lesions it may clinically mimic. Here, we report a rare case of poorly differentiated epithelioid angiosarcoma on the trunk initially diagnosed as poorly differentiated squamous cell carcinoma (SCC) on shave biopsy. A 56-year-old man presented for a full-body skin examination significant for a 5.6 x 2.8 mm brown-black macule on the left superomedial posterior trunk and a 5.0 x 2.7 mm pink, ulcerated papule on the right inferior posterior trunk. On histopathology, one lesion was found to be melanoma in situ, and the other showed ulcerated and infiltrative, poorly differentiated SCC. Post-excision histologic analysis of the right inferior back lesion revealed tumor cells immunoreactive to vimentin, ERG, and CD31 (PECAM-1). These findings suggested that the lesion previously diagnosed as SCC was an invasive, poorly differentiated epithelioid angiosarcoma. The patient underwent 40 sessions of radiotherapy. Epithelioid angiosarcoma occurring outside of the conventional epidemiologic profile is believed to portend higher mortality rates. This case highlights the importance of thorough clinical evaluation and appropriate histopathologic diagnosis of an aggressive neoplasm unlikely to present in this location. Further studies are required to explore potential risk factors, effective treatments, and long-term outcomes in patients with epithelioid angiosarcoma.
皮肤血管肉瘤是一种罕见的内皮细胞来源恶性肿瘤,常见于老年人的头颈部,淋巴结转移率和死亡率都很高。组织病理学特征存在很大的变异性,免疫组化有助于将上皮样血管肉瘤与其临床上可能模仿的病变区分开来。在此,我们报告了一例罕见的躯干上皮样血管肉瘤病例,该病例最初在剃须活检中被诊断为分化不良的鳞状细胞癌(SCC)。一名 56 岁的男子接受了全身皮肤检查,发现左侧躯干后上方有一个 5.6 x 2.8 毫米的棕黑色斑丘疹,右侧躯干后下方有一个 5.0 x 2.7 毫米的粉红色溃疡性丘疹。组织病理学检查发现,一个病变为原位黑色素瘤,另一个病变为溃疡和浸润性、分化不良的 SCC。切除后对右下背部病灶的组织学分析显示,肿瘤细胞对波形蛋白、ERG和CD31(PECAM-1)有免疫反应。这些结果表明,之前被诊断为 SCC 的病变是一种浸润性、分化不良的上皮样血管肉瘤。患者接受了 40 次放疗。上皮样血管肉瘤发生在传统流行病学特征之外,被认为预示着较高的死亡率。该病例强调了对这种不太可能出现在该部位的侵袭性肿瘤进行全面临床评估和适当组织病理学诊断的重要性。我们需要进一步研究上皮样血管肉瘤患者的潜在风险因素、有效治疗方法和长期预后。
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引用次数: 0
Type I Collagen Matrix with Polyhexylmethylene Biguanide in a Chronic Lower Extremity Wound I 型胶原蛋白基质与聚己亚甲基双胍在慢性下肢伤口中的应用
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.16
K. Aoki, Simona Bartos
Chronic wounds pose a significant healthcare challenge, affecting millions of individuals and incurring substantial healthcare costs. In this case study, we present the successful treatment of a chronic lower extremity wound in an 80-year-old female using PuraPly™, a Type I collagen matrix embedded with polyhexylmethylene biguanide (PHMB). The patient initially sustained the injury, which had become infected and resistant to conventional treatments. After applying PuraPly™, the wound showed rapid improvement, ultimately resulting in complete closure. Our findings highlight the utility of PuraPly™ as an effective intervention for chronic wounds, thereby improving treatment outcomes and the quality of life for these patients.
慢性伤口是一项重大的医疗挑战,影响着数百万人,并产生大量医疗费用。在本案例研究中,我们介绍了使用 PuraPly™ 成功治疗一名 80 岁女性下肢慢性伤口的案例,PuraPly™ 是一种嵌入聚己基亚甲基双胍 (PHMB) 的 I 型胶原蛋白基质。患者最初受伤时已经感染,对传统治疗方法产生了抗药性。使用 PuraPly™ 后,伤口迅速得到改善,最终完全闭合。我们的研究结果凸显了 PuraPly™ 作为慢性伤口有效干预措施的实用性,从而改善了治疗效果和这些患者的生活质量。
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引用次数: 0
Safety and Efficacy of Combination Therapy of Upadacitinib and Biologic Agents for Treatment-Resistant Psoriasis and Psoriatic Arthritis 乌达帕替尼和生物制剂联合疗法治疗耐药银屑病和银屑病关节炎的安全性和有效性
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.17
Shivkar Amara, Aarav Patel, M. Lebwohl
Importance: Although there are many available treatments for psoriasis and psoriatic arthritis (PsA), there are patients resistant to standard conventional therapy. For these patients, combination therapy of biologic agents and upadacitinib may be a viable alternative option; however, there is limited data on concomitant usage. Objective: To evaluate the safety and efficacy of combination therapy of upadacitinib and biologic agents for the treatment of refractory psoriasis and psoriatic arthritis. Design: We report the results of a retrospective chart review of 3 patients in a single practice treated with a combination of upadacitinib and biologics for psoriasis and PsA. The risks, benefits, and safety warnings of combination therapy of upadacitinib and biologic agents were discussed before initiation. After shared decision-making with the provider, patients were placed on a trial of combination therapy. Results: All three patients, previously refractory to monotherapy, experienced significant improvement of their symptoms while on combination therapy. No adverse reactions (malignancy, cardiovascular events, venous thromboembolism) occurred during treatment. However, one patient experienced a shingles outbreak and diverticulitis while on combination treatment. Further studies are needed to determine long-term efficacy with a larger sample size.  
重要性:尽管银屑病和银屑病关节炎(PsA)的现有治疗方法很多,但仍有一些患者对标准常规疗法产生耐药性。对于这些患者来说,生物制剂和达帕替尼的联合疗法可能是一种可行的替代选择;然而,目前有关联合用药的数据还很有限。目的评估奥达替尼和生物制剂联合治疗难治性银屑病和银屑病关节炎的安全性和有效性。设计:我们报告了对一家医疗机构的 3 名患者进行回顾性病历审查的结果,这些患者接受了奥达帕替尼和生物制剂联合治疗银屑病和 PsA。在开始使用达帕替尼和生物制剂联合疗法前,患者已讨论了其风险、益处和安全警告。在与医疗服务提供者共同决策后,患者接受了联合疗法试验。结果这三位患者之前对单药治疗无效,在接受联合治疗后症状均有明显改善。治疗期间未出现不良反应(恶性肿瘤、心血管事件、静脉血栓栓塞)。不过,一名患者在接受联合治疗期间出现了带状疱疹爆发和憩室炎。要确定长期疗效,还需要进行更多的样本研究。
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引用次数: 0
Scratching the Surface: A Paraneoplastic Pruritus Phenomenon in a Mediastinal Mass 表面划痕:纵隔肿块中的副肿瘤性瘙痒现象
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.24
Devine Thomas, Yousef Salem, Naser Asfoor, Mohamad Jabin, Mojahed M. K. Shalabi, Stephen Baker, Shovendra Gautam
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引用次数: 0
Unusual Presentation of Marginal Zone Lymphoma 边缘区淋巴瘤的不寻常表现
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.9
Akash Rau, Anne Opalikhin, Kaitlin Kreuser, R. Ashack, Kurt Ashack
Primary cutaneous B-cell lymphomas (pcBCLs) are non-Hodgkin lymphomas that present with exclusive cutaneous disease at the time of diagnosis. The lesions may be indolent, subtle, or waxing and waning, making the diagnosis challenging. The ear is an uncommon location, and its frequency of involvement remains unknown. A retrospective analysis of a single patient with primary cutaneous marginal zone lymphoma of the ear. The patient was a 44-year-old male who presented with erythematous tender nodules on the left superior posterior helix. Shave biopsy revealed mixed dermal lymphoid infiltrate with eosinophils and telangiectasias. The patient was diagnosed with angiolymphoid hyperplasia with eosinophilia. Multiple treatment regiments were trialed but failed due to lack of therapeutic response, relapse, and clinical progression. The patient ultimately underwent Mohs surgery with biopsy findings that demonstrated CD-20 positive cells. Evaluation with B-cell histopathology and gene rearrangement studies confirmed the diagnosis of marginal zone lymphoma. This case report shows a unique presentation of marginal zone lymphoma of the ear, initially diagnosed as angiolymphoid hyperplasia with eosinophilia. Our findings demonstrate a relatively rare presentation for this disease and highlight the challenges in diagnosing primary cutaneous marginal zone lymphoma.
原发性皮肤 B 细胞淋巴瘤(pcBCL)是一种非霍奇金淋巴瘤,确诊时仅表现为皮肤疾病。病变可能不明显、不明显或时好时坏,因此诊断具有挑战性。耳部淋巴瘤并不常见,其受累频率仍不清楚。本文对一名耳部原发性皮肤边缘区淋巴瘤患者进行了回顾性分析。患者是一名 44 岁的男性,左耳后上螺旋出现红斑触痛性结节。刮片活检显示真皮淋巴混合浸润,伴有嗜酸性粒细胞和毛细血管扩张。患者被诊断为血管淋巴细胞增生症伴嗜酸性粒细胞增多。患者接受了多种治疗方案,但都因缺乏治疗反应、复发和临床进展而失败。患者最终接受了莫氏手术,活检结果显示细胞 CD-20 呈阳性。B 细胞组织病理学评估和基因重排研究证实了边缘区淋巴瘤的诊断。本病例报告显示了耳部边缘区淋巴瘤的独特表现,最初诊断为血管淋巴样增生伴嗜酸性粒细胞增多。我们的研究结果表明,这种疾病的表现形式相对罕见,并强调了诊断原发性皮肤边缘区淋巴瘤所面临的挑战。
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引用次数: 0
Primary Cutaneous Apocrine Carcinoma: Diagnostic and Management Difficulties of an Uncommon Neoplasm 原发性皮肤腺分泌癌:一种不常见肿瘤的诊断和管理难题
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.20
Mounia Sidki, M. Aboudourib, Maryam El Ouazzani, A. Belbachir, H. Rais, Yassine Benchamkha, O. Hocar, S. Amal
Primary cutaneous apocrine carcinoma (PCAC) is a rare adnexal neoplasm. Due to its clinical presentation, it can mimic other tumors, especially metastasis. We present a case of primary apocrine carcinoma of the axilla mimicking breast carcinoma metastasis in a 54-year-old woman. The patient presented with a painless, lobed, and erythematous nodule in the left axilla of one year duration. We suspected an adnexal neoplasm but also considered metastasis from breast carcinoma. Histological examination revealed apocrine proliferation. Immunohistochemistry stains of tumor cells were positive for AE1/AE3, and negative for CK7, CK20, p63 and GATA3. Radiological explorations did not reveal any signs of malignancy. Considering all these features, the conclusive diagnosis was primary cutaneous apocrine carcinoma. Treatment was wide excision without adjuvant therapy, and no recurrence was noted. Due to the rarity of this carcinoma, its various clinical presentations, and histological overlaps with breast cancer metastasis, making the diagnosis may be challenging. A comprehensive evaluation of all findings is necessary to rule out other tumors. There is any consensus on the management strategy. The standard treatment is surgical excision, but adjuvant therapies such as radiotherapy or chemotherapy can be proposed in advanced stage disease.
原发性皮肤腺癌(PCAC)是一种罕见的附件肿瘤。由于其临床表现,它可以模仿其他肿瘤,尤其是转移瘤。我们介绍了一例腋窝原发性腺分泌癌,患者为 54 岁女性,模仿乳腺癌转移。患者左侧腋窝无痛、分叶、红斑结节,病程一年。我们怀疑是附件肿瘤,但也考虑是乳腺癌转移。组织学检查显示该肿瘤为腺体增生。肿瘤细胞的免疫组化染色显示 AE1/AE3 阳性,CK7、CK20、p63 和 GATA3 阴性。放射学检查未发现任何恶性肿瘤迹象。考虑到所有这些特征,最终诊断为原发性皮肤腺分泌癌。治疗方法是大范围切除,没有辅助治疗,也没有发现复发。由于这种癌症非常罕见,临床表现多种多样,而且在组织学上与乳腺癌转移重叠,因此诊断可能具有挑战性。有必要对所有检查结果进行综合评估,以排除其他肿瘤。关于治疗策略,目前尚无共识。标准的治疗方法是手术切除,但对于晚期患者,可采用放疗或化疗等辅助疗法。
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引用次数: 0
Incontinentia Pigmenti In a Female Infant: A Case Report 一名女婴的猪尿失禁:病例报告
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.11
Rebecca Lapides, Hannah J Porter, Deborah Cook, Keith Morley
Incontinentia pigmenti (IP) is an X-linked disorder characterized by a variety of findings that affect different systems, including cutaneous, ophthalmologic, neurologic, and dental. The cutaneous findings occur in 4 consecutive stages, generally referred to as vesicular, verrucous, hyperpigmented, and atrophic. The first stage is often observed in infancy, and this may be the first indication of IP in an infant. Early diagnosis is critical so that interventions can be implemented as soon as possible to monitor for complications and mitigate the effects of IP given the potential for multisystem involvement. Here, we describe a case of IP that was diagnosed in a female infant with no prior family history of IP. Prompt diagnosis and family counseling is critical, as early interventions can help optimize patients’ quality of life and genetic testing may help families make informed future family planning decisions. 
色素失禁症(IP)是一种 X 连锁障碍性疾病,其特征是影响皮肤、眼科、神经和牙科等不同系统的多种病变。皮肤症状分为四个连续的阶段,一般称为水泡期、疣状期、色素沉着期和萎缩期。第一阶段通常在婴儿期出现,这可能是婴儿患 IP 的第一个迹象。鉴于 IP 可能累及多个系统,因此早期诊断至关重要,这样才能尽快采取干预措施,监测并发症并减轻 IP 的影响。在此,我们描述了一例被确诊为 IP 的女婴,她之前并无 IP 家族史。及时诊断和家庭咨询至关重要,因为早期干预有助于优化患者的生活质量,基因检测可帮助家庭做出明智的未来计划生育决定。
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引用次数: 0
Eruptive Epidermal Inclusion Cysts in a Renal Transplant Patient on Tacrolimus 一名服用他克莫司的肾移植患者出现的溃疡性表皮包涵囊肿
Pub Date : 2024-05-13 DOI: 10.25251/skin.8.3.13
Meredith Burns, Hoang Ho-Pham, Lauren Kole
Epidermal inclusion cysts are common cutaneous lesions that can develop anywhere on the body. While benign, these cysts can become inflamed and symptomatic. Symptomatic epidermal inclusion cysts can be treated with intralesional steroid injections or antibiotics, but surgical excision is often necessary for definitive treatment. We present a unique case of tacrolimus-induced eruptive epidermal inclusion cysts on the head and trunk of an adult male post-renal transplantation. Physicians should be familiar with this potential adverse effect, especially due to the difficulty of obtaining definitive treatment for a multitude of epidermal inclusion cysts.
表皮包涵囊肿是一种常见的皮肤病变,可发生在身体的任何部位。这些囊肿虽然是良性的,但也会发炎和出现症状。无症状的表皮包涵体囊肿可通过皮内注射类固醇或抗生素来治疗,但通常需要手术切除才能彻底治愈。我们介绍了一例肾移植术后由他克莫司诱发的成年男性头部和躯干爆发性表皮包涵囊肿的独特病例。医生应该了解这种潜在的不良反应,尤其是由于多种表皮包涵囊肿难以获得明确的治疗。
{"title":"Eruptive Epidermal Inclusion Cysts in a Renal Transplant Patient on Tacrolimus","authors":"Meredith Burns, Hoang Ho-Pham, Lauren Kole","doi":"10.25251/skin.8.3.13","DOIUrl":"https://doi.org/10.25251/skin.8.3.13","url":null,"abstract":"Epidermal inclusion cysts are common cutaneous lesions that can develop anywhere on the body. While benign, these cysts can become inflamed and symptomatic. Symptomatic epidermal inclusion cysts can be treated with intralesional steroid injections or antibiotics, but surgical excision is often necessary for definitive treatment. We present a unique case of tacrolimus-induced eruptive epidermal inclusion cysts on the head and trunk of an adult male post-renal transplantation. Physicians should be familiar with this potential adverse effect, especially due to the difficulty of obtaining definitive treatment for a multitude of epidermal inclusion cysts.","PeriodicalId":22013,"journal":{"name":"SKIN The Journal of Cutaneous Medicine","volume":"107 10","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140986107","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
期刊
SKIN The Journal of Cutaneous Medicine
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