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Pigmented Superficial Basal Cell Carcinoma of the Nipple-Areola Complex: A Case Report. 乳头-乳晕复合体色素浅表基底细胞癌1例报告。
IF 0.9 Q4 DERMATOLOGY Pub Date : 2025-01-16 eCollection Date: 2025-01-01 DOI: 10.1159/000542168
Xinru Chen, Na Zhang, Hongping Ge, Xiaoli Zhai, Meiyan Wang, Min Zhang

Introduction: Basal cell carcinoma (BCC) is the most common type of skin malignancy, accounting for approximately 80% of all non-melanoma skin cancers (NMSCs). Ultraviolet (UV) exposure is a significant risk factor for BCC development, which typically occurs in sun-exposed areas. BCC arising in non-sun-exposed regions, such as the nipple-areola complex (NAC), is exceedingly rare, with fewer than 100 cases reported globally. This report describes a case of pigmented superficial BCC in the NAC of a 76-year-old Asian woman.

Case presentation: A 76-year-old Asian female presented with a 5-year history of a slowly enlarging lesion on her left breast, with recent rapid growth. Physical examination revealed a 10 mm × 8 mm blue-gray, pearl-like plaque on the NAC. Histopathology confirmed pigmented superficial BCC. Preoperative imaging, including breast ultrasound, chest computed tomography (CT), SPECT-CT, and axillary lymph node ultrasound, showed no evidence of metastasis. The patient underwent standard surgical excision with a 10 mm margin, followed by pathologic evaluation, confirming clear margins. The patient was discharged on the second postoperative day and remained asymptomatic at a 3-month follow-up.

Conclusion: Pigmented superficial BCC of the NAC is an uncommon presentation due to the area's minimal sun exposure and lack of pilosebaceous units. This case underscores the importance of considering BCC in non-sun-exposed areas, particularly in elderly patients. While nonsurgical options such as photodynamic therapy may offer superior esthetic outcomes, the patient's financial constraints led to the selection of a cost-effective surgical excision, which successfully eliminated the tumor.

基底细胞癌(BCC)是最常见的皮肤恶性肿瘤,约占所有非黑色素瘤皮肤癌(NMSCs)的80%。紫外线(UV)暴露是BCC发展的重要危险因素,通常发生在阳光照射的地区。BCC发生在非阳光照射区域,如乳头乳晕复合体(NAC),是非常罕见的,全球报告的病例不到100例。本文报告一位76岁的亚洲女性,在NAC中发生色素浅表性基底细胞癌。病例介绍:76岁亚洲女性,5年左乳病变缓慢增大,近期快速增长。体格检查发现NAC上有一个10 mm × 8 mm的蓝灰色珍珠样斑块。组织病理学证实为色素浅表性基底细胞癌。术前影像学检查,包括乳房超声、胸部CT、SPECT-CT和腋窝淋巴结超声,均未发现转移的证据。患者接受标准手术切除,边缘为10毫米,随后进行病理评估,确认边缘清晰。患者于术后第二天出院,随访3个月无症状。结论:NAC的色素浅表性基底细胞癌是一种罕见的表现,因为该区域的阳光照射最少,缺乏毛囊皮脂腺单位。该病例强调了在非阳光照射区域,特别是老年患者中考虑基底细胞癌的重要性。虽然光动力疗法等非手术治疗可能会带来更好的美观效果,但由于患者经济拮据,他们选择了经济有效的手术切除,最终成功地消除了肿瘤。
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引用次数: 0
Verrucous Eccrine Angiomatous Hamartoma: A Case Report.
IF 0.9 Q4 DERMATOLOGY Pub Date : 2025-01-16 eCollection Date: 2025-01-01 DOI: 10.1159/000533667
Ali Sadeghinia, Yasaman Sadeghi, Alireza Ghanadan, Elham Mazaherpour

Eccrine angiomatous hamartoma (EAH) is a rare malformation presenting with the proliferation of eccrine glands and other dermal structures. It typically presents as a solitary lesion on extremities that appears at birth or during childhood. It is generally asymptomatic but may be associated with hyperhidrosis or pain. Here we report a case of verrucous EAH located on the heel of a 27-year-old man, which is an uncommon presentation of this tumor, and review the clinical characteristics, histologic findings, and prognosis of this rare condition.

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引用次数: 0
Frieden's Group-V Aplasia Cutis Congenita with in utero Scarring in a Dichorionic Twin: A Case Report and Comprehensive Literature Review. 弗里登氏v组先天性皮肤发育不全伴双绒毛膜双胞胎子宫瘢痕:1例报告及综合文献复习。
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-12-19 eCollection Date: 2025-01-01 DOI: 10.1159/000541676
Anvar Paraparambil Vellamgot, Rajesh Pattu Valappil, Ahmed Hosny Tomerak, Hakam Khatib, Khalil Salameh

Introduction: Aplasia cutis congenita (ACC) is a rare congenital disorder characterized by the localized absence of skin at birth, primarily affecting the scalp but also affecting the trunk and limbs. Nine different presentations have been reported. Group V-ACC (G-V ACC) is a rare type associated with fetus papyraceus. The pathogenesis is speculated to involve ischemia from acute hypovolemia or disseminated intravascular coagulation due to thrombotic tissue affecting the surviving twin. Management of ACC depends on lesion severity, focusing on preventing infection and promoting healing, with conservative measures in most cases and surgical intervention in some cases. The long-term prognosis for isolated G-V ACC is generally favorable unless associated with systemic complications.

Case presentation: We report a neonate diagnosed with Group G-V ACC associated with fetus papyraceus from a dichorionic diamniotic twin pregnancy. Born to a 28-year-old Asian mother, this female infant presented with extensive, healed, scar-like lesions on her flanks and upper thighs. These lesions were consistent with the "H"-shaped distribution commonly described in ACC linked to fetus papyraceus. Despite significant dermatological manifestations, the infant showed no other external abnormalities or systemic involvement, and comprehensive screenings, including ultrasounds and chromosomal microarray, were normal.

Conclusion: This discussion highlights the rarity of ACC associated with fetus papyraceus, with only a few over 100 cases reported globally. This case underscores the importance of recognizing the patterns and potential complications of ACC in twins, contributing to better diagnostic acumen and management strategies for neonatal care.

摘要:先天性皮肤发育不全(Aplasia cutis congenita, ACC)是一种罕见的先天性疾病,以出生时局部皮肤缺失为特征,主要影响头皮,但也影响躯干和四肢。有九种不同的报告。v型ACC (G-V型ACC)是一种罕见的与胎儿纸莎草病相关的类型。发病机制推测与急性低血容量引起的缺血或由于血栓组织影响幸存的双胞胎引起的弥散性血管内凝血有关。ACC的治疗取决于病变的严重程度,重点是预防感染和促进愈合,大多数情况下采取保守措施,部分情况下采取手术干预。孤立性G-V型ACC的长期预后通常是良好的,除非伴有全身并发症。病例介绍:我们报告一个新生儿诊断为G-V组ACC与双绒毛膜双羊膜双胎妊娠的胎儿纸赘。这名28岁的亚洲母亲所生的女婴,在她的侧腹和大腿上部出现了广泛的、愈合的疤痕样病变。这些病变与胎儿纸莎草病相关ACC常见的“H”形分布一致。尽管有明显的皮肤病学表现,但婴儿未显示其他外部异常或全身受累,包括超声和染色体微阵列在内的综合筛查均正常。结论:本研究强调了ACC合并胎儿纸莎草病的罕见性,全球仅有100例以上的病例报道。本病例强调了识别双胞胎ACC的模式和潜在并发症的重要性,有助于更好的诊断敏锐度和新生儿护理管理策略。
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引用次数: 0
Ecthyma Gangrenosum of Fungal Origin: A Case Report. 源于真菌的坏疽性外皮藓:病例报告
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-11-22 eCollection Date: 2024-01-01 DOI: 10.1159/000542105
Germán Andrés León-Sánchez, Heiler Lozada-Ramos, Jorge Enrique Daza-Arana, Andrés Darío Restrepo-Becerra, Ruben Varela-Miranda

Introduction: Ecthyma gangrenosum (EG) is usually a dermatologic manifestation of a Pseudomonas aeruginosa infection in an immunocompromised individual but may sometimes be caused by other bacteria or fungi in an immunocompromised or non-immunocompromised individual.

Case presentation: A 75-year-old woman with a history of high blood pressure and sequels of ischemic cerebral infarction presented with a 5-day history of general malaise, cough with yellow sputum, and respiratory distress. The patient had pale mucous membranes, temperature of 38.5°C, tachycardia, normal blood pressure, SaO2 of 85%, intercostal retractions, and severe bronchospasm upon hospital admission. No skin lesions were seen. The patient was admitted to the intensive care unit (ICU) because of her critical condition and was supported with invasive mechanical ventilation. Her blood count showed 8,100 leukocytes/mm3, neutrophils 79%, hemoglobin 10.1 g/dL, creatinine 1.1 mg/dL, and C-reactive protein 328 mg/dL. Arterial blood gases showed metabolic acidosis and moderate hypoxemia. The initial report of blood and urine cultures was negative for bacteria, and positive for influenza A H1N1. The patient was treated with oseltamivir and intravenous methylprednisolone for acute respiratory distress syndrome associated with the viral infection that occurred. Subsequently, violaceus macular and papular lesions appeared, which evolved into ulcerated lesions with erythematous border and necrotic center were seen in the anterior region of the chest and abdomen, from where Candida metapsilosis was isolated. EG was reported in this patient, who was also immunocompromised because of steroid use, had a prolonged stay in the ICU and received broad-spectrum antibiotics. Fungemia and urinary infection due to different fungi were also found.

Conclusion: It is worth mentioning that EG can be caused by germs other than P. aeruginosa and fungal infections should not be ruled out.

导言:坏疽性外皮藓(EG)通常是免疫力低下者感染铜绿假单胞菌后的皮肤病表现,但有时也可能由免疫力低下或非免疫力低下者感染的其他细菌或真菌引起:一名 75 岁的女性患者有高血压和缺血性脑梗塞后遗症,5 天前出现全身不适、咳嗽并伴有黄痰和呼吸困难。患者入院时粘膜苍白,体温 38.5°C,心动过速,血压正常,SaO2 85%,肋间回缩,支气管痉挛严重。未见皮肤损伤。由于病情危重,患者被送入重症监护室(ICU),并接受了有创机械通气。她的血细胞计数显示白细胞为 8100 个/立方毫米,中性粒细胞为 79%,血红蛋白为 10.1 克/分升,肌酐为 1.1 毫克/分升,C 反应蛋白为 328 毫克/分升。动脉血气显示代谢性酸中毒和中度低氧血症。血液和尿液培养的初步报告显示细菌阴性,甲型 H1N1 流感阳性。患者因病毒感染引发急性呼吸窘迫综合征,接受了奥司他韦和甲泼尼龙静脉注射治疗。随后,胸腹部前区出现大疱性和丘疹性病变,进而演变为溃疡性病变,边缘红斑,中心坏死,并从中分离出念珠菌性甲沟炎。该患者因使用类固醇而免疫力低下,在重症监护室住院时间较长,并接受了广谱抗生素治疗。此外,还发现了由不同真菌引起的菌血症和泌尿系统感染:值得一提的是,除铜绿假单胞菌外,其他病菌也可能引起 EG,因此不应排除真菌感染的可能性。
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引用次数: 0
Keratoderma-Like T-Cell Dyscrasia: A Case Report. 角化病样 T 细胞瘤:病例报告
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-10-25 eCollection Date: 2024-01-01 DOI: 10.1159/000541258
Asem Shadid, Sukaina Al Haddad, Rua'a Alharithy, Tariq J Al-Zaid

Introduction: Cutaneous T-cell dyscrasia (CTCD) encompasses a heterogeneous group of T-cell infiltrates, often clonal and epitheliotropic. While the etiology remains unclear, certain medications, including statins, have been linked to cutaneous T-cell lymphocytic infiltrate development.

Case description: A patient presented with a pruritic, scaly eruption on her palms and soles unresponsive to topical steroids for 1 month. Histopathological examination revealed compact orthokeratosis, mild lymphocytic infiltrate with focal exocytosis, and atypical lymphocytes. Immunophenotyping demonstrated a predominance of CD3+ T cells with a 1:1 CD4/CD8 ratio and reduced CD7 expression. The clinical presentation, histopathology, and immunophenotype supported a diagnosis of statin-induced CTCD.

Conclusion: Statin discontinuation led to complete symptom resolution, emphasizing the reversibility of drug-induced T-cell dyscrasia. This case highlights the importance of a detailed medication history as drug-induced T-cell dyscrasia, unlike classic CTCD with its characteristic lymphoid atypia, phenotypic abnormalities, and clonality leading to a refractory course, can be reversed by drug withdrawal.

导言:皮肤T细胞发育不良(CTCD)包括一组异质性的T细胞浸润,通常具有克隆性和上皮细胞性。虽然病因尚不清楚,但包括他汀类药物在内的某些药物与皮肤 T 细胞淋巴细胞浸润的发生有关:患者的手掌和脚底出现瘙痒性鳞屑性糜烂,外用类固醇激素治疗 1 个月无效。组织病理学检查显示,她的皮肤出现紧密角化、轻度淋巴细胞浸润并伴有局灶性外渗和非典型淋巴细胞。免疫分型显示,CD3+ T 细胞占多数,CD4/CD8 比率为 1:1,CD7 表达减少。临床表现、组织病理学和免疫分型均支持他汀类药物诱发 CTCD 的诊断:结论:停用他汀类药物后症状完全缓解,强调了药物诱导的T细胞病变的可逆性。本病例强调了详细用药史的重要性,因为药物诱导的T细胞异常不同于典型的CTCD,后者具有淋巴细胞不典型性、表型异常和克隆性等特征,会导致难治性病程,而停药则可逆转。
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引用次数: 0
Syringotropic Lichen Planus: An Unusual Presentation of a Common Dermatosis - A Report of 2 Cases. 注射性扁平苔藓:常见皮肤病的不寻常表现--2 例报告。
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-10-24 eCollection Date: 2024-01-01 DOI: 10.1159/000541695
Fatemeh Mohaghegh, Zohre Khodashenas, Mina Saber, Haniyeh Sohrabi

Introduction: Lichen planus (LP) is a chronic inflammatory dermatosis that causes plaques and itchy papules on the skin, as well as erosion and ulcers in the mucous membranes. LP is characterized by a dense dermal T-cell infiltration. Perieccrine inflammation can be seen in a variety of dermatoses, but genuine lymphocyte permeation of the secretory coil or lymphocytic syringotropism is a rare sign that is typically seen in mycosis fungoides.

Case presentation: In this study, we present 2 uncommon instances of lymphocytic syringotropism in LP. Histopathological examination revealed dense T-cell infiltration and lymphocytic involvement of eccrine glands, confirming syringotropism.

Conclusion: Lymphocytic syringotropism is an uncommon finding in LP. Its presence broadens the histopathological spectrum of LP and highlights the need to differentiate it from lymphoproliferative disorders like mycosis fungoides.

简介扁平苔藓(LP)是一种慢性炎症性皮肤病,会导致皮肤出现斑块和瘙痒性丘疹,以及黏膜糜烂和溃疡。LP 的特点是真皮层 T 细胞密集浸润。周皮炎症可见于多种皮肤病,但真正的淋巴细胞渗透分泌线圈或淋巴细胞鞘膜炎是一种罕见的体征,通常见于真菌病:在本研究中,我们发现了两例不常见的LP淋巴细胞虹吸现象。组织病理学检查显示,T细胞密集浸润,淋巴细胞累及泌尿生殖腺,证实了淋巴细胞性虹膜炎:结论:淋巴细胞性虹吸症在 LP 中并不常见。淋巴细胞嗜嗜酸性粒细胞增多症是 LP 中不常见的发现,它的出现拓宽了 LP 的组织病理学范围,突出了将 LP 与真菌病等淋巴增生性疾病区分开来的必要性。
{"title":"Syringotropic Lichen Planus: An Unusual Presentation of a Common Dermatosis - A Report of 2 Cases.","authors":"Fatemeh Mohaghegh, Zohre Khodashenas, Mina Saber, Haniyeh Sohrabi","doi":"10.1159/000541695","DOIUrl":"https://doi.org/10.1159/000541695","url":null,"abstract":"<p><strong>Introduction: </strong>Lichen planus (LP) is a chronic inflammatory dermatosis that causes plaques and itchy papules on the skin, as well as erosion and ulcers in the mucous membranes. LP is characterized by a dense dermal T-cell infiltration. Perieccrine inflammation can be seen in a variety of dermatoses, but genuine lymphocyte permeation of the secretory coil or lymphocytic syringotropism is a rare sign that is typically seen in mycosis fungoides.</p><p><strong>Case presentation: </strong>In this study, we present 2 uncommon instances of lymphocytic syringotropism in LP. Histopathological examination revealed dense T-cell infiltration and lymphocytic involvement of eccrine glands, confirming syringotropism.</p><p><strong>Conclusion: </strong>Lymphocytic syringotropism is an uncommon finding in LP. Its presence broadens the histopathological spectrum of LP and highlights the need to differentiate it from lymphoproliferative disorders like mycosis fungoides.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"226-233"},"PeriodicalIF":0.9,"publicationDate":"2024-10-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11501101/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142495746","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
DRESS Syndrome That Resembles Graft-Versus-Host Disease after Chemotherapy in a Pediatric Patient: A Case Report. 一名儿科患者化疗后出现类似移植物抗宿主病的 DRESS 综合征:病例报告。
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-10-08 eCollection Date: 2024-01-01 DOI: 10.1159/000541046
Marian Rolón, Mateo Barros, Clara Ortiz, Sergio Danilo Cruz Romero, Johanna Álvarez

Introduction: Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a potentially life-threatening, drug-induced adverse reaction characterized by skin eruptions, lymphadenopathy, fever, and a broad range of other bodily manifestations. The spectrum of histopathologic and clinical presentations is wide; therefore, DRESS syndrome can mimic other diseases.

Case presentation: We present a case of a 4-year-old male patient who started chemotherapy with vincristine, cytarabine, and etoposide. The first clinical signs were fever, hemodynamic in-stability, and maculopapular erythema. Biopsies of skin lesions were taken, and hyperkeratosis, focal parakeratosis, acanthosis with slight spongiosis, and intraepithelial dyskeratotic cells were observed. There was a perivascular lymphoid infiltrate with abundant eosinophils in the dermis, and eosinophil permeations to the acrosyringium and epithelium were found.

Conclusion: DRESS syndrome is a drug-induced reaction that shares histopathological findings in skin biopsies with those seen in graft-versus-host disease. Although the histological findings are non-pathognomonic, they were characteristic enough to be of importance in the differential diagnosis.

简介:伴有嗜酸性粒细胞增多和全身症状的药物反应(DRESS)综合征是一种可能危及生命的药物诱发不良反应,以皮肤糜烂、淋巴结病、发热和一系列其他身体表现为特征。组织病理学和临床表现的范围很广,因此,DRESS 综合征可能会模仿其他疾病:本病例为一名 4 岁男性患者,开始接受长春新碱、阿糖胞苷和依托泊苷化疗。最初的临床症状是发热、血流动力学不稳定和斑丘疹性红斑。对皮损进行了活检,观察到角化过度、局灶性角化不全、伴有轻微海绵状增生的棘皮症和上皮内角化不良细胞。真皮内有大量嗜酸性粒细胞的血管周围淋巴浸润,并发现嗜酸性粒细胞渗透到棘层和上皮细胞:结论:DRESS 综合征是一种药物诱导的反应,其皮肤活检组织病理学结果与移植物抗宿主病相同。虽然这些组织学发现不具病理特征,但在鉴别诊断中具有重要意义。
{"title":"DRESS Syndrome That Resembles Graft-Versus-Host Disease after Chemotherapy in a Pediatric Patient: A Case Report.","authors":"Marian Rolón, Mateo Barros, Clara Ortiz, Sergio Danilo Cruz Romero, Johanna Álvarez","doi":"10.1159/000541046","DOIUrl":"https://doi.org/10.1159/000541046","url":null,"abstract":"<p><strong>Introduction: </strong>Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a potentially life-threatening, drug-induced adverse reaction characterized by skin eruptions, lymphadenopathy, fever, and a broad range of other bodily manifestations. The spectrum of histopathologic and clinical presentations is wide; therefore, DRESS syndrome can mimic other diseases.</p><p><strong>Case presentation: </strong>We present a case of a 4-year-old male patient who started chemotherapy with vincristine, cytarabine, and etoposide. The first clinical signs were fever, hemodynamic in-stability, and maculopapular erythema. Biopsies of skin lesions were taken, and hyperkeratosis, focal parakeratosis, acanthosis with slight spongiosis, and intraepithelial dyskeratotic cells were observed. There was a perivascular lymphoid infiltrate with abundant eosinophils in the dermis, and eosinophil permeations to the acrosyringium and epithelium were found.</p><p><strong>Conclusion: </strong>DRESS syndrome is a drug-induced reaction that shares histopathological findings in skin biopsies with those seen in graft-versus-host disease. Although the histological findings are non-pathognomonic, they were characteristic enough to be of importance in the differential diagnosis.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"221-225"},"PeriodicalIF":0.9,"publicationDate":"2024-10-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521526/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142543921","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Severe Nodulocystic Acne Induced by Mercury-Containing Skin-Whitening Cream: A Case Report and Call for Public Awareness. 含汞美白霜诱发的严重结节囊肿性痤疮:病例报告和提高公众认识的呼吁。
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-09-19 eCollection Date: 2024-01-01 DOI: 10.1159/000541141
Sami Alsuwaidan, Khalid Nabil Nagshabandi, Abdulmajeed Alajlan

Introduction: Skin-whitening products, often containing mercury, are used worldwide for cosmetic purposes but pose significant health risks. Mercury, a melanotoxin, can cause severe dermatological, renal, and neurological complications.

Case presentation: We report the case of a 27-year-old woman who developed severe nodulocystic acne due to chronic use of a mercury-containing skin-whitening cream. Treatment with isotretinoin and prednisolone led to significant improvement within 3 months.

Conclusion: This case underscores the need for public awareness and stricter regulatory actions to prevent mercury-related health hazards from skin-whitening products.

导言:美白产品通常含有汞,在全球范围内被用于美容目的,但会对健康造成严重危害。汞是一种黑色素毒素,可引起严重的皮肤病、肾病和神经系统并发症:我们报告了一例 27 岁女性的病例,她因长期使用含汞的美白霜而患上了严重的结节性痤疮。使用异维A酸和泼尼松龙治疗后,病情在 3 个月内明显好转:本病例强调了提高公众认识和采取更严格监管行动的必要性,以防止美白产品中与汞有关的健康危害。
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引用次数: 0
Treatment of a Complex Case of Scleromyxedema Using a Novel Intravenous Immunoglobulin Preparation. 使用新型静脉注射免疫球蛋白制剂治疗复杂的硬肌水肿病例
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-08-27 eCollection Date: 2024-01-01 DOI: 10.1159/000540519
Anastasia Sophie Vollmer, Nadine Wiedenmayer, Julia K Winkler, Alexander H Enk

Introduction: We provide a case report on a patient with scleromyxedema treated with high-dose intravenous immunoglobulins (IVIgs) who reported less fatigue and fewer headaches after transitioning to Yimmugo, a novel IVIg preparation. Scleromyxedema is a rare and chronic cutaneous mucinosis, frequently associated with monoclonal gammopathy and systemic complications, which may be life-threatening. The disease is characterized by papular eruptions and sclerosis of the skin. Treatment of scleromyxedema is challenging because of its unclear pathogenesis. High-dose IVIgs are a promising treatment option to improve cutaneous manifestations.

Case presentation: We present the case of a 57-year-old patient with scleromyxedema and monoclonal gammopathy with end-stage renal failure on hemodialysis (3 times a week). Stabilization of skin symptoms was finally achieved by high-dose IVIg therapy administered at 2 g per kg bodyweight distributed over 2 days every 3 weeks. However, disease stabilization came at the expense of flu-like side effects that significantly affected daily life. After transitioning to Yimmugo®, the patient reported an improved quality of life.

Conclusion: Further follow-up is essential to conclusively evaluate effectiveness and tolerability of this novel IVIg preparation.

导言:我们报告了一例接受大剂量静脉注射免疫球蛋白(IVIgs)治疗的硬肌水肿患者的病例,该患者在改用新型 IVIg 制剂 "Yimmugo "后,疲劳减轻,头痛减少。硬化性粘液性水肿是一种罕见的慢性皮肤粘液病,常伴有单克隆丙种球蛋白病和全身并发症,可能危及生命。该病的特征是皮肤丘疹性糜烂和硬化。由于发病机制不明确,硬皮水肿的治疗具有挑战性。大剂量静脉注射是改善皮肤表现的一种很有前景的治疗方法:本病例是一名 57 岁的硬肌水肿和单克隆丙种球蛋白病患者,终末期肾衰竭,接受血液透析治疗(每周 3 次)。通过每公斤体重 2 克、每 3 周注射 2 天的大剂量 IVIg 治疗,患者的皮肤症状最终得到稳定。然而,疾病的稳定是以流感样副作用为代价的,这些副作用严重影响了患者的日常生活。在转用 Yimmugo® 后,患者的生活质量有所改善:结论:要最终评估这种新型 IVIg 制剂的有效性和耐受性,必须进行进一步的随访。
{"title":"Treatment of a Complex Case of Scleromyxedema Using a Novel Intravenous Immunoglobulin Preparation.","authors":"Anastasia Sophie Vollmer, Nadine Wiedenmayer, Julia K Winkler, Alexander H Enk","doi":"10.1159/000540519","DOIUrl":"https://doi.org/10.1159/000540519","url":null,"abstract":"<p><strong>Introduction: </strong>We provide a case report on a patient with scleromyxedema treated with high-dose intravenous immunoglobulins (IVIgs) who reported less fatigue and fewer headaches after transitioning to Yimmugo, a novel IVIg preparation. Scleromyxedema is a rare and chronic cutaneous mucinosis, frequently associated with monoclonal gammopathy and systemic complications, which may be life-threatening. The disease is characterized by papular eruptions and sclerosis of the skin. Treatment of scleromyxedema is challenging because of its unclear pathogenesis. High-dose IVIgs are a promising treatment option to improve cutaneous manifestations.</p><p><strong>Case presentation: </strong>We present the case of a 57-year-old patient with scleromyxedema and monoclonal gammopathy with end-stage renal failure on hemodialysis (3 times a week). Stabilization of skin symptoms was finally achieved by high-dose IVIg therapy administered at 2 g per kg bodyweight distributed over 2 days every 3 weeks. However, disease stabilization came at the expense of flu-like side effects that significantly affected daily life. After transitioning to Yimmugo<sup>®</sup>, the patient reported an improved quality of life.</p><p><strong>Conclusion: </strong>Further follow-up is essential to conclusively evaluate effectiveness and tolerability of this novel IVIg preparation.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"210-215"},"PeriodicalIF":0.9,"publicationDate":"2024-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521433/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142543923","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Large Spontaneous Subcutaneous Abscess Formation due to Finegoldia magna in a Diabetic Patient: A Case Report. 糖尿病患者因细金丝桃而自发形成大面积皮下脓肿:病例报告。
IF 0.9 Q4 DERMATOLOGY Pub Date : 2024-08-23 eCollection Date: 2024-01-01 DOI: 10.1159/000540636
Toshiyuki Sato, Mayuka Tomita, Atsuhiro Kohno, Satomi Chujo, Yuma Waki, Yoshimasa Nobeyama, Masaaki Kawase, Akihiko Asahina

Introduction: Finegoldia magna is a member of the Gram-positive anaerobic cocci group and constitutes the flora of the skin and other parts of the body. It sometimes colonizes diabetic foot and rarely infects skin or soft tissue of non-immunocompromised patients.

Case presentation: Here, we report the case of a severe subcutaneous abscess on the back caused by F. magna involving an immunocompromised patient with poorly controlled diabetes. A 48-year-old woman with diabetes mellitus and anemia associated with uterine fibroids was referred to us with a 1-month history of a skin manifestation on her back, with a body temperature of 35.9°C and blood pressure of 115/73 mm Hg. The manifestation involved a subcutaneous mass of 36 × 45 cm with a foul odor, partly covered with necrotic tissue, which had the appearance of a tortoiseshell-like pattern. Blood examination revealed C-reactive protein of 21.4 mg/dL and hemoglobin A1c of 9.1%. Contrast-enhanced computed tomography showed a subcutaneous abscess with internal emphysema. Emergency debridement was performed, resulting in drainage of foul-smelling gray-green pus. F. magna was detected in the pus and skin tissue.

Conclusion: Skin and soft tissue infectious disease caused by F. magna is extremely rare, but the disease tends to become severe once developing in an immunocompromised patient, such as a patient with poorly controlled diabetes. Therefore, physicians should consider F. magna as a causative agent when poorly controlled diabetic patients suffer from severe infectious cutaneous manifestations.

简介细金葡菌(Finegoldia magna)属于革兰氏阳性厌氧球菌,是皮肤和身体其他部位的菌群成员。它有时会定植于糖尿病足,但很少感染非免疫力低下患者的皮肤或软组织:在此,我们报告了一例由 magna 真菌引起的背部严重皮下脓肿病例,该病例涉及一名免疫力低下的糖尿病患者。一名患有糖尿病和贫血并伴有子宫肌瘤的 48 岁女性转诊至我院,她的背部皮肤症状已有 1 个月的病史,体温为 35.9°C,血压为 115/73 mm Hg。症状表现为 36 × 45 厘米的皮下肿块,有恶臭,部分被坏死组织覆盖,外观呈玳瑁状。血液检查显示,C 反应蛋白为 21.4 毫克/分升,血红蛋白 A1c 为 9.1%。对比增强计算机断层扫描显示,患者皮下有脓肿,并伴有内部气肿。紧急清创后,排出了恶臭的灰绿色脓液。在脓液和皮肤组织中检测到蛆虫:结论:由巨大芽孢杆菌引起的皮肤和软组织感染性疾病极为罕见,但一旦在免疫力低下的患者(如糖尿病控制不佳的患者)中发病,病情往往会变得严重。因此,当病情控制不佳的糖尿病患者出现严重的皮肤感染表现时,医生应将麦地那龙线虫视为致病菌。
{"title":"Large Spontaneous Subcutaneous Abscess Formation due to <i>Finegoldia magna</i> in a Diabetic Patient: A Case Report.","authors":"Toshiyuki Sato, Mayuka Tomita, Atsuhiro Kohno, Satomi Chujo, Yuma Waki, Yoshimasa Nobeyama, Masaaki Kawase, Akihiko Asahina","doi":"10.1159/000540636","DOIUrl":"https://doi.org/10.1159/000540636","url":null,"abstract":"<p><strong>Introduction: </strong><i>Finegoldia magna</i> is a member of the Gram-positive anaerobic cocci group and constitutes the flora of the skin and other parts of the body. It sometimes colonizes diabetic foot and rarely infects skin or soft tissue of non-immunocompromised patients.</p><p><strong>Case presentation: </strong>Here, we report the case of a severe subcutaneous abscess on the back caused by <i>F</i>. <i>magna</i> involving an immunocompromised patient with poorly controlled diabetes. A 48-year-old woman with diabetes mellitus and anemia associated with uterine fibroids was referred to us with a 1-month history of a skin manifestation on her back, with a body temperature of 35.9°C and blood pressure of 115/73 mm Hg. The manifestation involved a subcutaneous mass of 36 × 45 cm with a foul odor, partly covered with necrotic tissue, which had the appearance of a tortoiseshell-like pattern. Blood examination revealed C-reactive protein of 21.4 mg/dL and hemoglobin A1c of 9.1%. Contrast-enhanced computed tomography showed a subcutaneous abscess with internal emphysema. Emergency debridement was performed, resulting in drainage of foul-smelling gray-green pus. <i>F</i>. <i>magna</i> was detected in the pus and skin tissue.</p><p><strong>Conclusion: </strong>Skin and soft tissue infectious disease caused by <i>F</i>. <i>magna</i> is extremely rare, but the disease tends to become severe once developing in an immunocompromised patient, such as a patient with poorly controlled diabetes. Therefore, physicians should consider <i>F</i>. <i>magna</i> as a causative agent when poorly controlled diabetic patients suffer from severe infectious cutaneous manifestations.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"202-209"},"PeriodicalIF":0.9,"publicationDate":"2024-08-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521465/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142550044","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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