Case Reports in Dermatology最新文献

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Focal Hypertrichosis during Topical Tacrolimus Therapy for Vitiligo: A Case Report. 局部他克莫司治疗白癜风期间局灶性多毛:1例报告。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-04-01 eCollection Date: 2025-01-01 DOI: 10.1159/000545115
Lingyu Mao, Yan Lin, Xueping Yu, Huaxu Liu
{"title":"Focal Hypertrichosis during Topical Tacrolimus Therapy for Vitiligo: A Case Report.","authors":"Lingyu Mao, Yan Lin, Xueping Yu, Huaxu Liu","doi":"10.1159/000545115","DOIUrl":"10.1159/000545115","url":null,"abstract":"<p><strong>Introduction: </strong>Tacrolimus is widely used in treating vitiligo. Drug-induced hypertrichosis is a well-known side effect of cyclosporine. It is less commonly associated with tacrolimus or with topical calcineurin inhibitors. Four reports of hypertrichosis attributed to topical or systemic tacrolimus have been previously reported in the literature. In this report, we describe a case of focal hypertrichosis due to long-term use of topical tacrolimus for vitiligo.</p><p><strong>Case presentation: </strong>We present a 15-year-old boy with focal hypertrichosis of white hair for 4 months, after treatment with topical tacrolimus 0.1% ointment. He denied ever using other ointment to the area. After he stopped using tacrolimus for half a month, the focal hypertrichosis improved.</p><p><strong>Conclusion: </strong>We propose that the focal hypertrichosis was induced by tacrolimus. The hair growth-stimulating effects of tacrolimus have been established. It is reported that tacrolimus directly stimulates mouse whiskers in vitro and prolongs the anagen phase of hair growth when it is applied topically in mice. We believe that a greater awareness of this rare side effect of tacrolimus is warranted as tacrolimus is widely used in treating vitiligo.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"106-109"},"PeriodicalIF":0.9,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11961153/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143763168","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
Drug Reaction with Eosinophilia and Systemic Symptoms Syndrome following Dalbavancin and Oritavancin Administration in a Patient with Osteomyelitis. 1例骨髓炎患者服用达尔巴万星和奥利塔万星后嗜酸性粒细胞增多和全身症状综合征的药物反应。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-03-28 eCollection Date: 2025-01-01 DOI: 10.1159/000545359
Jina Bai, Emily Frech Preciado, Mary Baxter Harlow, Nina Blank
{"title":"Drug Reaction with Eosinophilia and Systemic Symptoms Syndrome following Dalbavancin and Oritavancin Administration in a Patient with Osteomyelitis.","authors":"Jina Bai, Emily Frech Preciado, Mary Baxter Harlow, Nina Blank","doi":"10.1159/000545359","DOIUrl":"https://doi.org/10.1159/000545359","url":null,"abstract":"<p><strong>Introduction: </strong>Dalbavancin and oritavancin are newer long-acting antibiotics with potent activity against gram-positive organisms, including methicillin-resistant <i>Staphylococcus aureus</i> (MRSA). To our knowledge, there have been no reported cases of drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome in a patient treated with dalbavancin and oritavancin.</p><p><strong>Patient presentation: </strong>A woman in her 20s presented with a right thumb abscess and cellulitis that failed to respond to several courses of oral antibiotics, resulting in recurrent emergency room visits over 3 weeks. Approximately 1 month after the initial skin infection, magnetic resonance imaging revealed osteomyelitis of the right thumb. She was treated with a single dose of oritavancin followed by two weekly doses of dalbavancin, which successfully resolved the infection. However, she subsequently developed fever and a rash consistent with DRESS syndrome, likely triggered by oritavancin or dalbavancin. Given the prolonged half-life of these medications, she required treatment with high-dose steroids for an extended duration.</p><p><strong>Conclusion: </strong>Dalbavancin and oritavancin are second-generation lipoglycopeptide antibiotics that provide coverage for gram-positive organisms, including MRSA. They are approved for the treatment of acute bacterial skin and skin structure infections and are used off-label for bacteremia, endocarditis, and osteomyelitis. Their prolonged half-lives - 257 h for dalbavancin and 195 h for oritavancin - allow for less frequent dosing. However, a long half-life also leads to prolonged drug exposure in the event of adverse effects. Here, we report the first case of DRESS syndrome in a patient treated with dalbavancin and oritavancin.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"128-136"},"PeriodicalIF":0.9,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12040303/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143954851","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
Amelanotic Melanoma Arising in Surgical Cicatrix: A Case Report. 手术瘢痕无色素黑色素瘤一例报告。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-03-28 eCollection Date: 2025-01-01 DOI: 10.1159/000545287
Anna Louise Norling, Christian Beltoft Brøchner, Linnea Langhans
{"title":"Amelanotic Melanoma Arising in Surgical Cicatrix: A Case Report.","authors":"Anna Louise Norling, Christian Beltoft Brøchner, Linnea Langhans","doi":"10.1159/000545287","DOIUrl":"https://doi.org/10.1159/000545287","url":null,"abstract":"<p><strong>Introduction: </strong>Amelanotic melanoma (AM) is a rare subtype of melanoma characterized by the absence of pigmentation, making it difficult to diagnose. This case report describes AM arising in a cicatrix following cardiac surgery, highlighting diagnostic challenges and the importance of reassessment when the clinical findings do not fit the initial diagnosis.</p><p><strong>Case presentation: </strong>A 76-year-old male underwent coronary artery bypass grafting in March 2022. Nine months later, he noticed a mass with ulceration in the sternal cicatrix. A punch biopsy showed inflammation and fibrosis, leading to a diagnosis of suture granuloma. Despite the lesion's progression over 8 months, no further biopsies or treatments were performed. In September 2023, the patient was referred to the Department of Plastic Surgery at Copenhagen University Hospital, Rigshospitalet, where an excision biopsy revealed an AM with a thickness of 20 mm. Preoperative PET-CT showed no evidence of metastasis, and re-excision with sentinel lymph node biopsy was performed. Adjuvant oncologic treatment was administered due to a positive sentinel node.</p><p><strong>Conclusion: </strong>This case emphasizes the diagnostic challenges of AM, especially when arising in a cicatrix. When the clinical picture does not align with the initial diagnosis, it is crucial to reconsider and conduct further evaluations to avoid delays in treatment.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"123-127"},"PeriodicalIF":0.9,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12005706/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143962334","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
Guillain-Barré Syndrome Secondary to Herpes Zoster: A Rare Case Report of Death and Literature Review. 带状疱疹继发格林-巴利综合征1例死亡报告及文献复习。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-03-25 eCollection Date: 2025-01-01 DOI: 10.1159/000544954
Jingyu Wang, Xinyi Hou, Jin Zhang, Bo Ren, Bin Lu
{"title":"Guillain-Barré Syndrome Secondary to Herpes Zoster: A Rare Case Report of Death and Literature Review.","authors":"Jingyu Wang, Xinyi Hou, Jin Zhang, Bo Ren, Bin Lu","doi":"10.1159/000544954","DOIUrl":"https://doi.org/10.1159/000544954","url":null,"abstract":"<p><strong>Introduction: </strong>Herpes zoster (HZ) is a common infectious skin disease, and postherpetic neuralgia is the most feared and common complication. Guillain-Barré syndrome (GBS) is a rare complication of HZ.</p><p><strong>Case presentation: </strong>An exceptional case presenting GBS following HZ with a subsequent lethal issue is presented. A review of literature data revealed that middle-aged men with HZ were more likely to develop GBS. In the early stages of the disease, absent tendon reflexes and numbness of the limbs were common in patients.</p><p><strong>Conclusion: </strong>GBS should be kept in mind as rare HZ complication, particularly in middle-aged male patients.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"118-122"},"PeriodicalIF":0.9,"publicationDate":"2025-03-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12002728/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143964378","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
Disabling Plantar Keloids Treated with Brachytherapy and Free Flap Reconstruction: A Case Report. 近距离治疗和游离皮瓣重建致残性足底瘢痕1例报告。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-03-11 eCollection Date: 2025-01-01 DOI: 10.1159/000545053
Andrea Varazzani, Pierre Petolat, Louis Brochet, Alice Prevost, Nicolas Graillon, Antoine Pierrefeu
{"title":"Disabling Plantar Keloids Treated with Brachytherapy and Free Flap Reconstruction: A Case Report.","authors":"Andrea Varazzani, Pierre Petolat, Louis Brochet, Alice Prevost, Nicolas Graillon, Antoine Pierrefeu","doi":"10.1159/000545053","DOIUrl":"10.1159/000545053","url":null,"abstract":"<p><strong>Introduction: </strong>Keloids result from a disorganized fibroproliferative collagen response that extends beyond the original wound margins and fails to regress. They are associated with a high recurrence rate despite various treatment options. Keloids on the sole of the foot are exceedingly rare and pose significant challenges for both patients and surgeons. To date, only 15 cases of plantar keloids have been described in the English literature. Management options for this region include observation with custom-made footwear, steroid injections, surgery alone, or surgery followed by multimodal therapy. This report presents the first documented case of a plantar keloid treated with surgical excision, brachytherapy, and free flap reconstruction.</p><p><strong>Case presentation: </strong>We describe the case of a plantar keloid treated with surgical excision followed by high-dose-rate brachytherapy and free flap reconstruction. At 18 months postoperatively, the patient was ambulating independently without crutches, though hypersensitivity persisted. The scars at the donor site and the medial ankle exhibited keloids, but all scars subjected to brachytherapy were normal.</p><p><strong>Conclusion: </strong>The free flap approach may not be the optimal reconstruction method for plantar keloids, as skin grafts appear to have better outcomes, according to the literature. Also, in our patient, brachytherapy has proven its effectiveness in preventing further keloid formation as shown in the literature.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"96-105"},"PeriodicalIF":0.9,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11961157/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143763167","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
The Dynamic Sequential Comprehensive Treatment for Labia Majora Keloids: A Case Report and Literature Review. 动态顺序综合治疗大阴唇瘢痕疙瘩1例报告及文献复习。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-03-11 eCollection Date: 2025-01-01 DOI: 10.1159/000543932
Kun Yang, Jiayi Fu, Qiang Li
{"title":"The Dynamic Sequential Comprehensive Treatment for Labia Majora Keloids: A Case Report and Literature Review.","authors":"Kun Yang, Jiayi Fu, Qiang Li","doi":"10.1159/000543932","DOIUrl":"10.1159/000543932","url":null,"abstract":"<p><strong>Introduction: </strong>Keloids exhibit persistent growth beyond the original lesion, causing functional and cosmetic impairments. Keloids in the female genital region are especially rare.</p><p><strong>Case presentation: </strong>This report presents a unique case of bilateral labia majora keloids in a 35-year-old woman, persisting for over 20 years. Given the aesthetic importance of the labia majora, she underwent dynamic sequential comprehensive treatment centered on surgery. The patient was satisfied with the outcome, with no recurrence after 1 year. In addition to reviewing relevant literature, we evaluate the etiology, clinical manifestations, diagnosis, and treatment options for this condition.</p><p><strong>Conclusion: </strong>Dynamic sequential comprehensive treatment centered on surgery is effective in managing labia majora keloids. Emphasizing aesthetic outcomes is crucial. Early diagnosis and proper management are vital for alleviating symptoms and improving quality of life for affected individuals. Further research and increased awareness are essential for better understanding and managing labia majora keloids.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"110-117"},"PeriodicalIF":0.9,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11968096/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143779319","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
A Case of Juvenile Dermatomyositis Presenting with Inverse Gottron's Papules: A Case Report. 以逆Gottron丘疹为表现的青少年皮肌炎1例。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-03-11 eCollection Date: 2025-01-01 DOI: 10.1159/000544816
Renad AlKanaan, Iman I Nazer, Monira AlNasser
{"title":"A Case of Juvenile Dermatomyositis Presenting with Inverse Gottron's Papules: A Case Report.","authors":"Renad AlKanaan, Iman I Nazer, Monira AlNasser","doi":"10.1159/000544816","DOIUrl":"10.1159/000544816","url":null,"abstract":"<p><strong>Introduction: </strong>Inverse Gottron's papules are an uncommon presentation of dermatomyositis, with a paucity of studies published in children with juvenile dermatomyositis (JDM).</p><p><strong>Case presentation: </strong>A 5-year-old girl presented with progressive bilateral eyelid erythema and edema, mild erythematous rash over the face, painful palmar papules (inverse Gottron's papules), and proximal muscle weakness for 3 months. Additional symptoms included intermittent fever, decreased appetite, joint pain, and mild abdominal pain. Physical examination revealed upper and lower eyelid erythema with mild edema (heliotrope sign), Gottron's papules over the dorsal distal interphalangeal joint (DIP), Gottron's sign over the dorsal metacarpophalangeal (MCP) joints, proximal interphalangeal (PIP) joints and elbows, inverse Gottron's sign over the palmar surface of the hands (PIP and DIP), and ragged nailfold cuticles with no dilated capillaries. Laboratory investigations showed elevated muscle enzymes, positive myositis-specific antibodies, and elevated liver function tests. High-resolution computed tomography (HRCT) chest revealed no signs of interstitial lung disease (ILD). Whole-body magnetic resonance imaging (MRI) revealed bilateral upper limb subcutaneous edema and generalized myositis, particularly in the pelvis and thigh muscles, confirming the diagnosis of JDM. Treatment involved intravenous methylprednisolone 30 mg/kg/day for 3 days followed by tapering, and topical corticosteroids over dermatomyositis-affected areas.</p><p><strong>Conclusion: </strong>Inverse Gottron's papules are an uncommon cutaneous manifestation of dermatomyositis (DM) that have an association with ILD in both adults and children. Thus, patients with dermatomyositis and inverse Gottron's papules should be investigated for ILD. Our patient represents the first reported case of JDM with inverse Gottron's papules in Saudi Arabia.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"80-85"},"PeriodicalIF":0.9,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11952821/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143751323","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
Upadacitinib Monotherapy in Vitiligo Associated with Atopic Dermatitis: Killing Two Birds with One Stone. Upadacitinib单药治疗白癜风伴特应性皮炎:一石二鸟。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-02-28 eCollection Date: 2025-01-01 DOI: 10.1159/000544703
Lina Maria Magnanimi, Andrea De Berardinis, Maria Esposito, Maria Concetta Fargnoli
{"title":"Upadacitinib Monotherapy in Vitiligo Associated with Atopic Dermatitis: Killing Two Birds with One Stone.","authors":"Lina Maria Magnanimi, Andrea De Berardinis, Maria Esposito, Maria Concetta Fargnoli","doi":"10.1159/000544703","DOIUrl":"10.1159/000544703","url":null,"abstract":"<p><strong>Introduction: </strong>An increased risk of developing vitiligo has recently been described in patients with atopic dermatitis (AD). Vitiligo and AD can be associated because of shared pathogenetic pathways, including alterations in the Janus kinases/signal transducer and activator of transcription (JAK/STAT) signaling, suggesting JAK inhibitors as a promising new therapeutic approach in vitiligo.</p><p><strong>Case presentation: </strong>We describe a 25-year-old woman diagnosed with AD since childhood and subsequent onset of slowly progressive vitiligo at the age of 16. Systemic therapy with JAK1 inhibitor upadacitinib 15 mg daily was started, after a medical and laboratory evaluation to exclude pregnancy and other contraindications. Progressive improvement of AD was observed after the first weeks of treatment with clinical remission at week 16. At the same time, clear improvement of vitiligo was observed with an almost complete remission achieved at week 28 of treatment.</p><p><strong>Conclusion: </strong>The remission of both AD and vitiligo achieved with upadacitinib monotherapy supports the therapeutic utility of inhibition of JAK 1 signaling in these patients.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"91-95"},"PeriodicalIF":0.9,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11961158/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143763183","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
Recurrent Urticaria: A Rare Cryopyrin-Associated Periodic Syndrome - Muckle-Wells Syndrome. 复发性荨麻疹:一种罕见的低温素相关周期性综合征-粘液-井综合征。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-02-25 eCollection Date: 2025-01-01 DOI: 10.1159/000544705
Richa, Siddhi Gawhale
{"title":"Recurrent Urticaria: A Rare Cryopyrin-Associated Periodic Syndrome - Muckle-Wells Syndrome.","authors":"Richa, Siddhi Gawhale","doi":"10.1159/000544705","DOIUrl":"10.1159/000544705","url":null,"abstract":"<p><strong>Introduction: </strong>Muckle-Wells syndrome (MWS) is a rare cryopyrin-associated periodic syndrome (CAPS), an autoinflammatory disorder due to a loss of function mutation in the NACHT domain of the NLRP3 gene. The loss of cryopyrin activity brought on by this deficiency eventually causes dysregulated inflammation and increased release of the proinflammatory cytokine interleukin (IL)-1 beta. It has an autosomal dominant inheritance.</p><p><strong>Case presentation: </strong>We present here an 8-year-old girl with recurrent fever, recurrent urticarial rash, sensorineural hearing loss, raised inflammatory markers and serum amyloid levels, which did not respond to the anti-histaminic drugs, was wrongly diagnosed as tuberculosis, which on further genetic evaluation was diagnosed as MWS.</p><p><strong>Conclusion: </strong>Despite being uncommon, treating clinicians should take MWS into consideration given the specific clinical presentation. With the availability of appropriate medications, future complications can be prevented, and the prognosis is better.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"86-90"},"PeriodicalIF":0.9,"publicationDate":"2025-02-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11961156/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143763181","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
Bullous Vasculitis in Eosinophilic Granulomatosis with Polyangiitis: A Case Report. 嗜酸性肉芽肿伴多血管炎并发大疱性血管炎1例。
IF 0.9
Case Reports in Dermatology Pub Date : 2025-02-25 eCollection Date: 2025-01-01 DOI: 10.1159/000544815
Tiraporn Phumwiriya, Charussri Leeyaphan
{"title":"Bullous Vasculitis in Eosinophilic Granulomatosis with Polyangiitis: A Case Report.","authors":"Tiraporn Phumwiriya, Charussri Leeyaphan","doi":"10.1159/000544815","DOIUrl":"10.1159/000544815","url":null,"abstract":"<p><strong>Introduction: </strong>Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare systemic vasculitis affecting small- and medium-sized vessels. It is characterized by multiorgan involvement and can lead to severe outcomes if not diagnosed promptly. Cutaneous manifestations are common and typically include palpable purpura and subcutaneous nodules. Widespread bullous vasculitis affecting areas such as the forehead and ear presents an atypical presentation. We report a case of EGPA presenting with bullous vasculitis in an unusual location.</p><p><strong>Case presentation: </strong>A 40-year-old woman with a history of late-onset allergic rhinitis presented with a 2-week history of numbness in her right leg, along with multiple erythematous papules and vesicles, some with shallow erosions, located on the forehead and left ear. She also experienced fever, progressive dyspnea, and hemoptysis. She was diagnosed with pneumonitis, alveolar hemorrhage, and mononeuritis of the right leg. Laboratory findings revealed leukocytosis with eosinophilia, and the anti-myeloperoxidase antibody was positive. Histopathological examination of the bullous lesion on the forehead showed intraepidermal separation with necrotic keratinocytes and prominent eosinophil infiltration, along with focal leukocytoclastic vasculitis. The patient was diagnosed with EGPA and started on intravenous steroids and cyclophosphamide. EGPA is a rare disease characterized by multiorgan vasculitis, asthma, and granulomatous eosinophilic inflammation, which are its key hallmarks. While cutaneous involvement is common, bullous vasculitis is rarely observed on the forehead and ear.</p><p><strong>Conclusions: </strong>EGPA is a challenging diagnosis due to its variable presentation. While cutaneous manifestations are common, widespread bullous vasculitis may be atypical and rare clinical presentation. This case underscores the importance of considering EGPA in the differential diagnosis of bullous vasculitis, particularly when associated with systemic symptoms and eosinophilia. Early recognition and treatment are crucial for improving outcomes in this potentially life-threatening condition.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"74-79"},"PeriodicalIF":0.9,"publicationDate":"2025-02-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11936433/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143708674","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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