Case Reports in Dermatology最新文献

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Isolated Sclerodactyly Associated with Silica Exposure in a Stonemason: A Case Report. 石匠中与二氧化硅暴露相关的孤立性指硬症:一例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-11 eCollection Date: 2026-01-01 DOI: 10.1159/000552882
Vijay Selva, Arabella Wallett
{"title":"Isolated Sclerodactyly Associated with Silica Exposure in a Stonemason: A Case Report.","authors":"Vijay Selva, Arabella Wallett","doi":"10.1159/000552882","DOIUrl":"10.1159/000552882","url":null,"abstract":"<p><strong>Introduction: </strong>Sclerodactyly is commonly associated with systemic sclerosis (SSc), occurring alongside Raynaud's phenomenon and disease-specific autoantibodies. The combination of occupational silica exposure and SSc is known as Erasmus syndrome. However, isolated digital fibrosis in silica-exposed individuals without systemic features or SSc-specific autoantibody positivity has not yet been described.</p><p><strong>Case presentation: </strong>A 40-year-old stonemason presented with a 6-month history of progressive fingertip sclerosis, fissuring, and pain, in combination with hand dermatitis, which was initially diagnosed and treated as hand dermatitis alone. Examination revealed distal digital tapering, loss of skin markings, telangiectasia, and abnormal nailfold capillaries, together with dermatitis, without Raynaud's phenomenon or systemic involvement. High-resolution computed tomography demonstrated upper-lobe-predominant pulmonary nodules consistent with chronic silicosis. Autoimmune serology showed a weakly positive antinuclear antibody of 1:80 with negative SSc-specific autoantibodies. Under the 2013 ACR-EULAR criteria, the patient did not meet the diagnostic threshold for SSc. Dermatitis improved with exposure cessation and topical tacrolimus therapy, while sclerodactyly persisted.</p><p><strong>Conclusion: </strong>This case highlights isolated sclerodactyly as a potential manifestation of silica exposure in the absence of SSc or disease-specific autoantibodies. In this case, it permitted earlier detection and management of silicosis. Given the potential for progression to SSc, early identification and multidisciplinary surveillance may help prevent progression and associated morbidity.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"469-474"},"PeriodicalIF":0.7,"publicationDate":"2026-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13423326/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148629657","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
Allopurinol-Induced DRESS Syndrome in Patients with Pre-Existing Chronic Kidney Disease: A Case Report and Comprehensive Literature Review. 别嘌呤醇诱导的既往慢性肾病患者DRESS综合征:1例报告及综合文献综述
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-09 eCollection Date: 2026-01-01 DOI: 10.1159/000552298
Ashraf Ahmed, Alaa Abdelhamid, Hidaya Al-Tamimi, Raghad Elashie, Elhassan Mahmoud, Ali Ibrahim Rahil
{"title":"Allopurinol-Induced DRESS Syndrome in Patients with Pre-Existing Chronic Kidney Disease: A Case Report and Comprehensive Literature Review.","authors":"Ashraf Ahmed, Alaa Abdelhamid, Hidaya Al-Tamimi, Raghad Elashie, Elhassan Mahmoud, Ali Ibrahim Rahil","doi":"10.1159/000552298","DOIUrl":"10.1159/000552298","url":null,"abstract":"<p><strong>Introduction: </strong>Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a rare, potentially life-threatening hypersensitivity reaction that can be triggered by allopurinol, with increased risk in patients with chronic kidney disease (CKD). Its variable presentation and delayed onset pose diagnostic and therapeutic challenges.</p><p><strong>Case presentation: </strong>We describe a 76-year-old female with stage III CKD and multiple comorbidities who developed fever, diffuse erythematous scaly rash, eosinophilia, and acute kidney injury 5 weeks after initiating allopurinol. Her RegiSCAR score was 4 (probable DRESS), and the adverse drug reaction probability score was 7 (probable drug reaction). Following discontinuation of allopurinol and initiation of systemic corticosteroids, her cutaneous and renal manifestations improved, and she was successfully discharged with tapering steroids and outpatient follow-up.</p><p><strong>Conclusion: </strong>Allopurinol-induced DRESS in CKD patients is associated with high morbidity and mortality. Early recognition, drug withdrawal, and timely immunosuppressive therapy are crucial for favorable outcomes. Genetic screening and careful dose adjustment in high-risk populations may reduce incidence.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"426-437"},"PeriodicalIF":0.7,"publicationDate":"2026-06-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13399953/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148583662","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
Post-Infectious Purpura Fulminans in a Healthy Child: A Case Report. 健康儿童感染后暴发性紫癜1例报告
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-08 eCollection Date: 2026-01-01 DOI: 10.1159/000552745
Isabel Gainza Apraiz, Laura Blanch Rius, Angela Gainza Apraiz, Ana Sánchez Díez, Rosa María Izu Belloso
{"title":"Post-Infectious Purpura Fulminans in a Healthy Child: A Case Report.","authors":"Isabel Gainza Apraiz, Laura Blanch Rius, Angela Gainza Apraiz, Ana Sánchez Díez, Rosa María Izu Belloso","doi":"10.1159/000552745","DOIUrl":"10.1159/000552745","url":null,"abstract":"<p><strong>Introduction: </strong>Purpura fulminans (PF) is a rare and life-threatening thrombohemorrhagic disorder characterized by rapidly progressive purpuric skin lesions caused by dermal microvascular thrombosis and disseminated intravascular coagulation (DIC). Post-infectious PF is an uncommon variant in children and is typically associated with transient acquired deficiencies of natural anticoagulant proteins. We present a case of severe postinfectious PF that highlights the extensive cutaneous necrosis seen in this type of PF despite the absence of systemic shock.</p><p><strong>Case presentation: </strong>A previously healthy 3-year-old girl presented with rapidly progressive painful violaceous plaques on both shins. She was afebrile and hemodynamically stable. Laboratory studies revealed DIC and severe acquired Protein S deficiency with anti-Protein S antibodies. Diagnosis of PF was made. Microbiological testing was positive for <i>Streptococcus</i> species and metapneumovirus, suggesting a post-infectious trigger. Despite early treatment with antibiotics, anticoagulation, fresh frozen plasma, and intravenous immunoglobulins, the lesions progressed to full-thickness skin necrosis requiring surgical debridement and split-thickness skin grafting. Coagulation parameters and Protein S levels normalized within 2 weeks.</p><p><strong>Conclusion: </strong>Post-infectious PF should be suspected in children presenting with rapidly progressive purpuric lesions following recent infection. Early recognition and aggressive multidisciplinary management are essential to prevent life-threatening complications and limit permanent tissue damage.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"463-468"},"PeriodicalIF":0.7,"publicationDate":"2026-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13412243/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148618405","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
Acquired Digital Arteriovenous Malformation of the Toe Showing Tendency to Clinically Regress after Skin Biopsy: A Case Report. 获得性趾指动静脉畸形在皮肤活检后表现出临床倒退的趋势:1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-08 eCollection Date: 2026-01-01 DOI: 10.1159/000552851
Yoshihito Mima, Masako Yamamoto, Ken Iozumi
{"title":"Acquired Digital Arteriovenous Malformation of the Toe Showing Tendency to Clinically Regress after Skin Biopsy: A Case Report.","authors":"Yoshihito Mima, Masako Yamamoto, Ken Iozumi","doi":"10.1159/000552851","DOIUrl":"10.1159/000552851","url":null,"abstract":"<p><strong>Introduction: </strong>Acquired digital arteriovenous malformation (ADAVM) is a rare vascular anomaly characterized by direct arteriovenous shunts, typically occurring on the digits and often associated with trauma. Toe-localized ADAVM is particularly uncommon.</p><p><strong>Case presentation: </strong>A 65-year-old man presented with a persistent dark-purple macule on the dorsal aspect of the right fifth toe for over 3 years. Dermoscopy showed hairpin vessels with a white halo. Histopathological examination revealed epidermal hyperplasia, parakeratosis, elongation of rete ridges, and dilated vascular structures composed of capillaries, veins, and arterioles extending from the subepidermal dermis into the dermal papillae. Based on these findings, the patient was diagnosed with ADAVM. Following diagnostic skin biopsy, the lesion regressed without any additional intervention.</p><p><strong>Conclusion: </strong>We report a rare case of toe-localized ADAVM showing clinical regression after skin biopsy. This case suggests that biopsy-related alterations in local hemodynamics may contribute to lesion regression and highlights the importance of considering ADAVM in the differential diagnosis of persistent dark red macules on the digits.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"449-454"},"PeriodicalIF":0.7,"publicationDate":"2026-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13412241/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148618431","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
Diagnostic Challenge in Diagnosing Atypical Pityriasis Rosea: A Case Report. 诊断非典型玫瑰糠疹的挑战:1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-08 eCollection Date: 2026-01-01 DOI: 10.1159/000552340
Madeline N Pham, Benjamin Henry Taylor Bridges, Meghan Rosenquist
{"title":"Diagnostic Challenge in Diagnosing Atypical Pityriasis Rosea: A Case Report.","authors":"Madeline N Pham, Benjamin Henry Taylor Bridges, Meghan Rosenquist","doi":"10.1159/000552340","DOIUrl":"10.1159/000552340","url":null,"abstract":"<p><strong>Introduction: </strong>Pityriasis rosea is a self-limited inflammatory skin condition most commonly associated with systemic active viral infection, particularly human herpesvirus (HHV)-6 and HHV-7. Generally appearing as a herald patch, the rash subsequently spreads, commonly in a \"Christmas tree\" pattern on the back. There are atypical variants in which pityriasis rosea can present, making diagnosis challenging. This case is unique in that pityriasis rosea was misidentified, and it was only confirmed following histopathologic examination. Due to misidentification, there was an exacerbation of disease due to inappropriate treatment.</p><p><strong>Case presentation: </strong>A forty-year-old male on allopurinol for gout presented to the emergency department for 2-week history worsening rash on his abdomen and extremities. He was advised to discontinue allopurinol and was given oral corticosteroids for a suspected drug eruption. He followed up in an outpatient clinic 3 days later because the rash had worsened. At the outpatient clinic, he was suspected to have a viral exanthem; however, punch biopsy of the skin was taken and later confirmed the diagnosis of pityriasis rosea.</p><p><strong>Conclusion: </strong>Atypical presentations of pityriasis rosea can make diagnosis challenging, resulting in misidentification. Histopathology of inflammatory skin conditions can be used in the instances of diagnostic uncertainty. Reporting atypical cases of pityriasis rosea can raise awareness and aid physicians in identifying uncommon manifestations of the disease, which can potentially prevent unnecessary treatment and their adverse outcomes.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"487-491"},"PeriodicalIF":0.7,"publicationDate":"2026-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13441278/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148677131","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
When Conventional Therapy Fails: Interleukin-36 Inhibition for Severe Hydroxychloroquine-Induced Acute Generalized Exanthematous Pustulosis in Systemic Lupus Erythematosus - A Case Report. 当常规治疗失败:白细胞介素-36抑制严重羟氯喹诱导的系统性红斑狼疮急性全身性红斑性脓疱病- 1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-08 eCollection Date: 2026-01-01 DOI: 10.1159/000552623
Lu Yang, Jiayi Ying, Xiuxiu Wang, Chengfeng Zhang, Ruiming Hu, Ning Yu
{"title":"When Conventional Therapy Fails: Interleukin-36 Inhibition for Severe Hydroxychloroquine-Induced Acute Generalized Exanthematous Pustulosis in Systemic Lupus Erythematosus - A Case Report.","authors":"Lu Yang, Jiayi Ying, Xiuxiu Wang, Chengfeng Zhang, Ruiming Hu, Ning Yu","doi":"10.1159/000552623","DOIUrl":"10.1159/000552623","url":null,"abstract":"<p><strong>Introduction: </strong>Acute generalized exanthematous pustulosis (AGEP) is a rare but severe cutaneous adverse drug reaction characterized by the sudden onset of widespread sterile pustules on an erythematous and edematous base, often accompanied by fever and neutrophilia. Over 90% of cases are drug-induced. Its management can be particularly challenging in patients with autoimmune diseases such as systemic lupus erythematosus (SLE), where cutaneous manifestations and immune dysregulation may confound clinical assessment.</p><p><strong>Case presentation: </strong>We report a case of severe hydroxychloroquine-induced AGEP in a patient with active SLE. The disease was refractory to systemic corticosteroids and further worsened after intravenous immunoglobulin infusion. Given the emerging role of interleukin (IL)-36 pathway dysregulation in pustular dermatoses, the patient was treated with spesolimab, a monoclonal antibody targeting the IL-36 receptor, resulting in rapid defervescence and near-complete resolution of pustules within days.</p><p><strong>Conclusion: </strong>This case underscores the need for prompt recognition of drug-induced AGEP in patients receiving antimalarials or other immunomodulatory agents for connective tissue diseases. Crosstalk between IL-36 signaling and neutrophil extracellular traps may amplify inflammation, linking AGEP with autoimmune pathology. IL-36 inhibition with spesolimab represents a potential rescue therapy for severe, treatment-refractory AGEP, particularly in patients with underlying autoimmune disorders where conventional therapies fail.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"455-462"},"PeriodicalIF":0.7,"publicationDate":"2026-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13412247/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148618410","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
High-Potency Depigmenting Mask Delivered via 1,927-nm Fractional Laser in Refractory Melasma: A Pilot Case Series. 通过1927纳米分数激光在难治性黄褐斑中提供高效脱色面罩:一个试点案例系列。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-08 eCollection Date: 2026-01-01 DOI: 10.1159/000552673
Jaime Piquero-Casals, Juan Francisco Mir-Bonafé, Eduardo Rozas-Muñoz, Vanesa Piquero-Casals, Daniel Morgado-Carrasco
{"title":"High-Potency Depigmenting Mask Delivered via 1,927-nm Fractional Laser in Refractory Melasma: A Pilot Case Series.","authors":"Jaime Piquero-Casals, Juan Francisco Mir-Bonafé, Eduardo Rozas-Muñoz, Vanesa Piquero-Casals, Daniel Morgado-Carrasco","doi":"10.1159/000552673","DOIUrl":"10.1159/000552673","url":null,"abstract":"<p><strong>Introduction: </strong>Melasma is a chronic, relapsing hyperpigmentation disorder and may remain refractory despite multiple topical agents and procedural approaches.</p><p><strong>Case presentation: </strong>We report a case series of six women with Fitzpatrick skin types III-V and longstanding, treatment-resistant facial melasma managed with a multimodal protocol consisting of (1) a 16-week home regimen (twice-daily depigmenting cosmeceutical serum and daily high-protection sunscreen) and (2) two sessions of low-energy, low-density 1,927-nm fractional thulium laser at 4-week intervals, immediately followed by laser-assisted delivery of a compounded depigmenting mask (hydroquinone, retinoic acid, kojic acid, triamcinolone acetonide, vitamins C and E). Clinical response was assessed using the Melasma Severity Scale (MSS), standardized photography, and patient satisfaction. At week 16, all patients improved: 4 achieved >75% clinical improvement and 2 achieved 50-75% improvement, with high satisfaction and no significant adverse events beyond transient darkening and mild desquamation. Clinical benefits remained stable 12 weeks after the final laser session.</p><p><strong>Conclusion: </strong>This case series suggests that 1,927-nm fractional thulium laser-assisted delivery of a high-potency depigmenting compound, supported by topical priming and strict photoprotection, may be a feasible option for selected patients with refractory melasma.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"438-444"},"PeriodicalIF":0.7,"publicationDate":"2026-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13399954/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148583742","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
Cholesterotic Fibrous Histiocytoma: A Rare Variant of a Common Presentation: A Case Report. 胆固醇纤维组织细胞瘤:常见表现的罕见变体:1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-06-08 eCollection Date: 2026-01-01 DOI: 10.1159/000552887
Conall James McKinney, Kris Holte, David Alderdice
{"title":"Cholesterotic Fibrous Histiocytoma: A Rare Variant of a Common Presentation: A Case Report.","authors":"Conall James McKinney, Kris Holte, David Alderdice","doi":"10.1159/000552887","DOIUrl":"10.1159/000552887","url":null,"abstract":"<p><strong>Introduction: </strong>Here, we report a case of a 48-year-old man with a longstanding firm, asymptomatic nodule on the left upper arm who presented to clinic with a 1 year history of the lesion slowly enlarging to a size of 15 mm by 13 mm and becoming faintly pigmented.</p><p><strong>Case presentation: </strong>Initial concerns about the possibility of a nodular malignant melanoma meant that the lesion was excised with a 2 mm margin; however, histology demonstrated a hyperplastic epidermis with an underlying intradermal lesion with fibrocyte-like cells with a storiform and haphazard arrangement. Occasional histiocytes were present, many of which were xanthomatous. Numerous, almost confluent cleft-like spaces representing cholesterol deposition were noted as well as peripheral entrapment of collagen bundles.</p><p><strong>Conclusion: </strong>These findings confirmed the presence of a cholesterotic fibrous histiocytoma, a very rare variant of a common dermatological presentation.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"445-448"},"PeriodicalIF":0.7,"publicationDate":"2026-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13412357/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148618384","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
Eruptive Disseminated Superficial Porokeratosis and Multiple Dermatofibromas in a Patient with Systemic Lupus Erythematosus under Belimumab Therapy: A Case Report. 爆发性弥散性浅孔角化病和多发性皮肤纤维瘤在系统性红斑狼疮患者贝利单抗治疗:1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-05-23 eCollection Date: 2026-01-01 DOI: 10.1159/000552677
Mai Endo, Ryuto Mukaiyama, Toshiyuki Yamamoto
{"title":"Eruptive Disseminated Superficial Porokeratosis and Multiple Dermatofibromas in a Patient with Systemic Lupus Erythematosus under Belimumab Therapy: A Case Report.","authors":"Mai Endo, Ryuto Mukaiyama, Toshiyuki Yamamoto","doi":"10.1159/000552677","DOIUrl":"10.1159/000552677","url":null,"abstract":"<p><strong>Introduction: </strong>There have been several reports on non-actinic disseminated superficial porokeratosis in association with autoimmune diseases, malignant disorders, or immunosuppressive/immunomodulatory therapies.</p><p><strong>Case presentation: </strong>A 36-year-old female with systemic lupus erythematosus developed eruptive disseminated superficial porokeratosis on the lower legs, which appeared and increased in number after belimumab therapy was started. Physical examination showed 40 brownish superficial round plaques with slightly elevated borders on the bilateral lower legs. Histopathology revealed a narrow stack of parakeratotic corneocytes (cornoid lamella), compatible with porokeratosis. In addition, a total of 6 brownish, firm dermal nodules were observed on the thigh and lower back, which appeared a few years after the development of porokeratosis. Histopathological examination revealed dermatofibroma.</p><p><strong>Conclusion: </strong>To the best of our knowledge, there have been no reports of multiple porokeratosis under belimumab therapy. Biologic therapy may be associated with epigenetic changes in the candidate gene variation.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"405-410"},"PeriodicalIF":0.7,"publicationDate":"2026-05-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13354288/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148419120","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 Successfully Treated Dupilumab-Associated Generalized Pustular Psoriasis in a Pediatric Patient with Atopic Dermatitis: A Case Report. Upadacitinib成功治疗儿童特应性皮炎患者的dupilumab相关性全身性脓疱性银屑病1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-05-21 eCollection Date: 2026-01-01 DOI: 10.1159/000552609
Shufen Wang, Jiaguang Su
{"title":"Upadacitinib Successfully Treated Dupilumab-Associated Generalized Pustular Psoriasis in a Pediatric Patient with Atopic Dermatitis: A Case Report.","authors":"Shufen Wang, Jiaguang Su","doi":"10.1159/000552609","DOIUrl":"10.1159/000552609","url":null,"abstract":"<p><strong>Introduction: </strong>Atopic dermatitis (AD) and psoriasis are both T-cell-mediated inflammatory skin diseases. While dupilumab effectively treats moderate-to-severe AD, paradoxical psoriasiform eruptions, including rare cases of generalized pustular psoriasis (GPP), have been reported. These reactions often show poor response to conventional therapies, presenting significant therapeutic challenges. We present a case of a 6-year-old child with AD who developed GPP following dupilumab therapy and was successfully treated with upadacitinib.</p><p><strong>Case presentation: </strong>A 6-year-old girl with a 5-year history of AD was initiated on dupilumab therapy after an inadequate response to antihistamine treatment. Although initial symptoms improved, the patient rapidly developed GPP-like skin lesions throughout the body on day 11. Despite treatment with high-dose corticosteroids and cyclosporine, the patient experienced recurrent generalized pustules and developed erythrodermic psoriasis-like manifestations. Subsequently, upadacitinib was added at a dose of 15 mg every other day, following which the skin lesions gradually improved and completely resolved within 1 month. During the subsequent 6-month follow-up period on continued medication, no recurrence of skin lesions or adverse events was reported.</p><p><strong>Conclusion: </strong>This case provides a reference for the efficacy and safety of upadacitinib in a pediatric patient with AD who developed secondary GPP following dupilumab therapy. Upadacitinib may represent a valuable therapeutic option for dupilumab-associated paradoxical psoriasis (including GPP) in AD patients. Prospective studies are needed to establish its long-term safety and efficacy in this patient population.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"395-404"},"PeriodicalIF":0.7,"publicationDate":"2026-05-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13349372/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148418935","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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