Case Reports in Dermatology最新文献

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Use of Lebrikizumab in Moderate-to-Severe Atopic Dermatitis in Challenging Clinical Scenarios Including Atypical Presentations, Comorbidities, and Tattooed Skin Involvement: Four Case Reports. 在具有挑战性的临床情况下使用Lebrikizumab治疗中重度特应性皮炎,包括非典型表现、合并症和纹身皮肤受累:4例报告
IF 0.7
Case Reports in Dermatology Pub Date : 2026-04-02 eCollection Date: 2026-01-01 DOI: 10.1159/000551616
Mariateresa Rossi, Natale Schettini, Eugenia Veronica Di Brizzi, Luigi Gargiulo, Anna Balato
{"title":"Use of Lebrikizumab in Moderate-to-Severe Atopic Dermatitis in Challenging Clinical Scenarios Including Atypical Presentations, Comorbidities, and Tattooed Skin Involvement: Four Case Reports.","authors":"Mariateresa Rossi, Natale Schettini, Eugenia Veronica Di Brizzi, Luigi Gargiulo, Anna Balato","doi":"10.1159/000551616","DOIUrl":"10.1159/000551616","url":null,"abstract":"<p><strong>Introduction: </strong>Lebrikizumab is a high-affinity IgG4 monoclonal antibody that selectively binds soluble interleukin (IL)-13, approved for the treatment of moderate-to-severe atopic dermatitis (AD). In this case series, we described the experiences achieved with lebrikizumab in four Italian hospitals.</p><p><strong>Case presentation: </strong>The first case described a patient with moderate-to-severe AD and cheilitis complicated by bacterial impetiginization who previously received topical and systemic therapies, obtaining only a partial benefit; the 12-week treatment with lebrikizumab achieved skin clearance and cheilitis resolution. The second case highlighted the therapeutic value of lebrikizumab in a patient with sub-erythrodermic AD and concomitant cervical intraepithelial neoplasia grade 3. The third case illustrated the use of lebrikizumab in a patient with moderate-to-severe AD and concomitant asthma, confirming lebrikizumab tolerability and safety. The fourth case described the clinical improvement gained with lebrikizumab on eczema on the tattoo in a patient with obesity and dyslipidemia.</p><p><strong>Conclusion: </strong>In these challenging cases of moderate-to-severe AD where difficult-to-treat areas were involved, or conventional systemic treatments were contraindicated, lebrikizumab was associated with a marked improvement. No adverse events were reported during the observed follow-up. The presence of concomitant asthma or comorbidities, such as obesity and dyslipidemia, did not appear to hinder treatment feasibility.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"222-233"},"PeriodicalIF":0.7,"publicationDate":"2026-04-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13171092/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147940431","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
Unexpected Herpes Zoster in a Healthy 22-Month-Old: A Pediatric Case Report. 22个月大的健康婴儿意外出现带状疱疹:一份儿科病例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-04-02 eCollection Date: 2026-01-01 DOI: 10.1159/000551851
Nouf A Almagushi, Abdulaziz H Alhussain
{"title":"Unexpected Herpes Zoster in a Healthy 22-Month-Old: A Pediatric Case Report.","authors":"Nouf A Almagushi, Abdulaziz H Alhussain","doi":"10.1159/000551851","DOIUrl":"10.1159/000551851","url":null,"abstract":"<p><strong>Introduction: </strong>Herpes zoster (HZ), caused by varicella zoster virus reactivation, is uncommon in immunocompetent children without prior varicella exposure.</p><p><strong>Case presentation: </strong>We describe a 22-month-old boy who developed HZ 4 months after varicella vaccination, presenting with a unilateral dermatomal vesicular rash. Serological testing was positive for IgM. No maternal or postnatal varicella exposure was reported, and the child had no immunodeficiency.</p><p><strong>Conclusion: </strong>This case underscores the rare possibility of HZ in vaccinated young children lacking traditional risk factors, highlighting the need for clinical vigilance even in atypical populations. Further investigation into triggers of early VZV reactivation postvaccination is warranted.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"317-321"},"PeriodicalIF":0.7,"publicationDate":"2026-04-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13293594/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148326999","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
Case Report of Giant Myxoid Fibrosarcomatous Dermatofibrosarcoma Protuberans Compressing the Right Jugular Vein: A Multidisciplinary Approach to Complex Surgical Management. 压迫右颈静脉的巨大黏液样纤维肉瘤性隆突皮肤纤维肉瘤病例报告:复杂手术治疗的多学科方法。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-03-30 eCollection Date: 2026-01-01 DOI: 10.1159/000551789
Zhumei Wei, Chaocheng Liu, Andy Lee, Grace Xiong, Nawar Tarafdar, Dilys Chen, Xiaohong Chen
{"title":"Case Report of Giant Myxoid Fibrosarcomatous Dermatofibrosarcoma Protuberans Compressing the Right Jugular Vein: A Multidisciplinary Approach to Complex Surgical Management.","authors":"Zhumei Wei, Chaocheng Liu, Andy Lee, Grace Xiong, Nawar Tarafdar, Dilys Chen, Xiaohong Chen","doi":"10.1159/000551789","DOIUrl":"10.1159/000551789","url":null,"abstract":"<p><strong>Introduction: </strong>Dermatofibrosarcoma protuberans (DFSP) is a locally aggressive low-grade cutaneous neoplasm with high cure rates following complete surgical resection. However, 5-15% of classic DFSP cases undergo fibrosarcomatous transformation, resulting in fibrosarcomatous DFSP (FS-DFSP), a more aggressive variant with significantly higher rates of local recurrence, metastasis, and mortality. This case presents a rare myxoid variant of FS-DFSP with complex anatomical involvement requiring multidisciplinary management.</p><p><strong>Case presentation: </strong>A 56-year-old East Asian female presented with a giant recurrent chest mass measuring 15 × 12 × 6 cm, initially misdiagnosed as fibroma 19 years prior. Advanced imaging revealed an 8 × 13 × 12 cm poorly defined soft tissue mass involving the pectoralis major and extending to the sternocleidomastoid muscles, with compression of the proximal right jugular vein. Histopathological examination confirmed FS-DFSP with myxoid differentiation, characterized by diffuse spindle cell infiltration in a fascicular pattern. Immunohistochemistry revealed CD34+, EMA+, and actin+ staining with negative desmin, S100, and STAT-6. COL1A1-FISH analysis demonstrated COL1A1/PDGFB fusion in 8% of cells. Management included tumor vascular embolization, surgical resection with 3 cm margins, skin grafting, local flap repair, and adjuvant radiation therapy due to anatomical constraints and residual tumor involvement of the anterior sternum. Four-year follow-up shows no evidence of recurrence.</p><p><strong>Conclusion: </strong>This case highlights the myxoid variant of FS-DFSP, emphasizing the importance of comprehensive histopathological evaluation, multidisciplinary treatment planning, and long-term surveillance. The successful management with combined surgical and radiation therapy demonstrates the need for individualized treatment approaches in complex anatomical locations. Four-year remission demonstrated the efficacy of comprehensive multimodal therapy in preventing local recurrence in FS-DFSP.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"248-255"},"PeriodicalIF":0.7,"publicationDate":"2026-03-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13215662/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148052434","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
Scanning Acoustic Microscopy as a Novel Quantitative Tool for Assessing Dermal Sclerosis in Systemic Sclerosis: A Case Report. 扫描声学显微镜作为评估系统性硬化症中皮肤硬化的一种新的定量工具:一个病例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-03-20 eCollection Date: 2026-01-01 DOI: 10.1159/000550671
Kyoko Kanno, Satomi Igawa, Masaru Honma, Yuki Ogura, Tomonobu Ezure, Akemi Ishida-Yamamoto
{"title":"Scanning Acoustic Microscopy as a Novel Quantitative Tool for Assessing Dermal Sclerosis in Systemic Sclerosis: A Case Report.","authors":"Kyoko Kanno, Satomi Igawa, Masaru Honma, Yuki Ogura, Tomonobu Ezure, Akemi Ishida-Yamamoto","doi":"10.1159/000550671","DOIUrl":"10.1159/000550671","url":null,"abstract":"<p><strong>Introduction: </strong>Systemic sclerosis (SSc) is a chronic autoimmune disease characterized by skin fibrosis and internal organ involvement. Quantitative methods for assessing skin sclerosis beyond the modified Rodnan skin score (mRSS) are required to better monitor disease progression and treatment response.</p><p><strong>Case presentation: </strong>A 61-year-old woman with rapidly progressing SSc and early interstitial lung disease was treated with IVCY. Skin biopsies were obtained before and after treatment. Scanning acoustic microscopy (SAM) was used to measure the speed of sound within the skin tissue, reflecting tissue stiffness. A histological analysis was performed. Following IVCY, SAM significantly decrease the speed of sound within the affected skin tissue, preceding a measurable reduction in mRSS. Histological analysis showed improvements in collagen bundle architecture and reduced inflammatory cell infiltration.</p><p><strong>Conclusion: </strong>SAM may be a promising, objective, and quantitative technique for evaluating skin sclerosis in patients with SSc and for monitoring early therapeutic responses. This is an exploratory observation based on a single case. Further investigations in larger, controlled clinical trials are warranted to determine its clinical utility and predictive value.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"215-221"},"PeriodicalIF":0.7,"publicationDate":"2026-03-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13132523/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147811356","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
Squamous Cell Carcinoma of the External Auditory Canal in a Patient with Netherton Syndrome. 内瑟顿综合征1例外耳道鳞状细胞癌。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-03-20 eCollection Date: 2026-01-01 DOI: 10.1159/000551088
Misaki Hayashi, Takumi Kumai, Hisataka Ominato, Ryosuke Sato, Risa Wakisaka, Ryusuke Hayashi, Michihisa Kono, Hidekiyo Yamaki, Kenzo Ohara, Kan Kishibe, Miki Takahara
{"title":"Squamous Cell Carcinoma of the External Auditory Canal in a Patient with Netherton Syndrome.","authors":"Misaki Hayashi, Takumi Kumai, Hisataka Ominato, Ryosuke Sato, Risa Wakisaka, Ryusuke Hayashi, Michihisa Kono, Hidekiyo Yamaki, Kenzo Ohara, Kan Kishibe, Miki Takahara","doi":"10.1159/000551088","DOIUrl":"10.1159/000551088","url":null,"abstract":"<p><strong>Introduction: </strong>Netherton syndrome is a rare autosomal recessive disorder characterized by congenital ichthyosis, hair shaft abnormalities, atopic diathesis, and a broad spectrum of immunologic dysfunction. Although cutaneous malignancies are uncommon, affected individuals may develop early-onset tumors that can pose diagnostic and therapeutic challenges.</p><p><strong>Case presentation: </strong>We report the case of a 33-year-old woman with Netherton syndrome who presented with persistent left otorrhea and a granulomatous mass in the external auditory canal. Imaging demonstrated a soft-tissue lesion with focal bony erosion and suspected dural involvement, accompanied by fluorodeoxyglucose uptake in a cervical lymph node. Biopsy confirmed squamous cell carcinoma (SCC), staged as T4N1M0. The patient underwent superselective intra-arterial cisplatin infusion combined with radiotherapy, resulting in complete remission of both the primary lesion and nodal metastasis, with no evidence of recurrence over 4 years. Including this case, 9 patients with Netherton syndrome and cutaneous malignancies have been reported, with tumors frequently arising at a young age and showing variable HPV status. Interpretation of SCC antigen may be complicated by elevations associated with atopic dermatitis.</p><p><strong>Conclusion: </strong>This case highlights the elevated risk of cutaneous malignancies in Netherton syndrome and underscores the need for long-term surveillance and multidisciplinary care.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"234-241"},"PeriodicalIF":0.7,"publicationDate":"2026-03-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13175543/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147962221","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 Cellular Digital Fibroma on the Lateral Aspect of the Left Foot: A Case Report. 左脚侧面细胞性指状纤维瘤1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-03-16 eCollection Date: 2026-01-01 DOI: 10.1159/000551558
Yuuki Arai, Satoshi Nakamura
{"title":"A Case of Cellular Digital Fibroma on the Lateral Aspect of the Left Foot: A Case Report.","authors":"Yuuki Arai, Satoshi Nakamura","doi":"10.1159/000551558","DOIUrl":"10.1159/000551558","url":null,"abstract":"<p><strong>Introduction: </strong>Cellular digital fibroma (CDF) is a rare, benign CD34-positive fibrous tumor that typically arises on acral sites, most commonly the fingers and toes. Clinically, it may resemble acquired digital fibrokeratoma, digital fibroma, periungual fibroma, supernumerary digit, or verruca vulgaris, and histopathologically, it can be confused with other CD34-positive acral tumors, particularly superficial acral fibromyxoma. Therefore, accurate diagnosis requires careful clinicopathological and immunohistochemical evaluation.</p><p><strong>Case presentation: </strong>A 48-year-old woman presented with an asymptomatic, well-circumscribed, keratotic nodule measuring 5 mm in diameter on the lateral margin of the left foot, an uncommon location for CDF. Histopathological examination revealed compact orthokeratosis with an epidermal collarette and a dermal proliferation of uniformly slender spindle cells arranged in short interweaving fascicles. No cytologic atypia, mitotic figures, or myxoid stroma were observed. Immunohistochemically, the tumor cells were diffusely positive for CD34 and negative for α-smooth muscle actin, epithelial membrane antigen, S100 protein, and CD99. Based on these findings, a diagnosis of CDF was established. The lesion was completely excised, and no recurrence was observed during a 2-year follow-up period.</p><p><strong>Conclusion: </strong>Although rare, particularly at uncommon sites such as the lateral foot, CDF should be included in the differential diagnosis of small keratotic acral tumors. Awareness of its characteristic clinicopathological and immunohistochemical features is essential to distinguish CDF from both clinically similar benign lesions and other CD34-positive acral neoplasms, thereby avoiding unnecessary overtreatment.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"264-270"},"PeriodicalIF":0.7,"publicationDate":"2026-03-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13245965/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148210201","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
Nonpigmented Adenoid Subtype of Basal Cell Carcinoma: A Case Report. 非色素腺样亚型基底细胞癌1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-03-11 eCollection Date: 2026-01-01 DOI: 10.1159/000551351
Yasunori Ichimura, Shiro Niiyama, Akiko Hirata
{"title":"Nonpigmented Adenoid Subtype of Basal Cell Carcinoma: A Case Report.","authors":"Yasunori Ichimura, Shiro Niiyama, Akiko Hirata","doi":"10.1159/000551351","DOIUrl":"10.1159/000551351","url":null,"abstract":"<p><strong>Introduction: </strong>Basal cell carcinoma (BCC) is the most common skin cancer, with a rising incidence globally. We encountered a patient with the nonpigmented adenoid subtype of BCC. The nonpigmented subtype of BCC accounts for only a small percentage of cases of BCC in the Asian population, with the adenoid subtype of BCC also being rare in all human ethnicities.</p><p><strong>Case presentation: </strong>An 82-year-old woman presented to us with a growth in the inner canthus of the left eye, which had been slowly evolving for about 1 year. The lesion had failed to respond to a variety of topical treatments. Physical examination revealed a reddish plaque in the inner canthus of the left eye that measured 10 × 6 mm in size. Dermoscopic evaluation showed blue-gray globules and arborizing telangiectasia. Histopathological examination confirmed the diagnosis of adenoid BCC.</p><p><strong>Conclusion: </strong>We analyzed the data of patients with the adenoid subtype of BCC in the Japanese population and found the possibility that this variant of BCC was often nonpigmented. Furthermore, we collected the dermoscopic findings of patients with the adenoid subtype of BCC; however, the data revealed no other noteworthy characteristics at present. We expect that further characterization of these two variants of BCC through accumulation of data from clinical cases will contribute to facilitating early diagnosis.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"242-247"},"PeriodicalIF":0.7,"publicationDate":"2026-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13189552/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147986705","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
Adalimumab and Tofacitinib Combination Therapy for Pyoderma Gangrenosum: A Case Report. 阿达木单抗与托法替尼联合治疗坏疽性脓皮病1例。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-03-06 eCollection Date: 2026-01-01 DOI: 10.1159/000550618
Jingjing Yang, Shunlong Zhou, Chao Liu
{"title":"Adalimumab and Tofacitinib Combination Therapy for Pyoderma Gangrenosum: A Case Report.","authors":"Jingjing Yang, Shunlong Zhou, Chao Liu","doi":"10.1159/000550618","DOIUrl":"10.1159/000550618","url":null,"abstract":"<p><strong>Introduction: </strong>Pyoderma gangrenosum (PG) is a rare inflammatory skin disease causing progressive necrotizing ulcers with limited treatment options due to adverse effects and high recurrence rates.</p><p><strong>Case presentation: </strong>We present a case of a 70-year-old elderly patient diagnosed as PG, which was unresponsive to initial prednisolone treatment. After approximately 3 weeks of an individualized treatment regimen of adalimumab combined with tofacitinib, disease progression was halted, accompanied by a significant reduction in ulcer size, reduced exudation, and substantial pain relief. Subsequent tapering and discontinuation of prednisolone, followed by 6 weeks of combined adalimumab and tofacitinib and 8 weeks of tofacitinib monotherapy, resulted in complete ulcer healing by week 17. No recurrence of inflammatory activity was observed during the follow-up period of over 7 months.</p><p><strong>Conclusion: </strong>This case highlights the successful and rapid efficacy of adalimumab and tofacitinib combination therapy for PG, offering a novel strategic reference for individualized treatment, particularly in recurrent or cases unresponsive to initial treatment.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"177-181"},"PeriodicalIF":0.7,"publicationDate":"2026-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13061404/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147644006","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
Stevens-Johnson Syndrome following a Single Ampicillin Dose in an Elderly Patient: A Case Report. 老年患者单次氨苄西林后史蒂文斯-约翰逊综合征1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-02-27 eCollection Date: 2026-01-01 DOI: 10.1159/000550570
A'sem Mohammad Abedalqader Qannas, Syed Iftiqar Ahmed, Ketevan Machavariani, Nabil Tariq, Rana Hussain Abdul Rehman, Nino Abuladze, Ahmed Abdelkader
{"title":"Stevens-Johnson Syndrome following a Single Ampicillin Dose in an Elderly Patient: A Case Report.","authors":"A'sem Mohammad Abedalqader Qannas, Syed Iftiqar Ahmed, Ketevan Machavariani, Nabil Tariq, Rana Hussain Abdul Rehman, Nino Abuladze, Ahmed Abdelkader","doi":"10.1159/000550570","DOIUrl":"10.1159/000550570","url":null,"abstract":"<p><strong>Introduction: </strong>Stevens-Johnson syndrome (SJS) is a rare but life-threatening mucocutaneous disorder, most commonly triggered by medications.</p><p><strong>Case presentation: </strong>We present the case of a 75-year-old Armenian female who developed severe SJS following a single dose of ampicillin. She initially presented with fever, generalized weakness, and a rapidly progressing bullous rash affecting her face, neck, upper limbs, and chest. Her medical history included untreated rheumatoid arthritis and chronic bronchitis, for which she had been self-medicating with corticosteroids and other medications. Despite early initiation of high-dose corticosteroids and supportive therapy, her condition deteriorated, leading to septicemia and respiratory failure, necessitating admission to the Infectious Diseases Department. Multidisciplinary management, including immunosuppressive therapy with cyclosporine and targeted antimicrobial treatment, resulted in clinical stabilization and eventual recovery.</p><p><strong>Conclusion: </strong>This case highlights that even minimal exposure to beta-lactam antibiotics can precipitate severe SJS in elderly and immunocompromised patients. Early recognition and prompt multidisciplinary intervention are essential to improve outcomes.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"184-191"},"PeriodicalIF":0.7,"publicationDate":"2026-02-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13068386/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147670177","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
Ocular Manifestations Leading to the Diagnosis of Pseudoxanthoma Elasticum with a Novel Heterozygous ABCC6 Mutation: A Case Report. 具有新型杂合ABCC6突变的弹性假黄瘤的眼部表现:1例报告。
IF 0.7
Case Reports in Dermatology Pub Date : 2026-02-27 eCollection Date: 2026-01-01 DOI: 10.1159/000550862
Akihiro Fukuta, Yohei Iwata, Rena Yasuda, Akira Iwanaga, Hiroyuki Murota, Kazumitsu Sugiura
{"title":"Ocular Manifestations Leading to the Diagnosis of Pseudoxanthoma Elasticum with a Novel Heterozygous ABCC6 Mutation: A Case Report.","authors":"Akihiro Fukuta, Yohei Iwata, Rena Yasuda, Akira Iwanaga, Hiroyuki Murota, Kazumitsu Sugiura","doi":"10.1159/000550862","DOIUrl":"10.1159/000550862","url":null,"abstract":"<p><strong>Introduction: </strong>Pseudoxanthoma elasticum (PXE) is a progressive hereditary disease caused by the calcification and degeneration of elastic fibers, affecting the skin, retina, blood vessels, and gastrointestinal tracts. The gene responsible is the ATP-binding cassette subfamily C member 6 (<i>ABCC6</i>).</p><p><strong>Case presentation: </strong>A 47-year-old man presented to an ophthalmologist with a right eyelid contusion. Fundus examination revealed right macular hemorrhage and bilateral angioid streaks. PXE was suspected due to skin lesions around the neck and abdomen; he was referred to our dermatology department. Histopathological examination revealed degenerated anucleate elastic fibers with calcification in the mid-dermis. Genetic testing revealed a previously unreported heterozygous <i>ABCC6</i> mutation. We established a diagnosis of PXE based on characteristic skin lesions and histopathological findings triggered by ocular manifestations. Based on in silico analysis, the <i>ABCC6</i> gene mutation was predicted to be pathogenic.</p><p><strong>Conclusion: </strong>In some PXE cases, the <i>ABCC6</i> gene mutation can be undetectable or only one mutation is detected; PXE could be diagnosed based on clinical and histopathological symptoms. When characteristic skin and eye lesions are observed, appropriate evaluation is required. Genetic testing facilitates early diagnosis and the discovery of novel mutations, contributing to a deeper understanding of the disease spectrum.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"18 1","pages":"192-198"},"PeriodicalIF":0.7,"publicationDate":"2026-02-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13075883/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147688583","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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