{"title":"Childhood Linear IgA Dermatosis Successfully Treated with the Combination of Dapsone and Sulfasalazine.","authors":"Ashley S Kim, Olav Sundnes","doi":"10.1159/000546155","DOIUrl":"10.1159/000546155","url":null,"abstract":"<p><strong>Introduction: </strong>Linear IgA dermatosis (LAD) is a rare subepidermal autoimmune blistering skin disorder characterized by the linear deposition of IgA along the basal membrane. It affects primarily young children and adults, and is the most common bullous disease in the paediatric population. Dapsone represents the mainstay of treatment, to which the majority of patients show excellent initial responses with long-term remission. In recalcitrant cases, sulfonamides (sulfapyridine, sulfasalazine, sulfamethoxypyridazine) are considered second-line options either as monotherapy or in conjunction with dapsone. Most published cases of sulfonamides in childhood LAD report use of sulfapyridine or sulfamethoxypyridazine, with no published reports on sulfasalazine use in young children.</p><p><strong>Case presentation: </strong>We present a case of a 1-year-old child with confirmed LAD who did not respond adequately to dapsone alone. Sulfapyridine is not available in Norway, while sulfasalazine is accessible and considered a safe option for other autoimmune disorders. The addition of sulfasalazine resulted in rapid complete remission.</p><p><strong>Conclusion: </strong>This case thus supports sulfasalazine as a pragmatic, accessible alternative to sulfapyridine as the second-line treatment in childhood LAD.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"185-190"},"PeriodicalIF":0.9,"publicationDate":"2025-05-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12140607/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144233274","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}
Martin Gschnell, Iris Hennighausen, Can Alpagut, Julia Nadina Föhr, Jacqueline Kussini, Lisa Krönig
{"title":"Therapy-Resistant Erosions in the Genital Area of Elderly Patients: A Differential Diagnostic Challenge - A Case Report.","authors":"Martin Gschnell, Iris Hennighausen, Can Alpagut, Julia Nadina Föhr, Jacqueline Kussini, Lisa Krönig","doi":"10.1159/000546135","DOIUrl":"10.1159/000546135","url":null,"abstract":"<p><strong>Introduction: </strong>Genital erosions occur in many dermatological conditions and can sometimes be difficult to classify, especially in older patients with multiple morbidities.</p><p><strong>Case presentation: </strong>We report the case of an 81-year-old patient with therapy-resistant genital erythema. Under topical antifungal therapy for a suspected diagnosis of Candida intrigue, the scrotal findings continued to progress, accompanied by genital erosions and new erosions on the nose and right knee. With evidence of IgG and C3 deposition on the basement membrane on direct immunofluorescence and subepidermal cleft formation on histology, a diagnosis of bullous pemphigoid with predominant genital involvement was made.</p><p><strong>Conclusion: </strong>In conclusion, this case study illustrates that skin biopsies should be taken in cases of therapy-resistant erythema with erosions in order to clarify the dignity and ultimately to be able to adapt the treatment. In addition, it is important to examine the entire integument, including the mucous membranes close to the skin, to avoid overlooking groundbreaking findings. Bullous pemphigoid is the most common blistering autoimmune skin disease and occurs mainly in older people. However, the classic picture of bulging blisters is not always present. Bullous pemphigoid may masquerade clinically as pruritic eczema, erythroderma, or erosions. Predominant genital involvement has only been described in a few case reports.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"191-195"},"PeriodicalIF":0.9,"publicationDate":"2025-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12148320/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144257429","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}
{"title":"Successful Treatment of Necrobiosis Lipoidica with Abrocitinib: A Case Report.","authors":"Yuping Zhang, Yirong Li, Fengjiao Meng, Chen Li","doi":"10.1159/000546134","DOIUrl":"10.1159/000546134","url":null,"abstract":"<p><strong>Introduction: </strong>Necrobiosis lipoidica (NL) is a rare granulomatous disease with no standardized treatment guidelines.</p><p><strong>Case presentation: </strong>We present a case of a patient who experienced ulcerative NL of the left leg for 5 years, during which traditional treatments proved ineffective. The patient was administered oral abrocitinib at a dosage of 100 mg per day. After 2 weeks of treatment, the patient's skin lesions ceased exuding and began to show improvement. By the 10-week mark, the patient's symptoms had significantly improved, with the ulcer healing and no new lesions appearing. No adverse events were reported during the 4-month follow-up period.</p><p><strong>Conclusion: </strong>Abrocitinib may represent a rapid and safe treatment option for patients with refractory localized NL. Further studies are warranted to confirm these findings and evaluate long-term efficacy.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"181-184"},"PeriodicalIF":0.9,"publicationDate":"2025-04-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12140610/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144233275","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}
{"title":"Kasabach-Merritt Phenomenon: Diagnosis, Management, and Outcome on Two Cases in a Sub-Saharan Country (Senegal).","authors":"Khadim Diop, Mame Téné Ndiaye Diop, Ibrahima Diop, Ines Mejri, Lana Yassine, Fatou Diassé, Fatime Tall, Idrissa Demba Ba, Maodo Ndiaye, Ousmane Ndiaye","doi":"10.1159/000546020","DOIUrl":"10.1159/000546020","url":null,"abstract":"<p><strong>Introduction: </strong>Kasabach-Merritt phenomenon (KMP) is a rare and life-threatening disease, characterized by the profound thrombocytopenia and consumptive coagulopathy associated with vascular tumors. In sub-Saharan Africa, KMP-related data are scarce and it poses significant challenges in management, particularly due to limited availability of treatment resources. We report 2 cases of KMP observed in a sub-Saharan Africa country (Dakar, Senegal).</p><p><strong>Case presentation: </strong>Case 1: a 45-day-old male infant was admitted for a tumor lesion located in the axillary fold that has been evolving since birth. The lesion had rapidly become aggressive, inflammatory, and purpuric. Blood tests showed normocytic normochromic anemia with severe thrombocytopenia and high D-dimer levels. The diagnosis of a kaposiform hemangioendothelioma complicated by a KMP was retained. Treatment with betamethasone was initiated, but death occurred 6 days later secondary to cerebral hemorrhage. Case 2: a 2-month-old female infant was admitted for a rapidly aggressive, inflammatory, infiltrating tumor lesion on the face that had been progressing for 1 month. Blood tests showed normocytic normochromic anemia with severe thrombocytopenia and high D-dimer levels. The diagnosis of a KMP was retained. Treatment with corticosteroids, then combined with vincristine, was administered. The outcome was favorable with a considerable regression of the mass and improvement in biological parameters after 6 months.</p><p><strong>Conclusion: </strong>To our knowledge, we report the first case report on KMP in sub-Saharan Africa, particularly in pediatric dermatology in Dakar. Besides their rarity, these cases highlight the challenges in the management of KMP in a country with limited therapeutic resources.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"174-180"},"PeriodicalIF":0.9,"publicationDate":"2025-04-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12119071/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144172721","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}
Xiaofei Gao, Bin Yu, Jiaotian Huang, Haixia Yang, Zhu Wei
{"title":"Extensive Nail Changes Are a Possible Clue Indicating Multisystem Involvement in Childhood Langerhans Cell Histiocytosis: A Case Report and Literature Review.","authors":"Xiaofei Gao, Bin Yu, Jiaotian Huang, Haixia Yang, Zhu Wei","doi":"10.1159/000545567","DOIUrl":"10.1159/000545567","url":null,"abstract":"<p><strong>Introduction: </strong>Langerhans cell histiocytosis (LCH) is a rare myeloid neoplasm that can involve nearly any organ, leading to multisystem damage. Nail involvement in LCH is particularly uncommon. Here we report a case of a young boy with multisystem LCH initially presenting with nail changes.</p><p><strong>Case presentation: </strong>We described a boy who presented with a 2-year history of asymptomatic changes characterized by onycholysis, subungual hyperkeratosis, and purpuric striae affecting most fingernails and toenails, initially attributed to onychomycosis. Two years later, he developed multisystem involvement affecting the pituitary gland, lungs, skin, liver, and spleen. The patient succumbed shortly after histopathological confirmation via skin biopsy due to massive gastrointestinal hemorrhage secondary to cirrhosis-induced portal hypertension.</p><p><strong>Discussion: </strong>Nail lesions may serve as the initial manifestation of LCH, often preceding other characteristic disease symptoms. This early presentation provides critical diagnostic opportunities for timely intervention. Consideration of LCH, biopsy, and comprehensive evaluation of organ involvement is essential to reduce the rate of misdiagnosis and the potential for unrecognized high-risk disease.</p><p><strong>Conclusion: </strong>Nail involvement in LCH, while rare, may serve as an early clinical indicator of multisystem disease.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"165-173"},"PeriodicalIF":0.9,"publicationDate":"2025-04-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12097764/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144126845","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}
Saman Al-Zahawi, Sara Masoomi, Alireza Ghanadan, Yasaman Sadeghi, Maryam Daneshpazhooh, Hamidreza Mahmoudi
{"title":"A Rare Case of Primary Cutaneous CD4+ Small/Medium Size T-Cell Lymphoproliferative Disorder Responding to Rituximab.","authors":"Saman Al-Zahawi, Sara Masoomi, Alireza Ghanadan, Yasaman Sadeghi, Maryam Daneshpazhooh, Hamidreza Mahmoudi","doi":"10.1159/000545747","DOIUrl":"https://doi.org/10.1159/000545747","url":null,"abstract":"<p><strong>Introduction: </strong>Primary cutaneous CD4+ small/medium T-cell lymphoproliferative disorder is a rare lymphoproliferative disorder (PCS-TCLPD) characterized by a solitary erythematous plaque or nodule on the face, neck, or upper trunk with a favorable outcome. Very rarely, multiple plaques or masses may be seen in which precise evaluation should be performed to exclude other primary cutaneous lymphoma.</p><p><strong>Case presentation: </strong>Here we report a rare case of recalcitrant primary cutaneous small-/medium-sized CD4+ lymphoproliferative disorder in a 55-year-old male patient who responded well to rituximab injection but had recurrence after 1 year from the injections.</p><p><strong>Conclusion: </strong>Aggressive treatment is avoided in solitary lesions of PCS-TCLPD, but multiple lesions with no response to the conventional modalities may need more sophisticated management, including anti-CD20 rituximab.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"150-156"},"PeriodicalIF":0.9,"publicationDate":"2025-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12064157/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143968940","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}
{"title":"Acquired Reactive Perforating Collagenosis Complicated by Diabetes Mellitus and Hypertension: A Case Report.","authors":"Meng Zhang, Ruiqi Chu, Junping Shen, Chunmei Liu, Yanan Wang, Shun Zhang, Xiangxiang Ren","doi":"10.1159/000545693","DOIUrl":"https://doi.org/10.1159/000545693","url":null,"abstract":"<p><strong>Introduction: </strong>Acquired reactive perforating collagenosis (ARPC) is a rare cutaneous disorder frequently associated with systemic diseases such as diabetes mellitus and chronic kidney disease. Its diagnosis remains challenging due to clinical overlap with other perforating dermatoses, necessitating advanced diagnostic tools for confirmation.</p><p><strong>Case presentation: </strong>A 55-year-old male with a 15-year history of diabetes mellitus and hypertension developed progressively pruritic papules and nodules on both lower limbs over 2 months. Initial misdiagnosis as a fungal infection led to unsuccessful antifungal therapy. Dermatoscopy revealed keratotic plugs with peripheral erythema, while histopathology demonstrated transepidermal elimination of degenerated collagen, confirming ARPC. Topical retinoic acid cream achieved marked symptomatic relief.</p><p><strong>Conclusion: </strong>This case highlights ARPC's strong association with metabolic comorbidities and underscores the necessity of dermatoscopic and histopathological evaluation to avoid diagnostic pitfalls. The therapeutic success of topical retinoids in resource-constrained settings reinforces their role as a pragmatic first-line intervention. Clinicians should consider ARPC in patients with chronic pruritic eruptions and systemic metabolic disorders, advocating for multidisciplinary collaboration to address underlying comorbidities.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"143-149"},"PeriodicalIF":0.9,"publicationDate":"2025-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12061362/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143962332","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}
Yoshihito Mima, Masako Yamamoto, Naoki Nishida, Yuta Norimatsu, Ken Iozumi
{"title":"Trauma-Induced Psoriatic Arthritis: A Deep Köbner Phenomenon.","authors":"Yoshihito Mima, Masako Yamamoto, Naoki Nishida, Yuta Norimatsu, Ken Iozumi","doi":"10.1159/000545694","DOIUrl":"https://doi.org/10.1159/000545694","url":null,"abstract":"<p><strong>Introduction: </strong>Psoriasis is a chronic systemic inflammatory disorder resulting from complex interactions between genetic and environmental factors. In addition to cutaneous manifestations, psoriasis is associated with psoriatic arthritis (PsA). Among the recognized mechanisms of disease exacerbation, the Köbner phenomenon (KP) plays a crucial role. KP refers to the appearance of new psoriatic lesions on previously healthy skin following trauma. A deeper variant of this phenomenon, the deep Köbner phenomenon (DKP), involves severe trauma triggering systemic inflammation, including the onset of PsA. Trauma-induced DKP represents unique disease mechanisms, linking external mechanical stress to immune-mediated joint pathology.</p><p><strong>Case presentation: </strong>We present a case of a 60-year-old male with psoriasis, who initially responded well to ixekizumab, an IL-17A inhibitor but subsequently developed PsA following a wrist injury. The PsA exacerbation was attributed to increased mechanical stress from daily activities and rehabilitation efforts. Adjustments were made to rehabilitation intensity and joint movement restrictions, leading to gradual symptom improvement over 3 months without intensifying biological therapy.</p><p><strong>Conclusion: </strong>This case highlights the pathophysiological relationship between trauma, DKP, and PsA. The rapid onset of PsA following trauma suggests that inflammatory mediators and neuropeptides triggered by DKP play significant roles. Given that rehabilitation plays crucial roles in recovery but may also exacerbate symptoms if improperly managed, tailored rehabilitation strategies are essential in managing trauma-induced PsA. This case also underscores the importance of comprehensive management, including biological therapy and personalized rehabilitation approaches. Further studies are needed to optimize inflammation control and joint function in patients experiencing post-trauma PsA.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"137-142"},"PeriodicalIF":0.9,"publicationDate":"2025-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12061363/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143978903","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}
Carrie A Forman, Noelle Desir, Michael M Ong, Shari R Lipner
{"title":"Congenital Malalignment of the Great Toenails with Secondary Retronychia: A Tale of Twin Toenails.","authors":"Carrie A Forman, Noelle Desir, Michael M Ong, Shari R Lipner","doi":"10.1159/000545462","DOIUrl":"https://doi.org/10.1159/000545462","url":null,"abstract":"<p><strong>Introduction: </strong>Congenital malalignment of the great toenail (CMGT) is characterized by deviation of the nail plate due to rotation of the nail matrix. CMGT may be complicated by secondary retronychia.</p><p><strong>Case presentation: </strong>We report a case of twin 14-year-old females who presented with CMGT with secondary retronychia with no clear inciting event. Physical examination demonstrated lateral deviation of the great toenails with yellowed, thickened, and layered nail plates, consistent with a diagnosis of CMGT with secondary retronychia. The patients were advised to make relevant lifestyle changes and treated with clobetasol proprionate 0.05% ointment to use on all toenails daily for 3 months, with 1 week off per month, under occlusion.</p><p><strong>Conclusion: </strong>We hypothesize that both genetic and environmental factors likely contributed to the development of CMGT with secondary retronychia in these twins. Increased awareness and recognition of this underdiagnosed condition, particularly in the pediatric population, is important for timely and effective management.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"157-164"},"PeriodicalIF":0.9,"publicationDate":"2025-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12077864/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144076237","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}
{"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}