{"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":"https://doi.org/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}
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":"https://doi.org/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}
{"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}
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":"https://doi.org/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}
{"title":"Recurrent Urticaria: A Rare Cryopyrin-Associated Periodic Syndrome - Muckle-Wells Syndrome.","authors":"Richa, Siddhi Gawhale","doi":"10.1159/000544705","DOIUrl":"https://doi.org/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}
{"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}
{"title":"<i>Curvularia lunata</i>-Induced Onychomycosis in an Unlikely Host: A Case Report.","authors":"Marwa Hallal, Grace Obeid","doi":"10.1159/000543525","DOIUrl":"10.1159/000543525","url":null,"abstract":"<p><strong>Introduction: </strong>Onychomycosis, a fungal infection of the nail apparatus, presents with various manifestations, including subungual hyperkeratosis, onycholysis, and nail plate destruction. Established risk factors include trauma, advanced age, and comorbidities such as diabetes and immunosuppression. <i>Trichophyton rubrum</i>, classified as a dermatophyte, is the primary causative agent, with dermatophytes contributing to 60-70% of cases. Nondermatophyte molds (NDM) account for 30-40% of onychomycosis with Candida species being the most commonly identified, while <i>Curvularia</i> species are rarely encountered. <i>Curvularia lunata</i>, a saprobic dematiaceous mold, is an infrequent human pathogen primarily residing in soil. While human diseases caused by <i>Curvularia</i> are rare, they encompass a spectrum from skin infections to endocarditis.</p><p><strong>Case presentation: </strong>This case report details a 53-year-old male engineer presenting with left big toenail discoloration, revealing a <i>C. lunata</i>-induced onychomycosis through positive KOH preparation and culture. Given the patient's concurrent use of ticagrelor, oral terbinafine hydrochloride was chosen as a therapeutic option, taking into account potential drug-drug interactions. This approach resulted in complete resolution. The discussion emphasizes distinctions in disease severity among immunocompromised and immunocompetent patients, the potential role of trauma in <i>Curvularia</i> infections, and the importance of comprehensive diagnostic examinations.</p><p><strong>Conclusion: </strong>This case report provides insight into the challenges of accurate diagnosis, emphasizing the need for routine fungal cultures. Additionally, it discusses the complexities of treatment options, considering factors such as drug interactions and patient-specific conditions, and highlighting the importance of tailored management plans. This case is reported because of the rarity of onychomycosis caused by <i>Curvularia</i> in immunocompetent individuals.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"61-67"},"PeriodicalIF":0.9,"publicationDate":"2025-02-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11882162/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143566023","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}
Bashayr S Alhubayshi, Abdulaziz A Alnoshan, Ahmed A Alhumidi, Asem Shadid
{"title":"Bullous Lichen Planus Treated with Adalimumab: A Case Report.","authors":"Bashayr S Alhubayshi, Abdulaziz A Alnoshan, Ahmed A Alhumidi, Asem Shadid","doi":"10.1159/000543523","DOIUrl":"10.1159/000543523","url":null,"abstract":"<p><strong>Introduction: </strong>Bullous lichen planus is a rare variant of lichen planus that is characterized by vesiculobullous lesions developing in the setting of preexisting LP lesions with a burning and painful feeling. Bullous lichen planus treatment is difficult since so few examples of the condition have been documented in the literature.</p><p><strong>Case presentation: </strong>A 46-year-old female presented to our outpatient clinic with pruritic violaceous bullae involving lower extremities and oral mucosa for 8 years. After extensive investigations, a diagnosis of bullous lichen planus was made and successfully treated with adalimumab after failure of other treatment options.</p><p><strong>Conclusion: </strong>Adalimumab may represent a promising, efficacious, and safe monotherapy option for the management of bullous lichen planus. The therapeutic value of this novel off-label use should be assessed in controlled clinical trials.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"42-47"},"PeriodicalIF":0.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11835416/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143448246","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}
Andrea Malagón-Liceaga, Alejandra Juárez-Flores, Ana Lilia Ruelas-Villavicencio
{"title":"Reconstruction Techniques for the Medial Canthus and Lateral Nasal Wall: A Case-Based Learning Approach.","authors":"Andrea Malagón-Liceaga, Alejandra Juárez-Flores, Ana Lilia Ruelas-Villavicencio","doi":"10.1159/000543603","DOIUrl":"10.1159/000543603","url":null,"abstract":"<p><strong>Introduction: </strong>Reconstruction of the medial canthus and lateral nasal wall poses a significant challenge in dermatologic surgery due to the area's complex, concave, three-dimensional anatomy, and proximity to critical structures like the nasolacrimal system.</p><p><strong>Case presentation: </strong>This case-based learning approach details three different techniques on the management of extensive defects involving the medial canthus and lateral nasal wall, focusing on three different surgical techniques: the Mustardé flap, a combination of flaps with a graft, and a supraclavicular skin graft. All three approaches achieved satisfactory clinical and cosmetic outcomes.</p><p><strong>Conclusion: </strong>Successful reconstruction in this region requires a multidisciplinary approach that carefully integrates facial esthetic units while preserving the functional integrity.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"48-53"},"PeriodicalIF":0.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11839213/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143457019","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}
Aljoharah Al Saud, Reem Alsergani, Ahmed Alhumidi, Abdullah Aleisa
{"title":"Multiple Primary Cutaneous Anaplastic Large Cell Lymphoma: A Case Report.","authors":"Aljoharah Al Saud, Reem Alsergani, Ahmed Alhumidi, Abdullah Aleisa","doi":"10.1159/000543349","DOIUrl":"10.1159/000543349","url":null,"abstract":"<p><strong>Introduction: </strong>Anaplastic large cell lymphomas (ALCL) are a subtype of non-Hodgkin lymphoma categorized into systemic and cutaneous (cALCL) subtypes. cALCL are frequently characterized by their lack of ALK receptor expression, which differentiates them from their systemic counterparts. cALCLs present as solitary or grouped skin lesions. Due to their rarity, cALCLs are frequently misdiagnosed as other skin conditions.</p><p><strong>Case presentation: </strong>We report the case of a female with a 30-year history of expanding skin lesions on the upper and lower limbs, abdomen, and back. During her initial presentation, the lesions were thought to be due to an arthropod bite. However, lesion biopsy revealed ALK-negative cALCL and methicillin-resistant <i>Staphylococcus aureus.</i> A full workup excluded systemic ALCL and, therefore, confirmed the diagnosis of primary cALCL. The patient was placed on methotrexate to treat the lesions and sulfamethoxazole/trimethoprim to treat the infection.</p><p><strong>Conclusion: </strong>Our case highlights the need to consider cALCL in the differential diagnosis of patients with persistent skin lesions.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"68-73"},"PeriodicalIF":0.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11882159/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143566041","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}