Case Reports in Dermatology最新文献

Introduction: Necrobiosis lipoidica (NL) is a rare granulomatous disease with no standardized treatment guidelines.

Case presentation: 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.

Conclusion: 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.

Introduction: 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).

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Discussion: 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.

Conclusion: Nail involvement in LCH, while rare, may serve as an early clinical indicator of multisystem disease.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: Tubular apocrine adenoma (TAA) is a rare sweat gland tumor, usually presenting as a well-defined, red to brown nodule on the scalp. Generally, TAA is considered as benign tumor; however, it may be difficult to differentiate TAA from metastatic or primary carcinoma.

Case presentation: A woman in her 90s presented with a pale red nodule on her right posterior auricle for 3 years. Histology showed several tubular structures contained two layers in the dermis. These showed decapitation secretion. At the periphery of the lesion, squamoid tumor cells formed nest-like patterns. Some squamoid tumor cells exhibited multinucleation. Based on the histopathological, immunohistochemical, and molecular biology findings, the tumor was diagnosed as TAA with undefined malignant changes.

Conclusion: TAA is a rare sweat gland neoplasm. Although TAA is generally benign, it can exhibit aggressive features, making thorough pathological evaluation crucial to rule out malignancy.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: Dalbavancin and oritavancin are newer long-acting antibiotics with potent activity against gram-positive organisms, including methicillin-resistant Staphylococcus aureus (MRSA). To our knowledge, there have been no reported cases of drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome in a patient treated with dalbavancin and oritavancin.

Patient presentation: A woman in her 20s presented with a right thumb abscess and cellulitis that failed to respond to several courses of oral antibiotics, resulting in recurrent emergency room visits over 3 weeks. Approximately 1 month after the initial skin infection, magnetic resonance imaging revealed osteomyelitis of the right thumb. She was treated with a single dose of oritavancin followed by two weekly doses of dalbavancin, which successfully resolved the infection. However, she subsequently developed fever and a rash consistent with DRESS syndrome, likely triggered by oritavancin or dalbavancin. Given the prolonged half-life of these medications, she required treatment with high-dose steroids for an extended duration.

Conclusion: Dalbavancin and oritavancin are second-generation lipoglycopeptide antibiotics that provide coverage for gram-positive organisms, including MRSA. They are approved for the treatment of acute bacterial skin and skin structure infections and are used off-label for bacteremia, endocarditis, and osteomyelitis. Their prolonged half-lives - 257 h for dalbavancin and 195 h for oritavancin - allow for less frequent dosing. However, a long half-life also leads to prolonged drug exposure in the event of adverse effects. Here, we report the first case of DRESS syndrome in a patient treated with dalbavancin and oritavancin.