Case Reports in Dermatology最新文献

Introduction: Phytophotodermatitis is a cutaneous reaction resulting from skin contact with certain plant-derived phototoxic compounds, followed by ultraviolet exposure. Lime-induced phytophotodermatitis, sometimes called "lime disease" or "margarita photodermatitis," is one of the most common forms, caused by furocoumarins in lime juice or peel. Despite being well described, the condition remains underdiagnosed and is frequently mistaken for allergic dermatitis, burns, cellulitis, or even abuse, particularly when occurring in atypical linear or patterned distributions.

Case presentation: We report a case of lime-induced phytophotodermatitis in a 25-year-old woman who developed a striking, linear, hyperpigmented lesion on her forearm after spilling a mojito cocktail on her skin during prolonged sun exposure.

Conclusion: This case underscores the importance of recognizing the condition in the context of leisure and vacation settings, where patients may overlook or fail to report relevant exposures, and highlights the role of patient education to prevent recurrence.

Introduction: Pansclerotic morphea is a rare and debilitating variant of localized scleroderma characterized by extensive skin sclerosis. Conventional therapies may be ineffective, particularly in patients with comorbidities that limit treatment options, prompting the need for alternative modalities.

Case presentation: A 29-year-old African American male with autoimmune hepatitis presented with progressive sclerotic plaques on the trunk and extremities, accompanied by nonhealing leg ulcers. Biopsy and clinical findings confirmed pansclerotic morphea. Prior treatments, including topical and systemic corticosteroids, were unsuccessful, and methotrexate was contraindicated. Given the refractory nature of the disease, UVA-1 phototherapy was initiated as salvage therapy.

Conclusion: After 6 months of UVA-1 phototherapy, the patient showed marked clinical improvement, including softening of skin induration and complete wound healing, with no adverse effects. This case underscores the value of UVA-1 phototherapy as a safe and effective treatment for severe morphea, especially in patients unable to receive standard immunosuppressive therapies.

Introduction: Pyodermatitis-pyostomatitis vegetans (PDV-PSV) is a rare and chronic mucocutaneous inflammatory disorder strongly associated with inflammatory bowel disease, particularly ulcerative colitis (UC). It typically presents with exudative plaques that may affect the face, scalp, and intertriginous areas and also pustular lesions on the oral mucosa. Although several cases with cutaneous and mucosal involvement have been reported, eyelid involvement remains uncommon and sparsely reported in the literature.

Case presentation: We report a 28-year-old male patient recently diagnosed with UC who presented multiple asymptomatic vegetating pustular plaques in the bilateral groin area and multiple pustules on both upper and lower gingiva, lips and hard palate. In a rare manifestation of PDV-PSV, both upper and lower eyelids developed pustules and ulcers, predominantly affecting the left eye. Empirical antimicrobial therapies were initiated under the suspicion of an infectious etiology; however, no clinical improvement was observed. Histopathological evaluation revealed intraepidermal eosinophilic abscess with mild acantholysis and suprabasal clefting, and direct immunofluorescence study was negative supporting the diagnosis of PDV-PSV in the context of UC. The patient was successfully treated with Azathioporine and Adalimumab with significant improvement in gastrointestinal symptoms and mucocutaneous lesions after 3 months of therapy.

Conclusion: This case highlights the importance of considering PDV-PSV in UC patients who developed atypical mucocutaneous manifestations, especially when standard treatment regimen for supposed infectious etiologies fail. Although uncommon, eyelid involvement should be considered as a potential manifestation of PDV-PSV. Histopathological evaluation and direct immunofluorescence study remains essential for diagnosis, especially in cases presenting with unusual manifestations. Consideration of PDV-PSV as a mucocutaneous manifestation of UC is essential to avoid misdiagnoses and prevent unnecessary treatments. While periocular involvement may be rare, the presence of typical skin and oral lesions, along with distinctive histological and immunofluorescence findings, should guide clinicians toward the correct diagnosis.

Introduction: Cutaneous squamous cell carcinoma (cSCC) is one of the most common skin cancers, with an increasing incidence worldwide. Chronic sun exposure is a major risk factor and lesions are usually observed on sun-exposed areas such as the face, neck, scalp, and hands. Imiquimod cream, an immune response modifier, has shown promising results in the management of selected cases of cSCC.

Case report: A 70-year-old, Filipino male presented with 3-year history of a slow growing erythematous plaque on the abdomen. Physical examination revealed a solitary, well-defined, erythematous plaque measuring 5 cm on the left lower quadrant of the abdomen. Histopathologic examination confirmed cSCC. The patient declined surgery and opted for topical treatment with imiquimod 5% cream, applied over 24 weeks for a total of 104 applications. Serial biopsies taken at multiple follow-up points and anatomic sites confirmed complete histologic clearance.

Conclusion: This case highlights one of the few documented instances of histologic cure in a >5 cm cSCC treated solely with imiquimod cream. It underscores the potential of imiquimod as a nonsurgical alternative and expands current treatment for patients who may not be suitable candidates for conventional surgical excision.

Introduction: VEXAS (vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic) is a myeloid-driven, adult-onset inflammatory syndrome associated with hematological neoplasms and a wide range of manifestations, including fever, pancytopenia, vasculitis, polyarthritis, and pulmonary infiltrates. Skin lesions are present in up to 90% of cases but remain heterogeneously defined. We present 3 patients with VEXAS syndrome and cutaneous manifestations admitted to a tertiary referral hospital.

Case presentations: The 3 male patients with a median age of 66 years showed chronic muco-cutaneous xanthomatous lesions, persistent neutrophilic dermatosis en plaques, as well as eczematous lesions, respectively. Histology showed either dermal neutrophilic infiltrate, leukocytoclasia without vasculitis with abundant interstitial foamy CD68-positive macrophages, superficial and deep neutrophilic dermatitis and panniculitis, or spongiotic dermatitis. Somatic mutations in UBA1 were detected in all 3 patients and 2 also had the same UBA1 mutation in the skin.

Conclusion: VEXAS syndrome is associated with several cutaneous manifestations, the features of which may be atypical and pose a diagnostic and classification challenge. Histology often shows neutrophilic dermatosis with leukocytoclasia without frank leukocytoclastic vasculitis and an occasional accumulation of foamy macrophages in the superficial and deep dermis. The cutaneous lesions likely reflect either the activation of autoinflammatory pathways or the presence of myelodysplastic cells in the skin. They may represent either "non-specific" reactive findings or "specific" cutaneous manifestations associated with clonal infiltration of abnormal myeloid cells.

Introduction: Generalized pustular psoriasis (GPP) is a rare and potentially life-threatening form of psoriasis, characterized by widespread sterile pustules and systemic symptoms. While the pathogenesis remains incompletely understood, the interleukin 36 (IL-36) pathway has emerged as a key contributor. Spesolimab, a humanized monoclonal antibody targeting the IL-36 receptor, has shown rapid and marked clinical improvement in patients during acute GPP flares.

Case presentation: We report three cases of GPP flares treated with spesolimab in Denmark, administered prior to its commercial availability through a compassionate use program. All patients presented with severe GPP and were assessed using the Physician's Global Assessment for Generalized Pustular Psoriasis. Treatment with spesolimab resulted in rapid improvement across all cases.

Conclusion: These real-world observations support the clinical efficacy of spesolimab for managing acute GPP flares and demonstrate its potential as a therapeutic option in severe cases, even before commercial access.

Introduction: Hidradenitis suppurativa (HS) is a chronic inflammatory condition of the hair follicles in apocrine gland-rich areas, characterized by painful nodules, abscesses, sinus tracts, and fibrosis. Treatment aims to reduce inflammation, control symptoms, and prevent progression, with TNF-α inhibitors like adalimumab being effective for moderate-to-severe cases.

Case presentation: This report presents a 26-year-old female with recurrent HS lesions in the intermammary fold, treated with adalimumab and intralesional corticosteroids for a keloid-like lesion. While systemic therapy improved inflammatory lesions, the keloid-like lesion progressed, prompting biopsy to exclude malignancy. Histopathology revealed benign fibromatous fibrosis, highlighting the diagnostic challenges of HS-associated lesions.

Conclusion: This case underscores the efficacy of TNF-α inhibitors, the complexity of managing concomitant keloids, and the need for vigilant monitoring and histological evaluation to differentiate benign from malignant conditions. A multidisciplinary approach is essential for optimal HS management, particularly in patients with atypical or treatment-resistant lesions.

Introduction: Facial discoid dermatosis (FDD) is a rare dermatological condition, first described in 2010. Its aetiology remains unknown and it is distinguished by its resistance to several treatments, both topical and systemic. The lack of documentation in the medical literature makes this disease a significant diagnostic and therapeutic challenge.

Case presentation: A 51-year-old woman consulted for the appearance of pruritic skin lesions on the face, neck and upper chest of 2 months' duration. She underwent various topical and systemic therapies without complete resolution over a period of 9 months. Histological examination revealed psoriasiform irregular epidermal hyperplasia, mild spongiosis, parakeratosis, and hypogranulosis. In the superficial dermis, perivascular lymphocytic infiltrates were observed. Given the findings described and the refractoriness to multiple treatments employed, the diagnosis of FDD was established. Low-dose isotretinoin administration led to complete resolution of the skin lesions, with no associated adverse effects observed during the course of treatment.

Conclusion: We present the first reported case of FDD in a middle-aged woman who was successfully treated with low-dose isotretinoin, resulting in complete resolution of the condition. This finding underlines the potential of isotretinoin as an effective and well-tolerated therapeutic option, especially in women of childbearing age. We also provide a comprehensive review of the treatments for FDD that have been documented in the literature.

Introduction: Disseminated herpes zoster (HZ) is uncommon and typically occurs in immunocompromised populations, including patients with HIV/AIDS, transplantation recipients, and patients with autoimmune diseases. However, disseminated HZ may also occur in healthy patients. Many studies have found that its incidence is positively correlated with age, and the severity is also positively correlated with age. Central nervous system (CNS) complications during or following HZ are extremely rare. Encephalitis has been reported to affect only 0.1%-0.2% of HZ patients. The occurrence of disseminated HZ with acute encephalitis in immunocompetent patients is even less frequently reported.

Case presentation: An 85-year-old male presented with disseminated HZ and acute encephalopathy, later confirmed as varicella zoster virus (VZV) encephalitis via cerebrospinal fluid analysis and neuroimaging. Treatment with intravenous acyclovir and methylprednisolone led to gradual improvement in consciousness, and the skin lesions regressed, though dizziness persisted.

Conclusion: Elderly patients with disseminated HZ should be closely monitored for potential neurological complications such as HZ encephalitis. Once clinical manifestations such as fever, headache, meningeal irritation, cranial nerve impairment, or even cognitive changes appear, the possibility of CNS infection should be suspected.

Introduction: Lichen planopilaris (LPP) is a well-known cicatricial alopecia with a frontal fibrosing variant. LPP has a prevalence rate of 13.4 per 100,000 persons. It is understood that LPP favors the female populations. What is not commonly known is that LPP can be preceded by other signs and symptoms such as pili torti.

Case report: We present a case report of a 70-year-old female whose initial flare of LPP was preceded by a flare of pili torti. This patient's case is also complicated by her history of ectodermal dysplasia. She experienced progressive itching, shedding, and mid-scalp hair loss along with brittle curls which failed to improve with intralesional steroids and minoxidil, which prompted further investigation.

Conclusion: This case highlights how early intervention and detection can allow for treatment and prevention of further hair loss in patients with LPP and as well as other alopecias. Treatment of patients at onset of disease is crucial to prevent permanent hair loss.