Case Reports in Dermatology最新文献

Psoriasis is a common chronic skin disease mainly located in areas of friction. Psoriasis of the lips as an exclusive presentation is rare and often misdiagnosed. Different anti-psoriatic therapies have been proposed, but the literature is limited to case studies with partial results. Biologic therapies have revolutionized the management of many dermatologic conditions, including psoriasis, and they are approved for pediatric use. We report the case of a 14-year-old boy with a 2-year history of white-yellowish scaling lesions on his lips, without intraoral involvement. Lip biopsy showed a psoriasiform pattern. Treatment with adalimumab 40 mg every other week was started, and after 6 months of therapy, we obtained a complete remission of the patient's lip psoriasis.

Linear IgA disease (LAD) is a rare autoimmune bullous disease characterized by IgA deposition in the basement membrane zone (BMZ). A 66-year-old male was treated for myelodysplastic syndrome at our hospital for 5 years, during which his condition remained stable. He visited our department because of erythema with itching, which appeared 1 year ago and gradually exacerbated with the development of blisters and erosions. During the first visit, multiple erythemas with erosions and crusts on their periphery were observed on the trunk and lower limbs. Histopathological examination revealed subepidermal blisters with inflammatory cell infiltration, mainly constituting of neutrophils, eosinophils, and lymphocytes. Direct and indirect immunofluorescence showed linear IgA deposits in the BMZ and IgA anti-BMZ antibodies, respectively, while immunoblotting using a concentrated culture supernatant of HaCaT cells detected IgA antibodies reactive to 120-kDa LAD-1. Accordingly, the patient was diagnosed with lamina lucida-type LAD. Subsequent colonoscopy revealed multiple colorectal polyps and rectal adenocarcinoma (Tis, N0, and M0). Multigene panel test showed an ATM variant of unknown significance but did not detect any pathogenic variants associated with intestinal polyposis syndrome. The skin lesions quickly resolved with oral diaphenylsulfone 50 mg/day and resection of the colorectal polyps and adenocarcinoma. To our knowledge, this is the first reported case of LAD associated with multiple colorectal polyps and rectal adenocarcinoma. Additionally, we also analyzed reported cases of LAD associated with malignancy from the literature.

Reticular erythematous mucinosis is a rare and persistent form of primary idiopathic mucinosis, often referred to as plaque-like cutaneous mucinosis or midline mucinosis. It presents with reticulate patches or erythematous plaques with predilection for the anterior and posterior trunk. Affected patients are frequently asymptomatic. Pruritus or burning sensations were reported after exposure to the sun. The aetiology remains obscure; its pathogenesis is poorly understood, particularly in immunocompromised patients such as HIV-infected patients. The disease associations are not uniformly documented. Antimalarial agents significantly improve and shorten the course of the disease. We report a case of a 31-year-old African woman with underlying HIV infection who displayed the classical clinical and histological features of reticular erythematous mucinosis. This condition is rare among the HIV-infected patients, particularly in those of African descent, in whom lichen myxoedematosus/scleromyxoedema variants and acral persistent papular mucinoses were most frequently reported. The higher incidence of photosensitivity in HIV-infected individuals including the patients with skin of colour may play a potential role in reticular erythematous mucinosis. Its relationship with lupus erythematosus and photosensitivity in the context of HIV infection is discussed. To the best of our knowledge, this is the first reported case of reticular erythematous mucinosis in an African HIV-infected patient. This case highlights the need for diagnostic awareness in cases presenting with erythematous plaques and patches in a net-like pattern developing on the midline and sun-exposed areas of the trunk.

The large cell transformation of mycosis fungoides (MF-LCT) is a phenomenon observed in the advanced stages of mycosis fungoides (MF), which is the most common primary cutaneous lymphoma. The diagnostic criteria of MF-LCT are a minimum of 25% of large cells or a formation of microscopic nodules of them in the histological examination of skin samples. The clinical outcomes for MF-LCT are poor, as less than 20% of patients survive 5 years after diagnosis, but the expression of the CD30 antigen is generally considered to be associated with a better prognosis. We present a case of a patient with the diagnosis of MF with LCT, with an ulcerated tumor lesion approximately 30 × 20 cm in size on the right lateral abdominal wall. Brentuximab vedotin (BV) treatment was started due to the presence of the CD30 antigen, with a quick and impressive regression of the cutaneous lesion and tumor mass and good treatment tolerance. After follow-up of 20 months, patient remains in complete remission. A schedule of treatment for MF-LCT is directed mainly by the clinical stage of the disease and the comorbidities; the more severe clinical course of the disease requires systemic treatment. If at least 5% of the cells found in the skin lesions biopsy sample express the CD30 antigen, a beneficial effect of BV treatment could be expected. It may seem that the use of BV is one of the optimal therapeutic options in patients with advanced MF-LCT showing expression of CD30.

Oral and laryngeal cutaneous T-cell lymphoma (CTCL) is rare and usually associated with poor prognosis. Here, we discuss 2 cases of oral CTCL that developed in heavily pretreated patients and provide a review of the literature. The first case is of a 46-year-old African American male with rapidly progressive disease, presenting with a lesion on his hard palate 6 months after being diagnosed with a CD4+CD8+ CTCL. His cutaneous disease was widespread with tumors on >80% of his body surface area. Unfortunately, the patient died 2 ½ years after his CTCL diagnosis and 7 months after developing the oral CTCL lesion. The second case is of a 38-year-old African American male with stage IIb CD3+CD4+CD30+ mycosis fungoides (MF), who developed a tumor on the hard palate 6 months after diagnosis. He received palliative radiation to the oral lesion and multiple lines of systemic therapy for pulmonary, laryngeal, esophageal, and gastric involvement. Biopsy of the gastric lesions showed a CD30+ T-cell lymphoma with the same clonal peak as in his skin but with large cell transformation. Brentuximab vendoin was started, and the patient is now in complete remission, 30 months later. From the 76 cases of oral CTCL that have been reported in the English language, six were of transformed MF. The most common sites affected were the tongue and palate, and the most common presentation were erythematous or ulcerated tumors, plaques, or nodules associated with dysphagia and pain. Oral CTCL typically occurs years after the initial diagnosis of CTCL and portend a poor prognosis with an average survival of just over 1 year after development of oral lesions.

Neonatal lupus erythematosus (NLE) is a rare autoimmune disorder of newborns and infants, born to usually asymptomatic mothers with lupus erythematosus. Clinical manifestations include variable cutaneous findings, with possible cardiac or hepatic involvement. We present a case of a 3-month-old baby girl with NLE, born to an asymptomatic mother. Her atypical clinical presentation included hypopigmented atrophic scars on the temples. She improved with topical pimecrolimus cream, with almost complete resolution of the facial lesions and improvement in atrophy noted at the 4-month follow-up visit. Cutaneous findings of hypopigmentation and atrophic scarring are less commonly reported. To our knowledge, no similar cases have been published in the Middle East. We aim to share this interesting case, highlight the different clinical presentations of NLE and raise awareness among physicians about this variable phenotype of NLE for timely diagnosis of this uncommon entity.

Granulomatous pigmented purpuric dermatosis (GPPD) is a rare histologic variant of pigmented purpuric dermatosis (PPD) characterized by dermal histiocyte-rich interstitial infiltration with or without granuloma formation in addition to the other typical features of PPD. GPPD was previously observed more frequently to affect Asians and was reported to be associated with dyslipidemia. However, our literature search of 45 documented GPPD cases revealed an increasing prevalence in Caucasians in addition to dyslipidemia and associated autoimmune diseases. To date, etiopathogenesis of GPPD is unknown but may involve dyslipidemia, genetic and immunological factors such as autoimmune dysregulation or a sarcoidal reaction associated with C. acnes. GPPD is usually persistent and recalcitrant to treatments. We report a case of GPPD in a 57-year-old Thai woman with underlying myasthenia gravis who presented with a pruritic eruption on both lower legs. After treatment with 0.05% clobetasol propionate cream and oral colchicine, the lesion improved with marked flattening and disappeared with residual postinflammatory hyperpigmentation. We provide a literature review of the epidemiology, etiopathogenesis, concomitant comorbidities, clinical symptoms, dermatoscopic features, and treatments of GPPD.

Adverse reactions in tattooed skin during magnetic resonance imaging (MRI) are rare but well known. Previous reports describe sudden burning pain in tattooed skin, sometimes accompanied by mild erythema and oedema when entering MRI scanners. The pathophysiology remains unclear, but simple direct thermal heating can be excluded. It has been hypothesized that MRI-triggered torque and traction create neural sensations from magnetic pigment particles. However, this case enlightens yet another possible mechanism. We present a 35-year-old woman experiencing reoccurring stinging sensations in three decorative black tattoos just seconds after the initiation of the MRI. Single-blind tests with handheld power magnets or a dummy could reproduce painful subjective feelings in her tattooed skin. Similar events were provoked during re-evaluation with MRI. Surprisingly, chemical analyses and electron microscopy of skin samples revealed carbon black as the colouring agent - no iron-based solids were detected. Our case demonstrates that MRI tattoo reactions are not limited to magnetic contaminants alone. More distinct subgroups of MRI-induced reactions may occur. We hypothesize that radiofrequency induction of surface currents in black carbon particles adjacent to sensory axons in the dermis may lead to neurosensations.

Lichen planus pigmentosus (LPP) is a distinctive variant of lichen planus described for the first time in 1974. A wide range of presentations and distribution patterns have been reported, mainly presenting as dark brown hyperpigmented macules and patches, typically involving sun-exposed areas such as the face, neck, and flexural folds. It is more common in dark-skinned patients with a female predominance. Infrequently, LPP may present as a Blaschkoid distribution, with only a few cases reported in the medical literature. We report an unusual case of LPP that developed with a unilateral Blaschkoid distribution on the left side of the trunk in a 32-year-old female following weight reduction surgery.

An acral fibrochondromyxoid tumor is a newly described type of benign soft tissue neoplasm that presents as a single nodular lesion on a finger or toe. There has only been one previous report on this tumor, a case series that described the initial pathologic and clinical findings; however, details on clinical history, physical examination, and outcome are unknown. In this report, we describe a case of a 39-year-old male who presented with a painful enlarging mass involving the distal right 3rd finger and hyponychium. Punch biopsy was performed and the lesion was identified as an acral fibrochondromyxoid tumor on microscopic examination. X-ray showed no bony involvement. The tumor was successfully excised with complete resolution of pain symptoms. We discuss the clinical features and immunohistochemistry findings of our case in the context of the current limited knowledge about this very rare tumor.