以皮肤水疱为主要表现的血管肉瘤1例

报告1例以皮肤水疱为主要表现的血管肉瘤。患者男,78岁,面部、额部、头皮紫红色肿物、水疱5月余,经组织病理学检查确诊为血管肉瘤,免疫组化检查显示CD31( )、CD34(血管 )。     

Marjolin's Ulcer Presenting with In-Transit Metastases: A Case Report and Literature Review

Marjolin''s ulcer is an aggressive cutaneous malignancy common in previously traumatized or chronically inflamed skin. It has high regional metastasis and fatality rates. Our patient presented with subcutaneous nodules and ulcerations on the right limb. He had a history of osteomyelitis of the fifth toe. Histopathological examination of the nodule and ulceration demonstrated squamous cell carcinoma. The nodules and ulcerations were in-transit metastases of Marjolin''s ulcer. Here, we present a case of squamous cell carcinoma arising at a site of a chronic osteomyelitis with resultant in-transit metastases.     

Congenital Smooth Muscle Hamartoma Presenting as a Linear Atrophic Plaque: Case Report and Review of the Literature

Abstract: Congenital smooth muscle hamartoma usually manifests as a well-circumscribed, hyperpigmented plaque, frequently hypertrichotic, on the trunk or extremities. We report such a lesion in a 7-month-old girl that presented as a linear, mottled, purplish red plaque appearing In areas to be atrophic, involving her right buttock, posterior thigh and leg, and fifth toe. Although the clinical appearance suggested linear morphea, a biopsy specimen had numerous haphazardly oriented bundles of smooth muscle in the reticular dermis. Masson trichrome staining, smooth muscle specific actin, and electron microscopic studies confirmed the smooth muscle nature of the cells. A diagnosis of smooth muscle hamartoma was made. To our knowledge, this linear clinical presentation has not been described previously.     

Neonatal Acute Hemorrhagic Edema of Childhood: Case Report and Review of the English-Language Literature

Acute hemorrhagic edema of childhood is an unusual form of leukocytoclastic vasculitis previously reported in children age 4 months to 3 years. The etiology remains unknown, however, many authors describe the process as an immune-mediated vasculitis in response to a variety of antigenic stimuli. We report a case of congenital acute hemorrhagic edema of childhood associated with maternal gastroenteritis six weeks before delivery.     

Subacute Cutaneous Lupus Erythematosus Presenting in Childhood: A Case Report and Review of the Literature

A 2‐year‐old African American, Hispanic boy presented with well‐defined, violaceous, annular dermal plaques without scale over the upper extremities, face, lower extremities, and buttocks. The clinical presentation and laboratory studies were consistent with a diagnosis of subacute cutaneous lupus erythematous (SCLE). SCLE presenting in childhood is exceedingly rare, with only eight cases previously reported. It is important to clinically differentiate SCLE from other eruptions more common to children, such as atopic dermatitis, urticarial drug eruptions, and psoriasis vulgaris, because progression to systemic lupus erythematous (SLE) may occur. SLE needs to be closely followed. We present the first case (to our knowledge) of SCLE in a child of African American or Hispanic descent and provide a table of other documented pediatric presentations of SCLE for comparison.     

Alopecia Neoplastica due to Gastric Adenocarcinoma Metastasis to the Scalp,Presenting as Alopecia: A Case Report and Literature Review

Alopecia neoplastica is defined as hair loss secondary to a visceral malignancy that has metastasized to the scalp. The scalp is a relatively common site of cutaneous metastasis, usually presenting as a single or multiple firm scalp nodules. Alopecia neoplastica is a well-recognized but rare presentation, and its pathogenesis is incompletely understood. Atrophy of the hair follicles due to tumor invasion of the scalp plays a role in the development of alopecia. Herein, we describe a 33-year-old woman with gastric adenocarcinoma who developed alopecia neoplastica while receiving cancer chemotherapy. Scalp biopsy revealed metastatic adenocarcinoma cells interspersed between collagen bundles and around hair follicles. Immunohistochemical analysis indicated that the tumor cells originated from the primary gastric adenocarcinoma. Therefore, she was diagnosed with alopecia neoplastica due to gastric adenocarcinoma. The findings from this report may be helpful for understanding the mechanism of alopecia neoplastica.     

Wegener's Granulomatosis: Case Report and Literature Review

Abstract: Wegener's granulomatosis (WG) is a necrotizing granulomatous vasculitis usually affecting the upper and lower respiratory tracts and kidneys. Any organ system can be affected by the pathologic process, which remains an etiologic enigma. Limited forms of the disease are recognized in which few extrapulmonary and no renal lesions occur. Cutaneous manifestations occur in 40% to 50% of patients with WG (1). Early diagnosis is imperative because treatment regimens reduce morbidity and mortality in this potentially fatal disease. We report WG in a 10-year- old boy whose upper respiratory tract symptoms began at age 8 years. The case is illustrative of the difficulties that can be encountered in attempting to make this diagnosis. Skin is an easily accessible organ for biopsy, thus an increasing familiarity with the typical biopsy specimen findings within a consistent clinical setting may aid in earlier diagnosis of WG.     

表现为血疱的持久性隆起性红斑1例

患者男,70岁。双手、足、肘、膝出现红斑、血疱6个月。皮损组织病理示:表皮下水疱,疱内可见大量嗜中性粒细胞,真皮内可见弥漫的嗜中性粒细胞浸润,血管管壁及管周可见纤维素样渗出及核碎裂。诊断:持久性隆起性红斑。临床医师应警惕以血疱为表现的持久性隆起性红斑。     

Lichen Nitidus Presenting with Nail Changes—Case Report and Review of the Literature

Lichen nitidus of the nail is rare and can precede the onset of skin lesions. Delayed diagnosis is common. We present an unusual case of lichen nitidus–associated nail changes that preceded the onset of skin lesions in a 4‐year‐old Indian girl. We also conduct a review of six other cases of lichen nitidus with nail involvement from the English‐language literature. Clues to the diagnosis of lichen nitidus include violaceous or pigmentary changes of the nail fold and subtle lichenoid papules on the affected digits. Lichen nitidus of the nails appears to be less severe than nail changes of lichen planus and is generally self‐limiting. Understanding the natural history of lichen nitidus of the nails will help physicians better counsel patients and their families.     

Benign Cephalic Histiocytosis: Case Report and Literature Review

Abstract: A 3-year-old boy had maculopapules on his face and neck since age 6 months. These were yellow-brown, asymptomatic, and clinically similar to flat warts. Histopathologic study revealed a fibrohistiocytic infiltrate in the superficial dermis. Ultrastructurally, comma-shaped bodies, desmosome-like junctions, and coated vesicles were seen; there were no lipid droplets or Birbeck granules. With these data, a diagnosis of benign cephalic histiocytosis was made. Twenty-five cases are reported in the literature: 17 males and 8 females (male:female ratio 2:1). Sixteen patients had lesions on parts of the body other than the head, neck, and shoulders.     

Congenital Erythrodermic Psoriasis: Case Report and Literature Review

Abstract: A 4-year-old girl was seen in our department for erythroderma, palmoplantar hyperkeratosis, and scalp desquamation present since birth. The dermatosis had run an intermittent course, with exacerbations after Infections and spontaneous remissions. A specimen from a skin biopsy performed at 1 year of age showed the characteristic features of psoriasis, findings that were confirmed in our biopsy specimen. Treatment with acltretin controlled the outbreaks. At 7 years of age she has developed, for the first time, plaque type psoriasis. Congenital erythroderma is an unusual form of psoriasis with a wide differential diagnosis.     

Cutaneous Sparganosis: A Case Report and Literature Review

A sixty‐nine‐year‐old male patient, without a significant prior medical history, presented with a rapidly enlarging, 2 cm non‐tender nodule on the right lower eyelid. The lesion persisted in spite of an incision and drainage. Following an excision, the histology revealed a diffuse infiltrate of atypical mononuclear cells within the dermis. The cells were large, monomorphic, with irregular to convoluted nuclei, prominent nucleoli, amphophilic cytoplasm. They were very mitotically active. Immunohistochemical stains for CD45, CD3, and CD30 were strongly positive. Pancytokeratin, CD20, and ALK (Anaplastic Lymphoma Kinase fusion protein) were negative. The histopathologic diagnosis was CD30 (Ki‐1) positive anaplastic large cell lymphoma. The margin was positive for involvement by lymphoma. The lesion was re‐excised with no evidence of residual involvement by lymphoma. A primary cutaneous anaplastic large cell lymphoma was favored over systemic involvement based on the clinical presentation of a single eyelid nodule and ALK negativity. He was referred to a hematology‐oncologist for further management. Anaplastic large cell lymphoma is a distinctive type of malignant lymphoma with a relatively favorable prognosis. It frequently involves the skin, however, to the best of our knowledge, presentation of this lymphoma type as a single lesion on the eyelid is extremely rare.     

Congenital Miliaria Crystallina: Case Report and Literature Review

Generalized congenital miliaria crystallina occurred in a black newborn boy. Although miliaria crystallina occurring in infancy and beyond is well established, congenital occurrence is very rare. The pathogenesis of the disorder is not well understood. We discuss some hypotheses of pathogenesis in the context of our patient, as well as a differential diagnosis and a comparison with a previously reported case. Miliaria crystallina should be considered in the differential diagnosis of vesiculobullous eruptions in newborns.     

Clear Cell Papulosis: Case Report and Literature Review

Abstract: Clear cell papulosis is a newly described skin disease characterized by multiple white papules. Histopathologically, diagnostic clear cells were seen among the basal cells of the epidermis. We report clear cell papulosis on the lumbar area and buttocks of a 1-year-old girl.     

Imipramine-Induced Facial Pigmentation: Case Report and Literature Review

Background

Patients who present with facial pigmentation can be a diagnostic challenge. ObjectiveThe goal of this study was to discuss the diagnosis and management of imipramine-induced facial pigmentation.

Methods

We describe a patient with facial pigmentation of 26 years’ duration that was associated with imipramine treatment for depression. We discuss light and election microscopic findings and review 11 previously reported cases of imipramine-induced skin pigmentation.

Results

Examination showed blue–gray facial pigmentation. Light microscopy showed perivascular pigment granule deposits in the upper dermis that stained positively with Fontana–Masson stain and negatively with Prussian blue stain. Electron microscopy showed electron-dense bodies within histiocytes without clearly identifiable melanin granules, consistent with drug-induced pigmentation. Six weeks after switching to sertraline the patient reported a slight improvement of her cutaneous pigmentation.

Conclusion

Imipramine is a rare cause of gray–blue facial pigmentation. Light microscopy consistently shows granular dermal deposits that stain positively with Fontana–Masson stain but negatively with iron stain.     

Pediatric Sweet Syndrome: Case Report and Literature Review

Abstract:   We report on an 18-month-old boy with a typical presentation of Sweet syndrome following an upper respiratory tract illness. No evidence of hematologic malignancy was found and he was successfully treated with oral prednisolone. A comprehensive literature review has summarized the features of pediatric Sweet syndrome from 66 reported cases. Cases occurring under the age of 3 years had a male predominance and no association with malignancy, whereas cases over the age of 3 years had an equal sex distribution and were strongly associated with myeloid line hematologic malignancies. Identified complications were serious with an overall mortality of 9% rising to 40% with cardiovascular involvement.     

Agminated Lentiginosis: Case Report and Review of the Literature

Abstract: Agminated lentiglnosis (AL) is characterized by numerous lentfgines confined to a body segment, with a sharp demarcation at the midline. So far, only 13 cases have been reported in the literature. We report a 30-year-old woman with a multifocal AL The cutaneous lesions first appeared at 5 years of age and gradually increased in number with time. Clinically, they appeared as numerous brown macules, ranging in size from 1 to 5 mm in diameter, in a peppered distribution over an area extending bilaterally on the neck, chin, and cheeks. On the trunk the lesions were localized to the left shoulder and breast, involving the axilla and upper part of the abdomen down to the umbilicus. Four cafe au lait macules ranging in diameter from 10 to 15 mm were present, with no evidence of neuroflbromas. Histopathology of a macute showed the features of lentigo; in addition, groupings of melanocytes were observed at the dermoepidermal junction. The differential diagnosis of AL includes speckled lentiginous nevus and segmental neurofibromatosis.     

Erythrokeratoderma Variabilis: Case Report and Review of the Literature

Abstract: A 5-month-old boy with erythrokeratoderma variabilis is presented. The parents noted that the asymptomatic erythematous plaques changed over the course of hours to days, Topical therapy with retinoic acid proved ineffectlve. A brief review of the literature is presented.     

Congenital Dermatofibrosarcoma Protuberans: A Case Report and Literature Review

Congenital dermatofibrosarcoma protuberans (DFSP) is an extremely rare skin tumor that is commonly misdiagnosed, or is often diagnosed long after the initial presentation. Although many cases of DFSP are diagnosed in adulthood, there are some differences between adult DFSP and congenital DFSP. We report a case of congenital DFSP that was initially misdiagnosed as a simple vascular lesion. The delay in diagnosis led to the considerable growth of the lesion, such that a huge scar was left after the surgical treatment. The major differences between adult and congenital DFSP are discussed through a literature review. Clinicians should be aware of the characteristics of congenital DFSP, to reduce misdiagnosis and the delay of diagnosis from the initial presentation.     

Nivolumab-Induced Alopecia Areata: A Case Report and Literature Review

Nivolumab (anti-PD-1) currently used in many cancers. With the usage of nivolumab increased, many cutaneous side effects were reported including maculopapular rash, lichenoid reactions, vitiligo, bullous disorders, psoriasis exacerbation, and alopecia areata (AA). Here, we report AA after nivolumab for treatment of hepatocellular carcinomas (HCC). A 55-year-old male presented with multiple hairless patch from 1 month ago. He suffered HCC and treated with nivolumab for 6 months after hepatectomy. He treated for hair loss with triamcinolone intra-lesional injection without improvement. We performed skin biopsy on the scalp. Histopathologic findings revealed decreased of hair follicles on the horizontal section with lymphocyte infiltration on the perifollicular area on the vertical section. Clinicopathologic findings were agreed with AA. Considering lack of previous history of AA and hairless patches with 6 months after nivolumab injection, we diagnosed him as nivolumab induced AA. Treatment included topical steroid, and minoxidil. No regrowth of hair was noted after 4 months of follow-up. Nivolimumab induced AA is rare side effect. Pathogenesis of nivolumab induced AA remain unclear. But our case is likely related to nivolumab, known to induce immune related adverse events, and given in the delay of a few months between introduction and the occurrence of the hair loss. Here, we reports nivolmumab induced AA; rare side effect.