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A case of adult-onset urticaria pigmentosa
( Seuk Hoon Moon ),( Hyeree Kim ),( Hei Sung Kim ),( Sang Hyun Cho ),( Jeong Deuk Lee ) 대한피부과학회 2015 대한피부과학회 학술발표대회집 Vol.67 No.1
Mastocytosis is a neoplastic disease characterized by the accumulation of mast cells in various tissues. Two main variants of the disease are recognized by the WHO: cutaneous mastocytosis and systemic mastocytosis. Cutaneous mastocytosis typically presents as urticaria pigmentosa, and systemic mastocytosis is characterized by neoplastic mast cell infiltrates in extracutaneous tissues like the bone marrow, GI tract, liver, kidney, and central nervous system. Urticaria pigmentosa, the most common skin manifestation of mastocytosis, often presents as erythematous to brownish macules and patches, typically on the trunk usually in childhood. A 27-year-old male presented with asymptomatic, multiple, 0.2~0.5cm-sized, brown macules and papules on the trunk and both upper extremities for several months. He did not show any other symptoms such as hot-flushing, dyspnea, palpitation, gastrointestinal symptoms, and splenomegaly. Laboratory findings were non-specific. Histopathologic findings revealed perivascular and interstitial lymphohistiocytic and mast cell infiltration and increased epithelial melanin pigments. We diagnosed our patient as urticaria pigmentosa and referred him to the hematologic department for systemic evaluation. Herein, we report a rare case of adult-onset urticaria pigmentosa.
Bullous urticarial pigmentosa in an infant
( Ye Jin Lee ),( Sang Hyung Lee ),( Chong Hyun Won ),( Sung Eun Chang ),( Mi Woo Lee ),( Jee Ho Choi ) 대한피부과학회 2015 대한피부과학회 학술발표대회집 Vol.67 No.1
Urticaria pigmentosa is the most common variant of cutaneous mastocytosis characterized by an increased number of mast cells in the skin. Most children with UP present at birth or arise during the infancy as typical tan to brown papules and less commonly as macules,averaging 1.0 to 2cm in diameter. Lesions frequently appear on trunk but can localized any surface except for the palms and soles. A few reports described urticarial pigmentosa with bullous lesions. Differential diagnosis of blisters in infants were bullous impetigo, disseminated herpes infection, arthropod bites, linear IgA bullous dermatosis, epidermolysis bullosa, and incontinentia pigmenti. A 1-year-old boy presented with 6 monthshistory of multiple grouped 5mm sized crusted papules and vesicles on scalp. The first impression was either bullous impetigo or herpes simplex infection. The patient had been treated with antibiotics and antiviral drugswhich showed no improvement. Four months later, the patient developed pruritic multiple grouped vesicles on left side of the trunk. Histopathologic examination of the lesional skin showed subepidermal bullae and infiltration of numerous mast cells in the papillary and reticular dermis. Based on clinical and histologic findings the diagnosis of bullous urticarial pigmentosa was made. Symptomatic treatment with oral steroid and antihistamines was effective.
김범준,탁우정,김명남,노병인,송계용 대한피부과학회 2002 대한피부과학회지 Vol.40 No.12
Urticaria pigmentosa is a form of urticaria characterized by abnormality of cutaneous mast cells. No prediction for either gender has been reported but it usually occurs in children. When it develops in an adult cutaneous manifestations often follows recalcitrant and persistent course. Though tendency of genetic inheritance with an autosomal dominant pattern has been suggested most patients have no family history of mastocytosis, We report a case of adult-onset urticaria pigmentosa with references. (Korean J Dermatol 2002;40(12) 1587∼1590)
[P068] A case of atypical urticaria pigmentosa
( Hye-jin Ahn ),( Hyoung-jin Park ),( June-hyuck Yim ),( Ki-heon Jeong ),( Mu-hyoung Lee ) 대한피부과학회 2017 대한피부과학회 학술발표대회집 Vol.69 No.1
Mastocytosis is characterized by abnormal accumulation of mast cells in one or more organs. In about two-thirds of cases, mastocytosis begins in childhood. The pediatric onset mastocytosis is usually cutaneous mastocytosis (CM) and in frequent order is urticaria pigmentosa (UP), mastocytoma and diffuse cutaneous mastocytosis. Typically, the lesions of UP are brownish macules or papules scattered on the trunk and Darier sign is positive. A 13-year-old female presented with pruritic pigmented maculopapules on the left anterior chest that had been present for 7 years. Clinical examination revealed a light brownish patch containing multiple brownish to reddish colored 0.2-0.5cm sized maculopapules on the left upper anterior chest. When her skin rubbed, the lesion became itchy and reddened. The histopathology evaluation demonstrated marked and dense dermal infiltration of mast cells. Immunohistochemistry findings were positive for Toluidine blue, Wright-Giemsa and CD117 (c- kit). Laboratory tests to evaluate for systemic disease were all in normal range. We report a rare case of atypical UP showing mastocytoma, histologically.
이나영,이병진,조옥자,박시룡 대한피부과학회 2003 大韓皮膚科學會誌 Vol.41 No.4
Mastocytosis is a heterogenous group of disorders characterized by abnormal growth and accumulation of mast cells in one or more organ systems. Urticaria pigmentosa represents the most common form of cutaneous rnastocytosis in children and adults. We report a case of adult-onset urticaria pigmentosa in a 53-year-old man. The patient had pruritic multiple scattered well-demarcated brownish macules and papules on whole body for 4 years. Histopathologic examination showed diffuse and perivascular infiltration of Iymphocytes and cuboidal mast cells in the upper dermis. Any sign or symptom of systemic involvement was not detected. (Korean J Dermatol 2003;41(4) : 509~512)
박주희 ( Ju Hee Park ),유광호 ( Kwang Ho Yoo ),한태영 ( Tae Young Han ),이갑석 ( Kap Sok Li ),서성준 ( Seong Jun Seo ),홍창권 ( Chang Kwun Hong ) 대한피부과학회 2009 대한피부과학회지 Vol.47 No.9
Urticaria pigmentosa (UP) is the most common variant of cutaneous mastocytosis. Primarily a disease of childhood, in over one-half of the cases onset is before 2 years of age, and in 90%, the disease is confined to the skin. UP precedes the diagnosis of systemic mastocytosis and is more common in adults than in children. Therefore, systemic examination should be performed in patients with UP. We report a case of adult-onset UP with systemic involvement. (Korean J Dermatol 2009;47(9):1060∼1062)
Cutaneous mastocytosis in Korean patients : a clinical features of 77 cases
( Jong Sic Kim ),( Ji Hong Lim ),( Sewon Hwang ),( Yuri Woo ),( Miri Kim ),( Hyun Jeong Park ) 대한피부과학회 2017 대한피부과학회 학술발표대회집 Vol.69 No.2
Background: Mastocytosis represents a group of rare disorders characterized by a neoplastic proliferation of abnormal mast cells in one or more tissues, involving mainly skin, bone marrow, gastrointestinal tract, and liver. Cutaneous mastocytosis is classified according to clinical presentation and is further defined by onset of disease. Objectives: To analyze clinical characteristics of cutaneous mastocytosis in Korean patients. Methods: We performed a retrospective review in 77 patients diagnosed as cutaneous mastocytosis. Patients were evaluated for onset of disease, sex, distribution, associated symptoms and distribution of lesions. Results: There were 16 (20.7%) cases of telangiectasia macularis eruptive perstans cases, 25 (32.5%) cases of urticaria pigmentosa, 22 (28.6%) cases of mastocytoma, and 14 (18.2%) cases of diffuse cutaneous mastocytosis. In 30% of cases disease onset was in the first year of life. There was a male predominance 2.1 : 1. The most common presentation was involvement of trunk together with both extremities. Conclusion: The most frequent clinical form of mastocytosis was urticarial pigmentosa followed by mastocytoma, telangiectasia macularis eruptiva perstans and diffuse cutaneous mastocytosis. Cutaneous mastocytosis is a benign disease in children without systemic involvement.