A case of primary cutaneous CD4-positive small/medium T-cell lymphoproliferative disorder

( Moonhyung You ),( Hyeri Kim ),( Joongoon Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.1

Primary cutaneous CD4-positive small/medium T-cell lymphoproliferative disorder is a term that was revised in the 2016 revision of the WHO classification of lymphoid neoplasms. It was classified as a lymphoma by WHO in 2008, but it had good prognosis and localized lesion. It is more common in women, and the mean age of diagnosis is known to be 50. Clinically, it is often found in the head and neck or upper part of the trunk in the form of a single or localized asymptomatic erythematous, purpuric papule, nodule or plaque. Four cases have been reported so far in Korea. A 46-year-old male patient visited our hospital with an erythematous plaque on his back with pruritus, which he had had for two years and six months. On histological examination, infiltration of lymphocytes, which have small to medium-sized uniform population with pale scanty cytoplasm, hyperchromatic irregular noncerebriform nuclei were observed. It showed monoclonality in the TCR gamma chain gene rearrangement. We diagnosed it as a primary cutaneous CD4-positive small/medium T-cell lymphoproliferative disorder. We report an interesting case of primary cutaneous CD4-positive small/medium T-cell lymphoproliferative disease. The lesion was improved with triamcinolone intralesional injection. In this disease, we should consider conservative local management, because the clinical behavior is almost always indolent, with most patients presenting with localized disease.

A case of cutaneous eosinophilic granuloma

( Moonhyung You ),( Hyeri Kim ),( Joongoon Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.1

Eosinophilic granuloma is a clinical variant of Langerhans histiocytosis characterized by localized lesion. It is most commonly found in bone alone and is often found in skull, pelvis, and iliac bone. A total of 114 cases have been reported in Korea and more than 1,000 cases have been reported worldwide. However, Eosinophilic granuloma confined to skin have not been reported in Korea. A 19-year-old male patient visited our hospital with a right axillary mass with pain and tenderness, which he had had for a month. The lesion was initially a nodule with peripheral erythema, and ulcers have developed after the patient exploded the lesion himself. On histological examination, there were many eosinophils and many Langerhans histiocytes showing nucleus of renal shape and nuclear groove. The liver enzyme level was elevated, but the abdominal computed tomography did not reveal any abnormality other than fatty liver disease, and the systemic bone scan showed no evidence of bone lesion. The entire lesion was excised and is being observed for 2 months. We report an interesting case of cutaneous eosinophilic granuloma, which was rarely reported in Korea.

A case of cutaneous B-lymphoblastic lymphoma

( Moonhyung You ),( Hyeri Kim ),( Joongoon Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.1

B-lymphoblastic lymphoma is a rare non-Hodgkin's lymphoma, accounting for less than 10% of all lymphoblastic lymphomas, and extranodal presentation is more common than in T-lymphoblastic lymphoma. The clinical presentation typically includes single or multiple firm papules and nodules that are erythematous to blue in color. The majority of the reported cases were in the head and neck region. It usually occurs in children and adolescents, 64% of patients being younger than 18 years old at the time of diagnosis. A total of seven cutaneous B-lymphoblastic lymphomas have been reported in Korea, six of which occurred in children under 18 years of age. An 18-year-old male patient visited our hospital with a purpuric, tender nodule on the scalp, which he had had for a month. The patient was diagnosed with alopecia areata in a local medical center 5 months ago, and the lesion was elevated after 6 cycles of intralesional injection of triamcinolone. On histological examination, monotonous diffuse dermal infiltrate consisting of medium-sized blasts with scanty basophilic cytoplasm, finely dispersed chromatin, and inconspicuous but distinct nucleoli was observed. On immunohistochemistry, positivity for CD10, CD79a, BCL2, and TdT were noted. Taken together, the features were consistent with the B-lymphoblastic lymphoma. We report an interesting case of cutaneous B-lymphoblastic lymphoma which was rarely reported in Korea.

A case of granulomatous pigmented purpuric dermatosis

( Moonhyung You ),( Hyeri Kim ),( Joongoon Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.1

Granulomatous pigmented purpuric dermatosis is a rare histologic subtype of pigmented purpuric dermatosis characterized by the infiltration of lymphocytes and histiocytes forming granulomas at the papillary dermis. The clinical presentation typically includes erythematous to purpuric patches on the distal extremities, particularly the lower legs. Many reported cases had underlying hyperlipemia. Although the disease was initially thought to be limited to Asian patients, today Caucasian individuals represent more than one-half of reported cases, and three cases have been reported in Koreans. A 51-year-old female patient visited our hospital with asymptomatic erythematous patches on the both dorsal feet, which she had had for a year. The lesions occurred a year ago and persisted. She stand for long periods of time almost every day because of her occupation, so her legs were swollen often. She had thyroid cancer and hyperlipemia. On histological examination, superficial perivascular lymphohistiocytic infiltrate with some extension to the mid dermis forming scattered, poorly formed nonnecrotizing granulomas was seen. RBC extravasation, hemosiderin deposition were also noted. Taken together, the features were consistent with the granulomatous pigmented purpuric dermatosis. We report a case of granulomatous pigmented purpuric dermatosis which was rarely reported in Korea.

피부 편평세포암종에서 PD-L1의 면역조직화학적 발현과 전이 위험과의 연관성

유문형 ( Moonhyung You ),신동훈 ( Donghoon Shin ),최종수 ( Jongsoo Choi ),배영경 ( Youngkyung Bae ),이지민 ( Jihmin Lee ) 대한피부과학회 2018 대한피부과학회지 Vol.56 No.7

Background: Programmed cell death ligand 1 (PD-L1) plays a major role in the immune responses of a variety of cancers. Recently, several studies revealed that PD-L1 is differently expressed in some cases of non-melanoma skin cancer. The expression of PD-L1 in cutaneous squamous cell carcinoma has not yet been described in Korea. Objective: To investigate the expression of PD-L1 in cutaneous squamous cell carcinoma and its association with variable clinicopathological factors. Methods: We performed immunohistochemical staining of 52 cutaneous cell carcinoma cases, including 28 high-risk cases and 24 low-risk cases. Cases were selected from patients who had visited the department of dermatology of our hospital from 2001 to 2017. The expression patterns were assessed using the H-score. Cases demonstrating at least 1+ of PD-L1 in more than 1% of tumor cells were considered positive. Results: PD-L1 expression of tumor cells was 19.2% (10/52) for all cases, 0.0% (0/24) for the low-risk group, and 35.7% (10/28) for the high-risk group. PD-L1 positive cutaneous squamous cell carcinoma cases showed a significantly higher proportion of large tumors and tumors with deep invasion and a higher lymphatic metastasis rate when compared to PD-L1 negative cutaneous squamous cell carcinoma cases. Conclusion: Our study shows that cutaneous squamous cell carcinoma exhibits PD-L1 expression in 19.2% of cases. PD-L1 positive tumors are associated with high-risk cases of cutaneous squamous cell carcinoma, which may help guide the choice of therapeutic strategy. (Korean J Dermatol 2018;56(7):415∼420)

원발성 피부 CD4 양성 소/중 T세포 림프구증식병 1예

유문형 ( Moonhyung You ),김혜리 ( Hyeri Kim ),김준군 ( Joongoon Kim ),신동훈 ( Donghoon Shin ),최종수 ( Jongsoo Choi ) 대한피부과학회 2019 大韓皮膚科學會誌 Vol.57 No.4

Causative species of sporotrichosis in Korea

( Hyeri Kim ),( Moonhyung You ),( Joongoon Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.2

Background: Sporotrichosis is a chronic cutaneous fungal infection caused by Sporothrix (S.) schenckii complex. Sporotrichosis was common deep mycosis in Korea. All strains were reported as S. schenckii. There was no molecular studies until 2016. In northeast China and Japan, S. globosa was main species of Sporothrix, and S. schenckii sensu stricto was very rare. Objectives: We want to know accurate species of Korean strains reported as S. schenckii. Methods: We searched the Korean strains of Sporothrix spp. at fungus collection centers or private collections and reviewed literatures of molecular studies for Korean strains. We found 6 strains of S. schenckii and 3 strains of S. globosa. Ribosomal DNA ITS sequences were compared with those of the species of Sporothrix schenckii complex. Results: Ribosomal ITS sequences of 9 strains were 100 % identical with S. globosa. There was no S. schenckii sensu stricto. We found one study about molecular analysis of Korean S. schenckii . It was published in 2004 by Ishizaki et al. All of 8 strains of Korean S. schenckii were mitochondrial subtype group 2, that was S. globosa. So all 17 strains of Korean S. schenckii complex were S. globosa. Conclusion: S. globosa was the causative species in tested Korean sporotrichosis.

A case of adult onset rhabdomyomatous mesenchymal hamartoma

( Joongoon Kim ),( Moonhyung You ),( Yeonwoong Kim ),( Byeongsu Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2016 대한피부과학회 학술발표대회집 Vol.68 No.1

Rhabdomyomatous mesenchymal hamartoma is an uncommon benign lesion, which is usually congenital, polypoid. This lesion is primarily located on the head and neck. This hamartoma typically presents as a nodule or skin tag, but has also been described as a solitary indurated plaque or depressed plaque. The critical histopathologic finding is the existence of individualized fascicles of striated muscle affecting the dermis and subcutaneous fat tissue. A 39-year-old male presented with a single walnut sized normal skin colored soft elevated plaque on the chin for about 2 years. We performed punch biopsy, and histopathologic exam showed skeletal muscles and adipose tissues in the dermis. Because of cosmetic morbidity due to large size of hamartoma, he didn’t want surgical treatment. We report a rare case of adult onset rhabdomyomatous mesenchymal hamartoma. Although our patient and other reported cases had no clinically observable abnormalities, we recommend systemic evaluation in the patient with rhabdomyomatous mesenchymal hamartoma because etiology of this disease can be associated with aberrancy in the embryonic migration.

Necrobiotic xanthogranuloma coexists with diffuse normolipidemic plane xanthoma and multiple myeloma

( Hyeri Kim ),( Moonhyung You ),( Joongoon Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.1

Necrobiotic xanthogranuloma (NXG), is a rare multisystem disease that manifests as cutaneous inflammatory lesions, and is commonly associated with lymphoproliferative disease. Diffuse normolipidemic plane xanthoma (NX), is also a rare, acquired disease that is often associated with systemic diseases such as lymphoproliferative disease. Both these diseases have been reported to be associated with monoclonal immunoglobulins (MIg). However, there are few cases in which these diseases co-exist. A 78-year-old woman, who had a known case of NX on the neck and axillary area, presented with an asymptomatic erythematous plaque on her left supraclavicular area for 6 months. Histopathological examination showed lymphoid aggregates, necrobiotic areas, and granulomatous inflammation in the dermis. Numerous foreign-body and Touton type giant cells were noticed. Serum protein immunoelectrophoresis showed an IgG kappa type monoclonal gammopathy. Lipid profile of serum was normal. Bone marrow examination showed plasma cell myeloma. Based on these histologic and laboratory results, we diagnosed this lesion as NXG coexisting with NX and multiple myeloma. She was started on treatment with bortezomib and melphalan for multiple myeloma, and high-dose systemic corticosteroid and triamcinolone intralesional injection for the skin lesion. After 3 months of treatment, the NXG skin lesion and MIg improved.

A case of keratoacanthoma successfully treated with intralesional methotrexate

( Joongoon Kim ),( Moonhyung You ),( Yeonwoong Kim ),( Byeongsu Kim ),( Donghoon Shin ),( Jongsoo Choi ) 대한피부과학회 2016 대한피부과학회 학술발표대회집 Vol.68 No.1

Solitary keratoacanthoma is a rather common, rapidly growing neoplasm appearing mainly on sun-exposed areas of the skin in elderly, light-complexioned person. The majority of these lesions occur on the face, forearms, and hands. It is thought that intralesional methotrexate is effective as nonsurgical treatment of keratoacanthoma. A 85-year-old female presented with history of a rapidly growing 1.5cm-sized mass on the right submandible for 2 months. Histopathologic examination showed eosinophilic and glassy cell within shell and resulted in diagnosis of keratoacanthoma. We performed methotrexate injection with 0.1cc (50mg/2cc) each, four times in a row, with an interval of 3 weeks between inspections. After 6 weeks of treatment, the skin lesion had almost completely resolved. We report a case of keratoacanthoma which was successfully treated by intralesional methotrexate. Intralesional methotrexate can promote rapid improvement so in patient with cosmetic concern, we recommend intralesional methotrexate as nonsurgical treatment of keratoacanthoma.