Hifu no kagaku
Online ISSN : 1883-9614
Print ISSN : 1347-1813
ISSN-L : 1347-1813
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COLOR LIBRARY
CASE REPORT
  • Michiko Nagahama, Yukiko Tanaka, Tadashi Nomura, Masatoshi Jinnin, ...
    2020 Volume 19 Issue 3 Pages 163-168
    Published: 2020
    Released: October 28, 2020
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    A 1-month-old girl was referred to our hospital with a complaint of capillary malformation on the right side of her head, body, arm and leg as well as hypertrophy of her right upper limb. The patient had no other vascular anomalies or complications. We therefore diagnosed this case with diffuse capillary malformation with overgrowth (DCMO), which was first described by Lee et al. in 2013 :The clinical presentation is extensive and, diffuse capillary malformation and variable hypertrophy without major complications. It is considered a distinct entity from overgrowth syndromes. Skin Research, 19 : 163-168, 2020

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  • Yuji Horiguchi, Takaya Fukumoto, Akihiko Kitoh
    2020 Volume 19 Issue 3 Pages 169-173
    Published: 2020
    Released: October 28, 2020
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    A79-year-old male suffered from itching dermatitis for several months, gradually spreading from the pelvic region to the trunk and central extremities. Six-month treatment with topical steroid ointment prescribed by the previous doctor showed only limited effectiveness. Physical examination disclosed numerous slightly pigmented new and old erythematous plaques over the body. Each eruption was smaller than 3 centimeters in diameter and slightly palpable. Tumorous lesions were absent. Biopsy examination disclosed extensive histiocytoid and mononuclear cell infiltrations in the upper dermis. However, the marked proliferation of these cells was not detected. Immunohistochemical examination demonstrated abundant CD1a + cells infiltration and much amount CD3+ cells, but CD20+ cells and CD68+ cells were only sparsely distributed. Further examination for whole body found no lymph node swelling, abnormality of peripheral blood count, or monoclonal rearrangement of T-cell receptor genes. NB-UVB irradiation therapy was started, and the eruptions diminished in the three subsequent months. A2-year follow-up revealed no recurrence of the skin disturbance. Not only typical cases of Langerhans cell histiocytosis, involving multisystemic and tumor-forming, neoplastic, and clonal proliferation of Langerhans cells occurring in infants and children, but also cases showing skin limited infiltration or proliferation of Langerhans cells are reported. They include self-healing cases of neonate and infant, and adult types that show wide spread eruptions. They are now called cutaneous Langerhans cell histiocytosis. This case is diagnosed as an early stage of cutaneous Langerhans cell histiocytosis. Skin Research, 19 : 169-173, 2020

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  • Marina Nishida, Nao Kusutani, Junko Osako, Takahiko Nakane, Masahi ...
    2020 Volume 19 Issue 3 Pages 174-179
    Published: 2020
    Released: October 28, 2020
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    A 60-year-old woman presented with a 1-year history of asymptomatic subcutaneous nodules on the lower extremities. Physical examination revealed multiple hard subcutaneous nodules with a smooth surface distributed on the bilateral lower extremities. Histopathological examination of a skin biopsy specimen from her left lower extremity revealed nodules with eosinophilic amorphous material in the dermis to the subcutaneous tissue. Congo red and direct fast scarlet staining tests revealed the amorphous material was amyloid, and immunohistochemical examination confirmed it to be light-chain (AL) amyloid. Additionally, a transbronchial biopsy specimen from a nodule of the left lung showed amyloid deposition. Further investigation showed involvement of no organ other than skin and the bilateral lungs. She has been stable over two years of follow-up. Systemic AL amyloidosis rarely presents with primary clinical features of multiple subcutaneous nodules of the extremities. Treatments and prognoses differ depending on disease types and organs affected by this condition. Early diagnosis is essential for appropriate treatment. Skin Research, 19 : 174-179, 2020

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  • Takako Shijimaya, Hirotsugu Tanimura, Mari Tsuda, Naoko Nagano, Sa ...
    2020 Volume 19 Issue 3 Pages 180-184
    Published: 2020
    Released: October 28, 2020
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    An 85-year-old Japanese man presented with a growing nodule for four months. A clinical investigation revealed a 3 cm-sized, bloody, dark-red, dome-shaped nodule on the root of the nose. Dermoscopy detected milky red areas, crust, and linear irregular vessels. The remaining tumor was totally excised inorder to stop bleeding 2 days after the biopsy. Histologically, the tumor was domeshaped, and was composed of sheet-like basophilic nodule in the dermis. The tumor cells had small round nuclei with fine granular chromatin with ample cytoplasm. N/C ratio was high, and atypical mitoses could be detected. Immunohistochemically, tumor cells were positive for synaptophysin,chromograninA, CD56, CK20, and Merkel cell polyomavirus large T-antigen (clone : CM2B4). They were negative for CK7, CD31, D2-40 and TTF-1. The present case was diagnosed as Merkel cell polyomavirus-positive Merkel cell carcinoma. A postoperative radiotherapy was performed. Since the identification of Merkel cell polyomavirus inMerkel cell carcinoma in2008, its positive rates have beenreported to vary greatly betweencountries. Based onthe approval of anti-PD-L1 antibody avelumab for unresectable Merkel cell carcinomas, an accumulation of further cases and analyses are necessary in Japan. Skin Research, 19 : 180-184, 2020

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  • Kei Sakano, Reiko Takahashi, Kunitaro Fukuyama
    2020 Volume 19 Issue 3 Pages 185-189
    Published: 2020
    Released: October 28, 2020
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    Case 1was a 45-year-old woman. She had a nodule on right cheek 14 months before her first visit to our hospital. CT revealed a cylindrical structure suggestive of an external dental fistula in the soft tissues. She received root canal treatment, and the nodule regressed. Case 2 was a 64-yearold man. He found a nodule on the left alar crease one year ago. He received electrocauterization,but it recurred. A biopsy revealed an intense inflammatory reaction. Any structures between the nodule and maxillary teeth were not found on a computed tomography (CT). While he was treated with oral minocycline, the nodule shrinked, but it enlarged again when he stopped it. An apical periodontitis was observed, and the nodule was diagnosed as external dental fistula based on cone beam computed tomography (CBCT). He received root canal treatment. The nodule became flat,and local recurrence has not appeared during 14 months of follow-up. External dental fistula is a facial drainage route originating from a tooth with chronic suppurative inflammation. It tends to take time to be made a diagnosis because of lacking acute tooth symptoms. CT is effective for external dental fistula diagnosis, especially when a sonde cannot find fistula, and CBCT is effective at detecting peri apical lesion. However, it should be considered to consult dentist when a nodule in face is intractable, even if any obvious structures or odontopathy are not find in imaging examinations, because they have limitations. Skin Research, 19 : 185-189, 2020

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  • Shoko Miyamoto, Yasutomo Imai, Yukako Inoue, Makoto Nagai, Masaru ...
    2020 Volume 19 Issue 3 Pages 190-196
    Published: 2020
    Released: October 28, 2020
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    Case 1 is a 54-year-old female. Erythema appeared on her right upper arm and left wrist 10 days before the first visit. As the erythema had spread to her entire body and pustules had appeared, she was referred to our hospital. Her drug history raised suspicions of acute generalized exanthematous pustulosis, but systemic corticosteroid therapy was ineffective. Her poor response to corticosteroid therapy and the finding of an IL36RN gene mutation in a genetic test led to a decision to treat her for generalized pustular psoriasis. Her symptoms were improved after treatment with an infliximab biosimilar. Case 2 is a 36-year-old female. She developed psoriasis vulgaris at about age 20, and she had been treated by a local dermatologist since then. Erythema with small pustules appeared 10 months before her first visit, and she had started taking apremilast 5 months before that visit. Her symptoms were not improved, and she was referred to our hospital. She was given a diagnosis of generalized pustular psoriasis and treated with granulocyte and monocyte adsorptions plus an infliximab biosimilar, and her symptoms were improved. Case 3 is a 66-year-old man. At about age 15, he developed erythema and scales on his head. At about age 33, he developed a fever, and erythema with pustules appeared on his trunk. He had a diagnosis of generalized pustular psoriasis by a local dermatologist. He started taking etretinate, and his symptoms were improved. However, 1 month before the first visit, his erythema with pustules recurred and he was referred to our hospital. His symptoms improved after treatment with an infliximab biosimilar. Infliximab biosimilar drugs are approved for psoriasis by extrapolated drug reaction, and their efficacy and safety for generalized pustular psoriasis in Japan are unclear. In our hospital, an infliximab biosimilar was effective in all three cases, although the combination of an infliximab biosimilar with granulocyte and monocyte adsorptions or etretinate may contribute to that results. Skin Research, 19 :190-196, 2020

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  • Michihiro Takiwaki, Yoshihumi Iwahashi, Tatsuya Shimomatsu, Shinji ...
    2020 Volume 19 Issue 3 Pages 197-201
    Published: 2020
    Released: October 28, 2020
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    We reported a case of a fibrous hamartoma of infancy occurring on the right waist of a one-year-old boy with neurofibromatosis type 1 (NF1). The subcutaneous tumor was first noticed at the age of 4 months. He presented with a 30×30 mm elastic soft mass, and vellus hairs were shown on the surface. Café-au-lait spots with halo and ectopic Mongolian spot also existed on his back. Histopathological findings from a biopsy specimen taken from it, we diagnosed the tumor as fibrous hamartoma of infancy (FHI). FHI is a benign soft tissue tumor, composed of fibrous tissue,undifferentiated mesenchymal cell nests and matured fat cells. In particular, FHI with vellus hairs or with NF 1 is a rare subcutaneous soft tissue tumor in children. It is significant that we distinguish FHI with other mesenchymal tumors with NF1. Skin Research, 19: 197-201, 2020

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  • Norika Kawamura, Akitoshi Yu, Hideaki Tanizaki, Teruo Kurokawa, Sh ...
    2020 Volume 19 Issue 3 Pages 202-207
    Published: 2020
    Released: October 28, 2020
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    A 34-year-old man was admitted to our department because of a brownish cutaneous tumor of the right hip from 3 years ago and pain 1 year ago. At the first examination, blackish brown mass was observed on the right hip, and a dermoscopy examination showed a central white patch and histopathological analysis revealed proliferation of spindle-shaped cells in the dermis, spaces lacking vascular endothelium, and hemosiderin deposition. Immunostaining revealed that spindle-shaped cells were partially positive for factor XIIIa and negative for CD34, and the patient was diagnosed with aneurysmal fibrous histiocytoma. This dermoscopy finding of the case reflects the image of cutaneous fibroma with hemosiderin deposits, and was considered to be helpful for the diagnosis of this disease. Skin Research, 19 : 202-207, 2020

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