6か月男児の下顎骨と体幹皮膚に生じた多臓器多病変型ランゲルハンス細胞組織球症の1例
Langerhans cell histiocytosis (LCH) is a relatively rare proliferative disorder of Langerhans cells. We herein report an extremely rare case of LCH arising in the mandible and trunk skin of a 6-month-old boy. A patient was referred to our clinic because of right cheek swelling. Computed tomography r...
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| Published in | 日本口腔外科学会雑誌 Vol. 66; no. 9; pp. 439 - 443 |
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| Main Authors | , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
社団法人 日本口腔外科学会
20.09.2020
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0021-5163 2186-1579 |
| DOI | 10.5794/jjoms.66.439 |
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| Abstract | Langerhans cell histiocytosis (LCH) is a relatively rare proliferative disorder of Langerhans cells. We herein report an extremely rare case of LCH arising in the mandible and trunk skin of a 6-month-old boy. A patient was referred to our clinic because of right cheek swelling. Computed tomography revealed broad bone expansion and a defect in the right mandibular molar region. An excisional biopsy was performed, and the lesion was histopathologically diagnosed as LCH (multi-system multi-site: MM type). Immunohistochemically, the histiocytes were positive for S -100 and CD1a. The patient subsequently underwent chemotherapy (vinblastine and prednisolone), and the lesion was reduced completely. Long-term follow-up should be practiced to monitor for tumor recurrence and hypoplasia of the teeth as a side effect of chemotherapy. There has been no recurrence after 2 and a half years of follow-up. |
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| AbstractList | Langerhans cell histiocytosis (LCH) is a relatively rare proliferative disorder of Langerhans cells. We herein report an extremely rare case of LCH arising in the mandible and trunk skin of a 6-month-old boy. A patient was referred to our clinic because of right cheek swelling. Computed tomography revealed broad bone expansion and a defect in the right mandibular molar region. An excisional biopsy was performed, and the lesion was histopathologically diagnosed as LCH (multi-system multi-site: MM type). Immunohistochemically, the histiocytes were positive for S -100 and CD1a. The patient subsequently underwent chemotherapy (vinblastine and prednisolone), and the lesion was reduced completely. Long-term follow-up should be practiced to monitor for tumor recurrence and hypoplasia of the teeth as a side effect of chemotherapy. There has been no recurrence after 2 and a half years of follow-up. |
| Author | 森下, 雄斗 中村, 知寿 鍋田, 剛志 渋谷, 恭之 福島, 麻子 加藤, 伸一郎 |
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| References | 17) 中谷倫子， 成川 玄， 他：コルチコステロイドの局所注射が奏功した下顎骨ランゲルハンス細胞組織球症の1例．日口外誌 56: 128-132, 2010. 19) 大久保 淳， 浜之上 聡， 他：ランゲルハンス細胞組織球症の臨床像と予後 −単一施設での37年間の経験−．日小児血液会誌 22: 354-359, 2008. 2) 8.1. JLSG-02protocolの概要：日本ランゲルハンス細胞組織球症研究グループ．Available at : http://www.jlsg.jp/protocol/img/JLSG02_protocol.pdf. Accessed June 17, 2019. 8) Madrigal-Martinez-Pereda C, Guerrero-Rodriguez V, et al : Langerhans cell histiocytosis: literature review and descriptive analysis of oral manifestations. Medicina oral, patologia oral y cirugia bucal 14: E222-228, 2009. 6) Hicks J and Flaitz CM : Langerhans cell histiocytosis: current insights in a molecular age with emphasis on clinical oral and maxillofacial pathology practice. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 100: 42-66, 2005. 18) 荒井直樹， 野中 水， 他：非典型的画像所見を呈し診断に苦慮した肺ランゲルハンス細胞組織球症の1例．日呼吸会誌 6: 58-62, 2017. 11) 今宿晋作：ランゲルハンス細胞組織球症 (LCH) 治療の問題点．医事新報 3969: 6-14, 2000. 12) Swerdelow SH, Campo E, et al : WHO classification of tumors heamatopoietic and lymphoid tissues. IARC press, Lyon, 2008, p280-282. 3) Stockschlaeder M and Sucker C : Adult Langerhans cell histiocytosis. European journal of haematology 76: 363-368, 2006. 13) 中島英行， 岩井俊憲， 他：下顎骨を含む多臓器型Langerhans’ cell histiocytosisの1例．日口外誌 63: 570-575, 2017. 15) Minkov M, Grois N, et al : Langerhans cell histiocytosis Histiocyte Society Evaluation and Treatment Guidelines. Available at : http://www.Histiocytesociety.org/document290. Accessed June 3, 2019. 9) 山下雅子， 篠崎泰久， 他：上顎に発生したランゲルハンス細胞組織球症の1例．日口外誌 56: 515-518, 2010. 5) Brown NA, Furtado LV, et al : High prevalence of somatic MAP2KI mutations in BRAF V600E-negative Langerhans cell histiocytosis. Blood 124: 1655-1658, 2014. 4) 馬屋原孝恒， 戸倉新樹：ランゲルハンス細胞組織球症の皮疹を捉える．小児内科 48: 520-523, 2016. 14) 森本 哲：組織球症の病態解明と治療の進歩．日小児血がん会誌 53: 428-435, 2016. 1) Wess LM, Grogan TM, et al : Tumors of haematopoietic and lymphoid tissues. WHO classification of tumors. IARC Press, Lyon, 2001, p280-282. 20) 渡辺賀子， 林祐太郎， 他：化学療法が重大な歯の形成障害をもたらしたと考えられる小児の1例．小児口外 22: 45-48, 2012. 7) Nakamura S, Bessho K, et al : Langerhans’ Cell histiocytosis cofined to the jaw. J oral Maxillofac Surg 63: 989-995, 2005. 10) Cai S, Zhang S, et al : Solitary Langerhans cell histiocytosis of frontal lobe: a case report and literature review. Chinese journal of cancer research. Chungkuo yen cheng yen chiu 26: 211-214, 2014. 16) 乳児ランゲルハンス細胞組織球症という病気について．日本ランゲルハンス細胞組織球症研究グループ．http://www.jlsg.jp/WhatsLCH/WhatsLCH.pdf,2010 Accessed June 7, 2019. |
| References_xml | – reference: 5) Brown NA, Furtado LV, et al : High prevalence of somatic MAP2KI mutations in BRAF V600E-negative Langerhans cell histiocytosis. Blood 124: 1655-1658, 2014. – reference: 13) 中島英行， 岩井俊憲， 他：下顎骨を含む多臓器型Langerhans’ cell histiocytosisの1例．日口外誌 63: 570-575, 2017. – reference: 2) 8.1. JLSG-02protocolの概要：日本ランゲルハンス細胞組織球症研究グループ．Available at : http://www.jlsg.jp/protocol/img/JLSG02_protocol.pdf. Accessed June 17, 2019. – reference: 3) Stockschlaeder M and Sucker C : Adult Langerhans cell histiocytosis. European journal of haematology 76: 363-368, 2006. – reference: 4) 馬屋原孝恒， 戸倉新樹：ランゲルハンス細胞組織球症の皮疹を捉える．小児内科 48: 520-523, 2016. – reference: 10) Cai S, Zhang S, et al : Solitary Langerhans cell histiocytosis of frontal lobe: a case report and literature review. Chinese journal of cancer research. Chungkuo yen cheng yen chiu 26: 211-214, 2014. – reference: 18) 荒井直樹， 野中 水， 他：非典型的画像所見を呈し診断に苦慮した肺ランゲルハンス細胞組織球症の1例．日呼吸会誌 6: 58-62, 2017. – reference: 11) 今宿晋作：ランゲルハンス細胞組織球症 (LCH) 治療の問題点．医事新報 3969: 6-14, 2000. – reference: 15) Minkov M, Grois N, et al : Langerhans cell histiocytosis Histiocyte Society Evaluation and Treatment Guidelines. Available at : http://www.Histiocytesociety.org/document290. Accessed June 3, 2019. – reference: 16) 乳児ランゲルハンス細胞組織球症という病気について．日本ランゲルハンス細胞組織球症研究グループ．http://www.jlsg.jp/WhatsLCH/WhatsLCH.pdf,2010 Accessed June 7, 2019. – reference: 12) Swerdelow SH, Campo E, et al : WHO classification of tumors heamatopoietic and lymphoid tissues. IARC press, Lyon, 2008, p280-282. – reference: 7) Nakamura S, Bessho K, et al : Langerhans’ Cell histiocytosis cofined to the jaw. J oral Maxillofac Surg 63: 989-995, 2005. – reference: 14) 森本 哲：組織球症の病態解明と治療の進歩．日小児血がん会誌 53: 428-435, 2016. – reference: 8) Madrigal-Martinez-Pereda C, Guerrero-Rodriguez V, et al : Langerhans cell histiocytosis: literature review and descriptive analysis of oral manifestations. Medicina oral, patologia oral y cirugia bucal 14: E222-228, 2009. – reference: 1) Wess LM, Grogan TM, et al : Tumors of haematopoietic and lymphoid tissues. WHO classification of tumors. IARC Press, Lyon, 2001, p280-282. – reference: 20) 渡辺賀子， 林祐太郎， 他：化学療法が重大な歯の形成障害をもたらしたと考えられる小児の1例．小児口外 22: 45-48, 2012. – reference: 17) 中谷倫子， 成川 玄， 他：コルチコステロイドの局所注射が奏功した下顎骨ランゲルハンス細胞組織球症の1例．日口外誌 56: 128-132, 2010. – reference: 9) 山下雅子， 篠崎泰久， 他：上顎に発生したランゲルハンス細胞組織球症の1例．日口外誌 56: 515-518, 2010. – reference: 6) Hicks J and Flaitz CM : Langerhans cell histiocytosis: current insights in a molecular age with emphasis on clinical oral and maxillofacial pathology practice. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 100: 42-66, 2005. – reference: 19) 大久保 淳， 浜之上 聡， 他：ランゲルハンス細胞組織球症の臨床像と予後 −単一施設での37年間の経験−．日小児血液会誌 22: 354-359, 2008. |
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| SubjectTerms | 6か月男児 ランゲルハンス細胞組織球症 下顎骨 体幹皮膚 |
| Title | 6か月男児の下顎骨と体幹皮膚に生じた多臓器多病変型ランゲルハンス細胞組織球症の1例 |
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