A Case of Refractory Oral Lichen Planus Successfully Controlled by Systemic Cancer-Chemotherapy for Recurrent Thymoma
Thymoma is a rare disease with an incidence of about 0.5 per 100,000 people, and 0.3-6.3% of thymoma are accompanied by painful and intractable oral lichen planus, OLP. Symptomatic relief of OLP by thymectomy is reported to be not effective, and treatment by systemic chemotherapy for recurrent thymo...
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| Published in | JOURNAL OF THE KYORIN MEDICAL SOCIETY Vol. 54; no. 3; pp. 139 - 145 |
|---|---|
| Main Authors | , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
The Kyorin Medical Society
06.10.2023
杏林医学会 |
| Subjects | |
| Online Access | Get full text |
| ISSN | 0368-5829 1349-886X |
| DOI | 10.11434/kyorinmed.54.139 |
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| Abstract | Thymoma is a rare disease with an incidence of about 0.5 per 100,000 people, and 0.3-6.3% of thymoma are accompanied by painful and intractable oral lichen planus, OLP. Symptomatic relief of OLP by thymectomy is reported to be not effective, and treatment by systemic chemotherapy for recurrent thymoma is rarely reported. We report the case of a woman in her 40s with no particular medical history. She was incidentally pointed out for a mediastinal tumor by a routine medical check-up, and diagnosed as thymoma pStage IVa. Thymoma was resected, but 6 months after resection thymoma recurrenced. Although local radiotherapy was performed four times in total, thymoma progressed, and resulted in radiotherapy-refractory Thymoma, accompanied by OLP. Systemic chemotherapy as PAC (cisplatin, adriamycin, cyclophosphamide) regimen for unresectable thymoma controlled not only thymoma, but also refractory OLP. After systemic chemotherapy for unresectable thymoma, there has been no recurrence of OLP. Survival period is approximately 8 years from surgery of thymoma. |
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| AbstractList | Thymoma is a rare disease with an incidence of about 0.5 per 100,000 people, and 0.3-6.3% of thymoma are accompanied by painful and intractable oral lichen planus, OLP. Symptomatic relief of OLP by thymectomy is reported to be not effective, and treatment by systemic chemotherapy for recurrent thymoma is rarely reported. We report the case of a woman in her 40s with no particular medical history. She was incidentally pointed out for a mediastinal tumor by a routine medical check-up, and diagnosed as thymoma pStage IVa. Thymoma was resected, but 6 months after resection thymoma recurrenced. Although local radiotherapy was performed four times in total, thymoma progressed, and resulted in radiotherapy-refractory Thymoma, accompanied by OLP. Systemic chemotherapy as PAC (cisplatin, adriamycin, cyclophosphamide) regimen for unresectable thymoma controlled not only thymoma, but also refractory OLP. After systemic chemotherapy for unresectable thymoma, there has been no recurrence of OLP. Survival period is approximately 8 years from surgery of thymoma. Thymoma is a rare disease with an incidence of about 0.5 per 100,000 people, and 0.3-6.3% of thymoma are accompanied by painful and intractable oral lichen planus, OLP. Symptomatic relief of OLP by thymectomy is reported to be not effective, and treatment by systemic chemotherapy for recurrent thymoma is rarely reported. We report the case of a woman in her 40s with no particular medical history. She was incidentally pointed out for a mediastinal tumor by a routine medical check-up, and diagnosed as thymoma pStage IVa. Thymoma was resected, but 6 months after resection thymoma recurrenced. Although local radiotherapy was performed four times in total, thymoma progressed, and resulted in radiotherapy-refractory Thymoma, accompanied by OLP. Systemic chemotherapy as PAC (cisplatin, adriamycin, cyclophosphamide) regimen for unresectable thymoma controlled not only thymoma, but also refractory OLP. After systemic chemotherapy for unresectable thymoma, there has been no recurrence of OLP. Survival period is approximately 8 years from surgery of thymoma. 胸腺腫は希な疾患であり，難治性口腔扁平苔癬（Oral Lichen Planus, OLP）を稀に随伴する。胸腺切除術によるOLPの症状緩和は限定的であり，切除不能胸腺腫においては全身化学療法による加療となるが，OLPに対する症状緩和の報告は殆どない。症例は既往歴に特記のない40歳代の女性。健診で偶発的に縦隔腫瘍を指摘され，胸腺腫Stage IVaの診断を得た。抗AChR陽性の無症候性の重症筋無力症を随伴していたが，口腔内に特記所見を認めなかった。胸腺腫切除術を行ったが再発を繰り返し，計4回の放射線治療を施行したものの病勢進行し，ステロイド局所療法に抵抗性の難治性OLPを発症した。切除不能胸腺腫にプラチナ系とアントラサイクリン系併用療法による全身化学療法を導入したところ，胸腺腫の病勢制御に加えて難治性OLPの改善を得た。以降OLPの再燃なく，胸腺腫手術から凡そ8年の生存期間を得ている。 |
| Author | Kamma, Hiroshi Ikeda, Tetsuya Kawai, Kirio Maesono, Tomohiro Shimoyamada, Hiroaki Fujiwara, Masachika Hironaka, Hidekazu Nagashima, Fumio |
| Author_FL | 河合 桐男 藤原 正親 Maesono Tomohiro 下山田 博明 池田 哲也 菅間 博 長島 文夫 廣中 秀一 |
| Author_FL_xml | – sequence: 1 fullname: 河合 桐男 – sequence: 2 fullname: Maesono Tomohiro – sequence: 3 fullname: 池田 哲也 – sequence: 4 fullname: 下山田 博明 – sequence: 5 fullname: 藤原 正親 – sequence: 6 fullname: 菅間 博 – sequence: 7 fullname: 長島 文夫 – sequence: 8 fullname: 廣中 秀一 |
| Author_xml | – sequence: 1 fullname: Nagashima, Fumio organization: Division of Medical Oncology, Kyorin University School of Medicine – sequence: 1 fullname: Maesono, Tomohiro organization: Division of Medical Oncology, Kyorin University School of Medicine – sequence: 1 fullname: Fujiwara, Masachika organization: Department of pathology, Kyorin University School of Medicine – sequence: 1 fullname: Kamma, Hiroshi organization: Nasu Institute of Medical Sciences – sequence: 1 fullname: Kawai, Kirio organization: Department of Pain and Palliative Medicine, Palliative Care Consultation Team, The University of Tokyo Hospital – sequence: 1 fullname: Shimoyamada, Hiroaki organization: Department of pathology, Kyorin University School of Medicine – sequence: 1 fullname: Ikeda, Tetsuya organization: Division of Oro-facial surgery, Kyorin University School of Medicine – sequence: 1 fullname: Hironaka, Hidekazu organization: Division of Medical Oncology, Kyorin University School of Medicine |
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| DocumentTitle_FL | 全身がん薬物療法による再発胸腺腫の病勢制御に伴い軽快した難治性口腔扁平苔癬の一例 |
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| References | 8) Yasumizu Yoshiaki, Ohkura Naganari, Murata Hisashi, Kinoshita Makoto, Funaki Soichiro, Nojima Satoshi, Kido Kansuke, Kohara Masaharu, Motooka Daisuke, Okuzaki Daisuke and others : Myasthenia gravis-specific aberrant neuromuscular gene expression by medullary thymic epithelial cells in thymoma. Nat Commun 13 : 4230, 2022. 2) Bernard C., Frih H., Pasquet F., Kerever S., Jamilloux Y., Tronc F., Guibert B., Isaac S., Devouassoux M., Chalabreysse L., and others : Thymoma associated with autoimmune diseases : 85 cases and literature review. Autoimmun Rev 15 : 82-92, 2016. 7) Dresser Laura, Wlodarski Richard, Rezania Kourosh, Soliven Betty : Myasthenia gravis : epidemiology, pathophysiology and clinical manifestations. J Clin Med 10 : 2235, 2021. 12) Ferrisse Tulio Morandin, de Oliveira Analu Barros, Palacon Mariana Paravani, da Silveira Heitor Albergoni, Massucato Elaine Maria Sgavioli, de Almeida Luciana Yamamoto, Leon Jorge Esquiche, Bufalino Andreia : Immunohistochemical evaluation of Langerhans cells in oral lichen planus and oral lichenoid lesions. Arch. Oral Biol 124 : 105027, 2021. 11) Carrion Daniela Calderon, Korkmaz Yuksel, Cho Britta, Kopp Marion, Bloch Wilhelm, Addicks Klaus, Niedermeier Wilhelm : Loss of Aβ -nerve endings associated with the Merkel cell-neurite complex in the lesional oral mucosa epithelium of lichen planus and hyperkeratosis. Int J Oral Sci 8 : 32-38, 2016. 14) Jiang Ruoyi, Hoehn Kenneth B., Lee Casey S., Pham Minh C., Homer Robert J., Detterbeck Frank C., Aban Inmaculada, Jacobson Leslie, Vincent Angela, Nowak Richard J. and others : Thymus-derived B cell clones persist in the circulation after thymectomy in myasthenia gravis. Proc Natl Acad Sci U S A 117 : 30649-30660, 2020. 5) Kunitoh H., Tamura T., Shibata T., Nakagawa K., Takeda K., Nishiwaki Y., Osaki Y., Noda K., Yokoyama A., Saijo N. : A phase-II trial of dose-dense chemotherapy in patients with disseminated thymoma : report of a Japan Clinical Oncology Group trial (JCOG 9605). Br. J. Cancer 101 : 1549-1554, 2009. 10) Anjum Rubina, Singh Jasmin, Kuduva S. : A clinicohistopathologic study and probable mechanism of pigmentation in oral lichen planus. World J Dent 3 : 330-334, 2012. 6) Loehrer Sr Patrick J., Chen Michael, Kim KyungMann, Aisner Seena C., Einhorn Lawrence H., Livingston Robert, Johnson David : Cisplatin, doxorubicin, and cyclophosphamide plus thoracic radiation therapy for limited-stage unresectable thymoma : an intergroup trial. J Clin Oncol 15 : 3093-3099, 1997. 9) Hana Anisa, Booken Dirk, Henrich Carmen, Gratchev Alexei, Maas-Szabowski Nicole, Goerdt Sergij, Kurzen Hjalmar : Functional significance of non-neuronal acetylcholine in skin epithelia. Life Sci 80 : 2214-2220, 2007. 15) Carbone Mario, Goss E., Carrozzo M., Castellano S., Conrotto D., Broccoletti Roberto, Gandolfo Sergio : Systemic and topical corticosteroid treatment of oral lichen planus : a comparative study with long-term follow-up. J Oral Pathol Med 32 : 323-329, 2003. 1) Elenbaas Andrea, Enciso Reyes, Al-Eryani Kamal : Oral lichen planus : a review of clinical features, etiologies, and treatments. Dentistry Review 2 : 100007, 2022. 4) Motegi Sei-ichiro, Uchiyama Akihiko, Yamada Kazuya, Toki Sayaka, Amano Hiroo, Ishikawa Osamu : Lichen planus complicated with thymoma : Report of three Japanese cases and review of the published work. J. Dermatol. 42 : 1072-1077, 2015. 13) Passuello Nicola, Pozza Gioia, Blandamura Stella, Valmasoni Michele, Sperti Cosimo : Thymoma metastatic to liver and pancreas : case report and review of the literature. J. Int. Med. Res 45 : 868-874, 2017. 3) Flamenbaum Helen S., Safai Bijan, Siegal Frederick P., Pahwa Savita : Lichen planus in two immunodeficient hosts. J. Am. Acad. Dermatol 6 : 918-920, 1982. |
| References_xml | – reference: 9) Hana Anisa, Booken Dirk, Henrich Carmen, Gratchev Alexei, Maas-Szabowski Nicole, Goerdt Sergij, Kurzen Hjalmar : Functional significance of non-neuronal acetylcholine in skin epithelia. Life Sci 80 : 2214-2220, 2007. – reference: 11) Carrion Daniela Calderon, Korkmaz Yuksel, Cho Britta, Kopp Marion, Bloch Wilhelm, Addicks Klaus, Niedermeier Wilhelm : Loss of Aβ -nerve endings associated with the Merkel cell-neurite complex in the lesional oral mucosa epithelium of lichen planus and hyperkeratosis. Int J Oral Sci 8 : 32-38, 2016. – reference: 13) Passuello Nicola, Pozza Gioia, Blandamura Stella, Valmasoni Michele, Sperti Cosimo : Thymoma metastatic to liver and pancreas : case report and review of the literature. J. Int. Med. Res 45 : 868-874, 2017. – reference: 7) Dresser Laura, Wlodarski Richard, Rezania Kourosh, Soliven Betty : Myasthenia gravis : epidemiology, pathophysiology and clinical manifestations. J Clin Med 10 : 2235, 2021. – reference: 2) Bernard C., Frih H., Pasquet F., Kerever S., Jamilloux Y., Tronc F., Guibert B., Isaac S., Devouassoux M., Chalabreysse L., and others : Thymoma associated with autoimmune diseases : 85 cases and literature review. Autoimmun Rev 15 : 82-92, 2016. – reference: 6) Loehrer Sr Patrick J., Chen Michael, Kim KyungMann, Aisner Seena C., Einhorn Lawrence H., Livingston Robert, Johnson David : Cisplatin, doxorubicin, and cyclophosphamide plus thoracic radiation therapy for limited-stage unresectable thymoma : an intergroup trial. J Clin Oncol 15 : 3093-3099, 1997. – reference: 3) Flamenbaum Helen S., Safai Bijan, Siegal Frederick P., Pahwa Savita : Lichen planus in two immunodeficient hosts. J. Am. Acad. Dermatol 6 : 918-920, 1982. – reference: 14) Jiang Ruoyi, Hoehn Kenneth B., Lee Casey S., Pham Minh C., Homer Robert J., Detterbeck Frank C., Aban Inmaculada, Jacobson Leslie, Vincent Angela, Nowak Richard J. and others : Thymus-derived B cell clones persist in the circulation after thymectomy in myasthenia gravis. Proc Natl Acad Sci U S A 117 : 30649-30660, 2020. – reference: 4) Motegi Sei-ichiro, Uchiyama Akihiko, Yamada Kazuya, Toki Sayaka, Amano Hiroo, Ishikawa Osamu : Lichen planus complicated with thymoma : Report of three Japanese cases and review of the published work. J. Dermatol. 42 : 1072-1077, 2015. – reference: 1) Elenbaas Andrea, Enciso Reyes, Al-Eryani Kamal : Oral lichen planus : a review of clinical features, etiologies, and treatments. Dentistry Review 2 : 100007, 2022. – reference: 5) Kunitoh H., Tamura T., Shibata T., Nakagawa K., Takeda K., Nishiwaki Y., Osaki Y., Noda K., Yokoyama A., Saijo N. : A phase-II trial of dose-dense chemotherapy in patients with disseminated thymoma : report of a Japan Clinical Oncology Group trial (JCOG 9605). Br. J. Cancer 101 : 1549-1554, 2009. – reference: 10) Anjum Rubina, Singh Jasmin, Kuduva S. : A clinicohistopathologic study and probable mechanism of pigmentation in oral lichen planus. World J Dent 3 : 330-334, 2012. – reference: 15) Carbone Mario, Goss E., Carrozzo M., Castellano S., Conrotto D., Broccoletti Roberto, Gandolfo Sergio : Systemic and topical corticosteroid treatment of oral lichen planus : a comparative study with long-term follow-up. J Oral Pathol Med 32 : 323-329, 2003. – reference: 8) Yasumizu Yoshiaki, Ohkura Naganari, Murata Hisashi, Kinoshita Makoto, Funaki Soichiro, Nojima Satoshi, Kido Kansuke, Kohara Masaharu, Motooka Daisuke, Okuzaki Daisuke and others : Myasthenia gravis-specific aberrant neuromuscular gene expression by medullary thymic epithelial cells in thymoma. Nat Commun 13 : 4230, 2022. – reference: 12) Ferrisse Tulio Morandin, de Oliveira Analu Barros, Palacon Mariana Paravani, da Silveira Heitor Albergoni, Massucato Elaine Maria Sgavioli, de Almeida Luciana Yamamoto, Leon Jorge Esquiche, Bufalino Andreia : Immunohistochemical evaluation of Langerhans cells in oral lichen planus and oral lichenoid lesions. Arch. Oral Biol 124 : 105027, 2021. |
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| SubjectTerms | choroid plexus ; astrocyte ; thymoma para-neoplastic symptom refractory oral lichen planus systemic cancer- chemotherapy 全身がん化学療法 胸腺腫 腫瘍随伴症状 難治性口腔扁平苔癬 |
| Title | A Case of Refractory Oral Lichen Planus Successfully Controlled by Systemic Cancer-Chemotherapy for Recurrent Thymoma |
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