Kindler syndrome with intractable squamous cell carcinoma arising in the oral cavity: A case report and review of cases

Kindler syndrome (KS) is an extremely rare subtype of epidermolysis bullosa characterized by blistering of the extremities, progressive polymorphous skin atrophy, and mucosal inflammation. There is no curative therapy for KS, only symptomatic treatment. Patients with KS generally present with squamo...

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Vydáno v:	Current problems in cancer. Case reports Ročník 19; s. 100379
Hlavní autoři:	Sugauchi, Akinari, Uchihashi, Toshihiro, Isomura, Emiko Tanaka, Seki, Soju, Kimura, Yuka, Kiyohara, Eiji, Fujimoto, Manabu, Tanaka, Susumu, Tanemura, Atsushi
Médium:	Journal Article
Jazyk:	angličtina
Vydáno:	Elsevier Inc 01.09.2025 Elsevier
Témata:	Hematology, Oncology, and Palliative Medicine Kindler syndrome Oral cavity Palliative care Squamous cell carcinoma Oral cavity OSCC Squamous cell carcinoma RT SCC IMRT PD Kindler syndrome Palliative care KS CR progressive disease complete response oral squamous cell carcinoma intensity-modulated radiation therapy radiation therapy
ISSN:	2666-6219, 2666-6219
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Abstract	Kindler syndrome (KS) is an extremely rare subtype of epidermolysis bullosa characterized by blistering of the extremities, progressive polymorphous skin atrophy, and mucosal inflammation. There is no curative therapy for KS, only symptomatic treatment. Patients with KS generally present with squamous cell carcinoma of the extremities and lips, which are more susceptible to irritation, with only a few cases involving the oral cavity. Herein, we report the case of a 53-year-old female patient with KS, who developed oral squamous cell carcinoma. While initial treatment with intensity-modulated radiation therapy was successful, shortly thereafter, she relapsed and was not responsive to multiple rounds of chemotherapy. Ultimately, palliative care at home was suggested. While the patient received radiation therapy and chemotherapy, she complained of difficulty breathing due to the tumor, especially during the last round of chemotherapy. As a result of aggressive weight reduction of tumor necrotic tissue, the patient's respiratory distress improved. Based on our experience in this case, we propose that the removal of oral tumor necrotic tissues, which is not often performed as part of conventional curative treatment approaches, can sometimes be efficacious for the maintenance of the quality of life, especially in a palliative context for OSCC patients with KS.
AbstractList	Kindler syndrome (KS) is an extremely rare subtype of epidermolysis bullosa characterized by blistering of the extremities, progressive polymorphous skin atrophy, and mucosal inflammation. There is no curative therapy for KS, only symptomatic treatment. Patients with KS generally present with squamous cell carcinoma of the extremities and lips, which are more susceptible to irritation, with only a few cases involving the oral cavity. Herein, we report the case of a 53-year-old female patient with KS, who developed oral squamous cell carcinoma. While initial treatment with intensity-modulated radiation therapy was successful, shortly thereafter, she relapsed and was not responsive to multiple rounds of chemotherapy. Ultimately, palliative care at home was suggested. While the patient received radiation therapy and chemotherapy, she complained of difficulty breathing due to the tumor, especially during the last round of chemotherapy. As a result of aggressive weight reduction of tumor necrotic tissue, the patient's respiratory distress improved. Based on our experience in this case, we propose that the removal of oral tumor necrotic tissues, which is not often performed as part of conventional curative treatment approaches, can sometimes be efficacious for the maintenance of the quality of life, especially in a palliative context for OSCC patients with KS. AbstractKindler syndrome (KS) is an extremely rare subtype of epidermolysis bullosa characterized by blistering of the extremities, progressive polymorphous skin atrophy, and mucosal inflammation. There is no curative therapy for KS, only symptomatic treatment. Patients with KS generally present with squamous cell carcinoma of the extremities and lips, which are more susceptible to irritation, with only a few cases involving the oral cavity. Herein, we report the case of a 53-year-old female patient with KS, who developed oral squamous cell carcinoma. While initial treatment with intensity-modulated radiation therapy was successful, shortly thereafter, she relapsed and was not responsive to multiple rounds of chemotherapy. Ultimately, palliative care at home was suggested. While the patient received radiation therapy and chemotherapy, she complained of difficulty breathing due to the tumor, especially during the last round of chemotherapy. As a result of aggressive weight reduction of tumor necrotic tissue, the patient's respiratory distress improved. Based on our experience in this case, we propose that the removal of oral tumor necrotic tissues, which is not often performed as part of conventional curative treatment approaches, can sometimes be efficacious for the maintenance of the quality of life, especially in a palliative context for OSCC patients with KS.
ArticleNumber	100379
Author	Sugauchi, Akinari Seki, Soju Fujimoto, Manabu Kimura, Yuka Kiyohara, Eiji Uchihashi, Toshihiro Tanaka, Susumu Tanemura, Atsushi Isomura, Emiko Tanaka
Author_xml	– sequence: 1 givenname: Akinari surname: Sugauchi fullname: Sugauchi, Akinari organization: Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, The Universty of Osaka, Japan – sequence: 2 givenname: Toshihiro surname: Uchihashi fullname: Uchihashi, Toshihiro email: uchihashi.toshihiro.dent@osaka-u.ac.jp organization: Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, The Universty of Osaka, Japan – sequence: 3 givenname: Emiko Tanaka surname: Isomura fullname: Isomura, Emiko Tanaka organization: Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, The Universty of Osaka, Japan – sequence: 4 givenname: Soju surname: Seki fullname: Seki, Soju organization: Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, The Universty of Osaka, Japan – sequence: 5 givenname: Yuka surname: Kimura fullname: Kimura, Yuka organization: Department of Dermatology, Graduate School of Medicine, The Universty of Osaka, Japan – sequence: 6 givenname: Eiji surname: Kiyohara fullname: Kiyohara, Eiji organization: Department of Dermatology, Graduate School of Medicine, The Universty of Osaka, Japan – sequence: 7 givenname: Manabu surname: Fujimoto fullname: Fujimoto, Manabu organization: Department of Dermatology, Graduate School of Medicine, The Universty of Osaka, Japan – sequence: 8 givenname: Susumu surname: Tanaka fullname: Tanaka, Susumu organization: Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, The Universty of Osaka, Japan – sequence: 9 givenname: Atsushi surname: Tanemura fullname: Tanemura, Atsushi organization: Department of Dermatology, Graduate School of Medicine, The Universty of Osaka, Japan
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Cites_doi	10.1002/humu.21576 10.1016/j.jdcr.2022.06.005 10.1200/JCO.1990.8.2.342 10.5021/ad.2021.33.4.382 10.1902/jop.2008.070167 10.1016/j.rpor.2016.07.004 10.1016/j.det.2009.10.012 10.1007/s12105-022-01441-w 10.1016/j.anorl.2017.05.003 10.1046/j.1365-2133.2001.04262.x 10.1002/1097-0142(19901015)66:8<1692::AID-CNCR2820660807>3.0.CO;2-Y 10.1186/s13023-019-1158-6 10.1111/j.1365-2133.1954.tb12598.x 10.1111/jcpe.12947 10.1097/MS9.0000000000000503
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Keywords	Oral cavity OSCC Squamous cell carcinoma RT SCC IMRT PD Kindler syndrome Palliative care KS CR progressive disease complete response oral squamous cell carcinoma intensity-modulated radiation therapy radiation therapy
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Snippet	Kindler syndrome (KS) is an extremely rare subtype of epidermolysis bullosa characterized by blistering of the extremities, progressive polymorphous skin... AbstractKindler syndrome (KS) is an extremely rare subtype of epidermolysis bullosa characterized by blistering of the extremities, progressive polymorphous...
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SubjectTerms	Hematology, Oncology, and Palliative Medicine Kindler syndrome Oral cavity Palliative care Squamous cell carcinoma
Title	Kindler syndrome with intractable squamous cell carcinoma arising in the oral cavity: A case report and review of cases
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