Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report
Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively...
Ausführliche Beschreibung
Autor*in: |
Ryoichi Goto [verfasserIn] Norio Kawamura [verfasserIn] Masaaki Watanabe [verfasserIn] Yasuyuki Koshizuka [verfasserIn] Souichi Shiratori [verfasserIn] Momoko Ara [verfasserIn] Shohei Honda [verfasserIn] Tomoko Mitsuhashi [verfasserIn] Yoshihiro Matsuno [verfasserIn] Tsuyoshi Shimamura [verfasserIn] Akinobu Taketomi [verfasserIn] |
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Format: |
E-Artikel |
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Sprache: |
Englisch |
Erschienen: |
2020 |
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Schlagwörter: |
Indolent T cell lymphoproliferative disorder |
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Übergeordnetes Werk: |
In: Surgical Case Reports - SpringerOpen, 2015, 6(2020), 1, Seite 7 |
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Übergeordnetes Werk: |
volume:6 ; year:2020 ; number:1 ; pages:7 |
Links: |
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DOI / URN: |
10.1186/s40792-020-00904-y |
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Katalog-ID: |
DOAJ052171051 |
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520 | |a Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. | ||
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10.1186/s40792-020-00904-y doi (DE-627)DOAJ052171051 (DE-599)DOAJbf49a3f4499740fa81cbe935f2109546 DE-627 ger DE-627 rakwb eng RD1-811 Ryoichi Goto verfasserin aut Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report 2020 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. Indolent T cell lymphoproliferative disorder Post-transplant lymphoproliferative disorder Living donor liver transplantation Surgery Norio Kawamura verfasserin aut Masaaki Watanabe verfasserin aut Yasuyuki Koshizuka verfasserin aut Souichi Shiratori verfasserin aut Momoko Ara verfasserin aut Shohei Honda verfasserin aut Tomoko Mitsuhashi verfasserin aut Yoshihiro Matsuno verfasserin aut Tsuyoshi Shimamura verfasserin aut Akinobu Taketomi verfasserin aut In Surgical Case Reports SpringerOpen, 2015 6(2020), 1, Seite 7 (DE-627)818040475 (DE-600)2809613-7 21987793 nnns volume:6 year:2020 number:1 pages:7 https://doi.org/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/article/bf49a3f4499740fa81cbe935f2109546 kostenfrei http://link.springer.com/article/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/toc/2198-7793 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 6 2020 1 7 |
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10.1186/s40792-020-00904-y doi (DE-627)DOAJ052171051 (DE-599)DOAJbf49a3f4499740fa81cbe935f2109546 DE-627 ger DE-627 rakwb eng RD1-811 Ryoichi Goto verfasserin aut Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report 2020 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. Indolent T cell lymphoproliferative disorder Post-transplant lymphoproliferative disorder Living donor liver transplantation Surgery Norio Kawamura verfasserin aut Masaaki Watanabe verfasserin aut Yasuyuki Koshizuka verfasserin aut Souichi Shiratori verfasserin aut Momoko Ara verfasserin aut Shohei Honda verfasserin aut Tomoko Mitsuhashi verfasserin aut Yoshihiro Matsuno verfasserin aut Tsuyoshi Shimamura verfasserin aut Akinobu Taketomi verfasserin aut In Surgical Case Reports SpringerOpen, 2015 6(2020), 1, Seite 7 (DE-627)818040475 (DE-600)2809613-7 21987793 nnns volume:6 year:2020 number:1 pages:7 https://doi.org/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/article/bf49a3f4499740fa81cbe935f2109546 kostenfrei http://link.springer.com/article/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/toc/2198-7793 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 6 2020 1 7 |
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10.1186/s40792-020-00904-y doi (DE-627)DOAJ052171051 (DE-599)DOAJbf49a3f4499740fa81cbe935f2109546 DE-627 ger DE-627 rakwb eng RD1-811 Ryoichi Goto verfasserin aut Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report 2020 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. Indolent T cell lymphoproliferative disorder Post-transplant lymphoproliferative disorder Living donor liver transplantation Surgery Norio Kawamura verfasserin aut Masaaki Watanabe verfasserin aut Yasuyuki Koshizuka verfasserin aut Souichi Shiratori verfasserin aut Momoko Ara verfasserin aut Shohei Honda verfasserin aut Tomoko Mitsuhashi verfasserin aut Yoshihiro Matsuno verfasserin aut Tsuyoshi Shimamura verfasserin aut Akinobu Taketomi verfasserin aut In Surgical Case Reports SpringerOpen, 2015 6(2020), 1, Seite 7 (DE-627)818040475 (DE-600)2809613-7 21987793 nnns volume:6 year:2020 number:1 pages:7 https://doi.org/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/article/bf49a3f4499740fa81cbe935f2109546 kostenfrei http://link.springer.com/article/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/toc/2198-7793 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 6 2020 1 7 |
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10.1186/s40792-020-00904-y doi (DE-627)DOAJ052171051 (DE-599)DOAJbf49a3f4499740fa81cbe935f2109546 DE-627 ger DE-627 rakwb eng RD1-811 Ryoichi Goto verfasserin aut Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report 2020 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. Indolent T cell lymphoproliferative disorder Post-transplant lymphoproliferative disorder Living donor liver transplantation Surgery Norio Kawamura verfasserin aut Masaaki Watanabe verfasserin aut Yasuyuki Koshizuka verfasserin aut Souichi Shiratori verfasserin aut Momoko Ara verfasserin aut Shohei Honda verfasserin aut Tomoko Mitsuhashi verfasserin aut Yoshihiro Matsuno verfasserin aut Tsuyoshi Shimamura verfasserin aut Akinobu Taketomi verfasserin aut In Surgical Case Reports SpringerOpen, 2015 6(2020), 1, Seite 7 (DE-627)818040475 (DE-600)2809613-7 21987793 nnns volume:6 year:2020 number:1 pages:7 https://doi.org/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/article/bf49a3f4499740fa81cbe935f2109546 kostenfrei http://link.springer.com/article/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/toc/2198-7793 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 6 2020 1 7 |
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10.1186/s40792-020-00904-y doi (DE-627)DOAJ052171051 (DE-599)DOAJbf49a3f4499740fa81cbe935f2109546 DE-627 ger DE-627 rakwb eng RD1-811 Ryoichi Goto verfasserin aut Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report 2020 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. Indolent T cell lymphoproliferative disorder Post-transplant lymphoproliferative disorder Living donor liver transplantation Surgery Norio Kawamura verfasserin aut Masaaki Watanabe verfasserin aut Yasuyuki Koshizuka verfasserin aut Souichi Shiratori verfasserin aut Momoko Ara verfasserin aut Shohei Honda verfasserin aut Tomoko Mitsuhashi verfasserin aut Yoshihiro Matsuno verfasserin aut Tsuyoshi Shimamura verfasserin aut Akinobu Taketomi verfasserin aut In Surgical Case Reports SpringerOpen, 2015 6(2020), 1, Seite 7 (DE-627)818040475 (DE-600)2809613-7 21987793 nnns volume:6 year:2020 number:1 pages:7 https://doi.org/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/article/bf49a3f4499740fa81cbe935f2109546 kostenfrei http://link.springer.com/article/10.1186/s40792-020-00904-y kostenfrei https://doaj.org/toc/2198-7793 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 6 2020 1 7 |
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Ryoichi Goto @@aut@@ Norio Kawamura @@aut@@ Masaaki Watanabe @@aut@@ Yasuyuki Koshizuka @@aut@@ Souichi Shiratori @@aut@@ Momoko Ara @@aut@@ Shohei Honda @@aut@@ Tomoko Mitsuhashi @@aut@@ Yoshihiro Matsuno @@aut@@ Tsuyoshi Shimamura @@aut@@ Akinobu Taketomi @@aut@@ |
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Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. 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Post-transplant indolent T cell lymphoproliferative disorder in living donor liver transplantation: a case report |
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Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. |
abstractGer |
Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. |
abstract_unstemmed |
Abstract Background Post-transplant lymphoproliferative disorder (PTLD) of T cell type has been rarely reported. Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required. |
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Accurate diagnosis of this life-threatening rare form of PTLD is important for the treatment strategy. Case presentation A 7-year-old boy had severe diarrhea and weight loss progressively at 7 years post-living donor liver transplantation (LDLT) for biliary atresia. Endoscopy in the gastrointestinal (GI) tract revealed multiple erosions and ulcer lesions with prominent intraepithelial lymphocytosis in the duodenum and terminal ileum. Immunohistochemical examination demonstrated that these accumulated lymphocytes mainly comprised small- to medium-sized T cells expressing CD3, CD4, CD5, CD7, and CD103, but lacking CD8, CD56, and Epstein-Barr virus-encoded small RNAs. In addition, T cell receptor β gene rearrangement was detected by polymerase chain reaction analysis. Comprehensively, the lesions were best interpreted as post-transplant indolent T cell lymphoproliferative disorder (LPD) of the intestine. Clinical remission was achieved by reducing the immunosuppressant. Conclusion A rarely reported indolent type of T cell LPD in post-LDLT was diagnosed by direct inspection and histological investigation. Although the histological classification and therapeutic strategy for post-transplant indolent T cell LPD have not been established, reducing immunosuppression allowed complete remission in our case. To prevent the incidence of PTLD and de novo malignancy, developing a methodology to set a proper dose of immunosuppressant is required.</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Indolent T cell lymphoproliferative disorder</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Post-transplant lymphoproliferative disorder</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Living donor liver transplantation</subfield></datafield><datafield tag="653" ind1=" " ind2="0"><subfield code="a">Surgery</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Norio Kawamura</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Masaaki Watanabe</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield 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