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Posttransplantation lymphoproliferative disorder after pediatric solid organ transplantation: experiences of 20 years in a single center

View Article: PubMed Central - PubMed

ABSTRACT

Purpose: To evaluate the clinical spectrum of posttransplantation lymphoproliferative disorder (PTLD) after solid organ transplantation (SOT) in children.

Methods: We retrospectively reviewed the medical records of 18 patients with PTLD who underwent liver (LT) or kidney transplantation (KT) between January 1995 and December 2014 in Seoul National University Children's Hospital.

Results: Eighteen patients (3.9% of pediatric SOTs; LT:KT, 11:7; male to female, 9:9) were diagnosed as having PTLD over the last 2 decades (4.8% for LT and 2.9% for KT). PTLD usually presented with fever or gastrointestinal symptoms in a median period of 7 months after SOT. Eight cases had malignant lesions, and all the patients except one had evidence of Epstein-Barr virus (EBV) involvement, assessed by using in situ hybridization of tumor tissue or EBV viral load quantitation of blood. Remission was achieved in all patients with reduction of immunosuppression and/or rituximab therapy or chemotherapy, although 1 patient had allograft kidney loss and another died from complications of chemotherapy. The first case of PTLD was encountered after the introduction of tacrolimus for pediatric SOT in 2003. The recent increase in PTLD incidence in KT coincided with modification of clinical practice since 2012 to increase the tacrolimus trough level.

Conclusion: While the outcome was favorable in that all patients achieved complete remission, some patients still had allograft loss or mortality. To prevent PTLD and improve its outcome, monitoring for EBV infection is essential, which would lead to appropriate modification of immunosuppression and enhanced surveillance for PTLD.

No MeSH data available.


Related in: MedlinePlus

Treatment and response. RI, reduction of immunosuppression; Chemo, chemotherapy; RTX, rituximab; GCV, ganciclovir; NR, no response; CR, complete remission; R-ICE, rituximab, ifosfamide, carboplatin, etoposide. *CCG 106B protocol. (GCV) GCV was used for CMV coinfection. †Expired from complication of chemotherapy (sepsis) after remission of posttransplantation lymphoproliferative disorder (PTLD). ‡Graft loss during chemotherapy after remission of PTLD.
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Figure 2: Treatment and response. RI, reduction of immunosuppression; Chemo, chemotherapy; RTX, rituximab; GCV, ganciclovir; NR, no response; CR, complete remission; R-ICE, rituximab, ifosfamide, carboplatin, etoposide. *CCG 106B protocol. (GCV) GCV was used for CMV coinfection. †Expired from complication of chemotherapy (sepsis) after remission of posttransplantation lymphoproliferative disorder (PTLD). ‡Graft loss during chemotherapy after remission of PTLD.

Mentions: Upon recognition of PTLD, maintenance immunosuppression was reduced in all patients, similar to what was reported in the literature1011), and additional treatments were applied as appropriate (Table 3, Fig. 2). Surgery was considered as the primary treatment for localized lesion and primary tumors were removed by surgery in one third of the patients (n=6, 33.3%). Rituximab (RTX) was administered in every case since 2013 and one malignant case in 2006 (72.2%). Before RTX was established as the first line treatment for PTLD, ganciclovir (GCV) had been tried as an initial treatment of PTLD (n=6). GCV was also used along with RTX when there was CMV coinfection (patient numbers 7 and 16). Chemotherapy was administered for malignant PTLD (n=8). The first line chemotherapy in our institute was CCG 106B protocol12) (prednisolone, vincristine, daunomycin, cyclophosphamide, and intrathecal chemotherapy of cytarabine, hydrocortisone, and methotrexate) along with RTX.


Posttransplantation lymphoproliferative disorder after pediatric solid organ transplantation: experiences of 20 years in a single center
Treatment and response. RI, reduction of immunosuppression; Chemo, chemotherapy; RTX, rituximab; GCV, ganciclovir; NR, no response; CR, complete remission; R-ICE, rituximab, ifosfamide, carboplatin, etoposide. *CCG 106B protocol. (GCV) GCV was used for CMV coinfection. †Expired from complication of chemotherapy (sepsis) after remission of posttransplantation lymphoproliferative disorder (PTLD). ‡Graft loss during chemotherapy after remission of PTLD.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC5383637&req=5

Figure 2: Treatment and response. RI, reduction of immunosuppression; Chemo, chemotherapy; RTX, rituximab; GCV, ganciclovir; NR, no response; CR, complete remission; R-ICE, rituximab, ifosfamide, carboplatin, etoposide. *CCG 106B protocol. (GCV) GCV was used for CMV coinfection. †Expired from complication of chemotherapy (sepsis) after remission of posttransplantation lymphoproliferative disorder (PTLD). ‡Graft loss during chemotherapy after remission of PTLD.
Mentions: Upon recognition of PTLD, maintenance immunosuppression was reduced in all patients, similar to what was reported in the literature1011), and additional treatments were applied as appropriate (Table 3, Fig. 2). Surgery was considered as the primary treatment for localized lesion and primary tumors were removed by surgery in one third of the patients (n=6, 33.3%). Rituximab (RTX) was administered in every case since 2013 and one malignant case in 2006 (72.2%). Before RTX was established as the first line treatment for PTLD, ganciclovir (GCV) had been tried as an initial treatment of PTLD (n=6). GCV was also used along with RTX when there was CMV coinfection (patient numbers 7 and 16). Chemotherapy was administered for malignant PTLD (n=8). The first line chemotherapy in our institute was CCG 106B protocol12) (prednisolone, vincristine, daunomycin, cyclophosphamide, and intrathecal chemotherapy of cytarabine, hydrocortisone, and methotrexate) along with RTX.

View Article: PubMed Central - PubMed

ABSTRACT

Purpose: To evaluate the clinical spectrum of posttransplantation lymphoproliferative disorder (PTLD) after solid organ transplantation (SOT) in children.

Methods: We retrospectively reviewed the medical records of 18 patients with PTLD who underwent liver (LT) or kidney transplantation (KT) between January 1995 and December 2014 in Seoul National University Children's Hospital.

Results: Eighteen patients (3.9% of pediatric SOTs; LT:KT, 11:7; male to female, 9:9) were diagnosed as having PTLD over the last 2 decades (4.8% for LT and 2.9% for KT). PTLD usually presented with fever or gastrointestinal symptoms in a median period of 7 months after SOT. Eight cases had malignant lesions, and all the patients except one had evidence of Epstein-Barr virus (EBV) involvement, assessed by using in situ hybridization of tumor tissue or EBV viral load quantitation of blood. Remission was achieved in all patients with reduction of immunosuppression and/or rituximab therapy or chemotherapy, although 1 patient had allograft kidney loss and another died from complications of chemotherapy. The first case of PTLD was encountered after the introduction of tacrolimus for pediatric SOT in 2003. The recent increase in PTLD incidence in KT coincided with modification of clinical practice since 2012 to increase the tacrolimus trough level.

Conclusion: While the outcome was favorable in that all patients achieved complete remission, some patients still had allograft loss or mortality. To prevent PTLD and improve its outcome, monitoring for EBV infection is essential, which would lead to appropriate modification of immunosuppression and enhanced surveillance for PTLD.

No MeSH data available.


Related in: MedlinePlus