Cidofovir for BK Virus–Associated Hemorrhagic Cystitis: A Retrospective Study

Cesaro, Simone; Hirsch, Hans H.; Faraci, Maura; Owoc‐Lempach, Joanna; Beltrame, Anna; Tendas, Andrea; Baltadakis, Ioannis; Dalle, Jean-Hughes; Koç, Yener; Toporski, Jacek; Styczyński, Jan; Yeşilipek, M. Akif; Heinz, Werner; Caniglia, Maurizio; Rascon, Jelena; Fauser, Axel A.; Michallet, Mauricette; López-Corral, Lucía; Neuburger, Stefan; Tridello, Gloria; Einsele, Herman

doi:10.1086/599829

Cited by 106 publications

(118 citation statements)

References 37 publications

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“…18 In a single-center study on pediatric patients and in a retrospective study on adult and pediatric patients, the lack of recovery from HC has been associated with both a higher transplant-related mortality (72% vs 40%), and lower survival (43% vs 52%). 3,14 One issue still debated is the optimal dosage of CDV. Bernhoff et al…”

Section: Discussionmentioning

confidence: 99%

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Relationship between clinical and BK virological response in patients with late hemorrhagic cystitis treated with cidofovir: a retrospective study from the European Group for Blood and Marrow Transplantation

Cesaro

Pillon

Tridello

et al. 2012

Bone Marrow Transplant

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To investigate the relationship between clinical response and modification of BK viremia, we assessed retrospectively 32 cases of hemorrhagic cystitis (HC) after allogeneic hematopoietic SCT that were treated with i.v. cidofovir (CDV). They were 22 men (69%) and 10 women (31%) with a median age of 24 years, range 3-62. The median number of CDV doses was 3, range 1-8, and the treatment lasted for a median of 3 weeks, range 1-10. Clinical improvement of HC was observed in 27 patients (84%). In 12 of 32 episodes (37.5%), BK viremia was determined before every CDV administration and a complete clinical response was observed in 10 of 12 patients (83%), the reduction of BK viremia load being X1 log by 2 weeks after starting CDV. Nephrotoxicity related to CDV was observed in nine patients. Among 26 patients with 100-day follow-up, 4 of 4 patients who had a complete clinical response by 30 days were alive vs 16 of 22 (73%) who did not have the resolution of HC in this time frame. We conclude that in patients with HC, the response to CDV treatment is usually associated with a significant reduction of BK viremia load.

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Section: Discussionmentioning

confidence: 99%

“…6 Indeed, limited data are available on virological response during CDV treatment. 13,14 For these reasons, we decided to extend the data collection on CDV-treated HC among EBMT centers. The aim was to further investigate the relationship between clinical response and BK viremia in CDV-treated patients.…”

Section: Introductionmentioning

confidence: 99%

Relationship between clinical and BK virological response in patients with late hemorrhagic cystitis treated with cidofovir: a retrospective study from the European Group for Blood and Marrow Transplantation

Cesaro

Pillon

Tridello

et al. 2012

Bone Marrow Transplant

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“…No significant differences were found in the incidence of IRM nor overall NRM between CB and BM/PB recipients. The cumulative incidence of IRM was 14% at 100 days for both groups, while the 1-year IRM was 30% (95%CI ) vs 27% (95%CI (22)(23)(24)(25)(26)(27)(28)(29)(30)(31)(32)(33)(34)(35)) at 1 year, and 33% (95%CI (21-46)) vs 30% (95%CI (25-34)) at 5 years for CB and BM/PB, respectively (P = 0.4). Regarding NRM, the cumulative incidence was 23% at 100 days for both groups, 1-year NRM was 42 vs 37%, and 5-year NRM was 45 vs 43% in CB and BM/PB recipients, respectively (P = 0.6) ( Figure 2).…”

Section: Patientsmentioning

confidence: 92%

“…20 Other non-CMV severe viral infections considered in this study included (i) EBV infection determined by exponential increase of positive EBV PCR, with or without evidence of post-transplant lymphoproliferative disease (EBV-PTLD); (ii) disseminated VZV infection, affecting two or more skin dermatomes and/or visceral involvement; (iii) Human Herpesvirus 6 as cause of encephalitis, diagnosed by positive PCR from cerebrospinal fluid; (iv) Adenovirus (ADV) disease, diagnosed when ADV was identified in any sample by immunohistochemistry; and (v) pneumonia due to a community respiratory virus (CRV) or BK polyomavirus-related hemorrhagic cystitis, as defined by accepted criteria. [21][22][23] However, it has to be remarked that monitoring of ADV, Human Herpesvirus 6 and toxoplasma was not extensively used in all centers. EBV monitoring was introduced around 2005-2006.…”

Section: Definitionsmentioning

confidence: 99%

Fungal and viral infections after allogeneic hematopoietic transplantation from unrelated donors in adults: improving outcomes over time

Parody

Martino

Cámara

et al. 2014

Bone Marrow Transplant

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Umbilical cord blood (CB) is increasingly used as an alternative source of stem cells in adult unrelated transplantation. Although registry studies report similar overall outcomes in comparison with BM/PB, comparative studies focusing on severe infections and infection-RM (IRM) with a long follow-up are scarce. A total of 434 consecutive unrelated transplants (1997)(1998)(1999)(2000)(2001)(2002)(2003)(2004)(2005)(2006)(2007)(2008)(2009) were retrospectively analyzed to compare overall outcomes, incidence and risk factors of severe viral and invasive fungal infections in CB (n = 65) vs BM/PB recipients (n = 369). The 5-year OS was 38 vs 43%, respectively (P = 0.2). CB transplantation (CBT) was associated with a higher risk of invasive aspergillosis (100-days-cumulative incidence 16 vs 6%, P = 0.04) and CMV infection without differences in RM. No statistically significant differences were found regarding NRM (NRM of 38% in CB vs 37% in BM/PB at 1 year) nor IRM (30% in CB vs 27% in BM/PB at 1 year). In the overall population, NRM and IRM improved in more recent years. In adults who receive a single CBT, the risk of severe infections is increased when compared with unrelated BM/PB recipients, but mortality from infections is similar, leading to similar NRM and survival. INTRODUCTIONInfections are a major concern after allo-HSCT, and the main cause of mortality in at least 35-45% of deaths.1-7 The use of unrelated donors (UD) in adults increases the risk of severe infectious complications, although recent reports have found similar survival with matched sibling and UD allo-HSCT in the setting of specific diseases, such as AML. 8,9 Moreover, the use of cord blood (CB) as an alternative source of HSC in adults who lack an HLA-matched UD BM or PB has shown to be a valid alternative for an allo-HSCT. Recent studies have shown that similar NRM and OS can be achieved with CB if an accurate choice of the CB unit(s) is made, in comparison with other alternative donor sources.10-15 However, slow hematologic and immune recovery after CB transplantation (CBT) still remains a concern, and a high rate of serious pre and post-engraftment infections has been reported. However, few studies have actually compared the incidence of severe infections and infection-RM (IRM) of adult CBT recipients as opposed to UD BMT/PBT.For this purpose, and as a continuation of a previously published study by our group, 16 we performed a multicenter 12-year retrospective study to analyze the impact of the source of HSC for UD transplantation, including single CBT and BM/PBT on overall transplantation outcomes, especially. We also analyzed the

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“…Because of full-matched HLA compatibility, anti-HLA antibodies were not tested. A major early complication was severe BK virus-related hemorrhagic cystitis (grade 4) that was managed with antiviral therapy with low-dose cidofovir (1 mg/kg twice weekly), 10 bladder irrigation with hyaluronate-based drug, and analgesic therapy. After initial mixed chimerism, neutrophil engraftment, achieved on day +23, was followed by complete secondary rejection on day +41.…”

Section: First Hematopoietic Stem Cell Transplantmentioning

confidence: 99%

Successful Second Unrelated Donor Hematopoietic Stem Cell Transplant in a Patient With Dyskeratosis Congenital After First Graft Rejection

Giardino¹,

Faraci²,

Lanino³

et al. 2023

Exp Clin Transplant

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Dyskeratosis congenita is a rare congenital telomeropathy characterized by cutaneous and nail dystrophy, oral leukoplakia, and bone marrow failure. Pulmonary fibrosis and cancers are late manifestations. Allogeneic hematopoietic stem cell transplant represents the only cure for those with bone marrow failure with this disease, but outcomes reported are overall poor, with organ toxicities, graft failure, and graft-versus-host disease as main issues. Although reduced intensity conditioning regimens seem to be related to better outcomes, a standard regimen for dyskeratosis congenita has never been defined. Here, we report a successful long-term outcome of an 8-year-old girl with dyskeratosis congenita who received 2 consecutive allogeneic hematopoietic stem cell transplants from different unrelated donors, because of rejection after the first one, both conditioned with fludarabine-based reduced intensity conditioning regimen. The second transplant was complicated by severe hemorrhagic cystitis and acute grade IV graft-versus-host disease in the early phase and mild chronic graft-versus-host disease and ureteral stenosis in the late phase. This experience confirms that dyskeratosis congenita is at high risk for transplant-related morbidity but that a fludarabine-based reduced intensity conditioning regimen is a safe and feasible option as a preparative regimen, as shown here in a second transplant after first graft rejection. To reduce the risk of graft-versushost disease, more effective prophylaxis schedules should be chosen in cases of unrelated donor, and haploidentical hematopoietic stem cell transplant with in vitro α/β+ and CD19+ depletion should be considered. Key words: Allogeneic hematopoietic stem cell transplant, Bone marrow failure, Reduced intensity conditioning regimen IntroductionDyskeratosis congenita (DC) is a rare heterogeneous inherited disorder, characterized by abnormal skin pigmentation, oral leukoplakia, nail dystrophy, and bone marrow failure. 1,2 Cancer predisposition with increased risk of squamous cell carcinoma and hepatolymphoid neoplasms and pulmonary fibrosis represent typical severe features of the disease. 3 About 60% to 70% of affected patients carry a mutation in telomerase genes (DKC1, TERC, TERT, NOP10, and NHP2), which are required to synthesize the tandem repeats of the TTAGGG sequencing or of the telosome/shelterin complex proteins (TINF2) acting at chromosome ends. 4 Their defect leads to telomere shortness and consequent chromosomal instability, with premature stem cell exhaustion, tissue failure, and high tissue sensitivity to toxic damage. 5 Moreover, high sensibility to oxidative stress has been demonstrated in DC-cultured cells. 6 Androgen therapy has been reported to improve bone marrow function 7 ; however, allogeneic hematopoietic stem cell transplant (HSCT) still represents the only curative option for DC-related bone marrow failure.Few reports on HSCT for DC have been published so far. Although overall survival has progressively improved over the years with ...

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Cidofovir for BK Virus–Associated Hemorrhagic Cystitis: A Retrospective Study

Abstract: Cidofovir may be a potentially effective therapy for BKV-HC, but evidence supporting its use requires randomized controlled trials.

Cited by 106 publications

References 37 publications

Relationship between clinical and BK virological response in patients with late hemorrhagic cystitis treated with cidofovir: a retrospective study from the European Group for Blood and Marrow Transplantation

Relationship between clinical and BK virological response in patients with late hemorrhagic cystitis treated with cidofovir: a retrospective study from the European Group for Blood and Marrow Transplantation

Fungal and viral infections after allogeneic hematopoietic transplantation from unrelated donors in adults: improving outcomes over time

Successful Second Unrelated Donor Hematopoietic Stem Cell Transplant in a Patient With Dyskeratosis Congenital After First Graft Rejection

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