Successful Anti-TNFα Treatment in a Child with Posttransplant Recurrent Focal Segmental Glomerulosclerosis

Leroy, Sylvie; Guigonis, Vincent; Brückner, Dieter; Emal-Aglaé, Violaine; Deschênes, Georges; Bensman, A; Ulinski, Tim

doi:10.1111/j.1600-6143.2009.02550.x

Cited by 50 publications

(26 citation statements)

References 16 publications

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“…Predictable recognition of this group might therefore facilitate accurate prognosis as well as identification of which patients to target for more intense therapy pretransplant, with strict emphasis on better control of nephrotic relapses earlier in their course of disease to avoid secondary steroid resistance. Therapy is also currently limited by a lack of consensus for the optimal management of post-transplant SRNS recurrence, such as ciclosporin versus tacrolimus, peritransplant plasmapheresis, rituximab (anti-CD20), 32 or infliximab/eternacept (anti-TNFa), 33 all strategies that have been used in attempts to control post-transplant disease with varying success.…”

Section: Discussionmentioning

confidence: 99%

Initial Steroid Sensitivity in Children with Steroid-Resistant Nephrotic Syndrome Predicts Post-Transplant Recurrence

Ding

Koziell

McCarthy

et al. 2014

Journal of the American Society of Nephrology

104

102

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Of children with idiopathic nephrotic syndrome, 10%-20% fail to respond to steroids or develop secondary steroid resistance (termed initial steroid sensitivity) and the majority progress to transplantation. Although 30%-50% of these patients suffer disease recurrence after transplantation, with poor long-term outcome, no reliable indicator of recurrence has yet been identified. Notably, the incidence of recurrence after transplantation appears reduced in patients with steroid-resistant nephrotic syndrome (SRNS) due to monogenic disorders. We reviewed 150 transplanted patients with SRNS to identify biomarkers that consistently predict outcome of SRNS after transplantation. In all, 25 children had genetic or familial SRNS and did not experience post-transplant recurrence. We reviewed phenotypic factors, including initial steroid sensitivity, donor type, age, ethnicity, time to ESRD, and time on dialysis, in the remaining 125 children. Of these patients, 57 (45.6%) developed post-transplant recurrence; 26 of 28 (92.9%) patients with initial steroid sensitivity recurred after transplantation, whereas only 26 of 86 (30.2%) patients resistant from the outset recurred (odds ratio, 30; 95% confidence interval, 6.62 to 135.86; P,0.001). We were unable to determine recurrence in two patients (one with initial steroid sensitivity), and nine patients did not receive initial steroids. Our data show that initial steroid sensitivity is highly predictive of post-transplant disease recurrence in this pediatric patient population. Because a pathogenic circulating permeability factor in nephrotic syndrome remains to be confirmed, we propose initial steroid sensitivity as a surrogate marker for post-transplant recurrence.

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Section: Discussionmentioning

confidence: 99%

Initial Steroid Sensitivity in Children with Steroid-Resistant Nephrotic Syndrome Predicts Post-Transplant Recurrence

Ding

Koziell

McCarthy

et al. 2014

Journal of the American Society of Nephrology

104

102

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“…Anti-TNF-a agents Leroy et al [69] Infliximab (3 mg/kg twice monthly) [70] Etanercept (twice weekly) 1 patient (< 18 yr) Partial remission Rituximab ® Pescovitz et al [28] Rituximab ® (6 doses, 375 mg/m 2 ) 1 patient (< 18 yr) Complete remission Hristea et al [74] Complete remission in 3; partial remission in 2; no response in 1 1/7 patients received one dose, 4/7 patients received 2 doses, and 1/7 received 4 doses; 1/7 patients experienced a severe reaction during first infusion and was excluded from the analysis Tsagalis et al [79] Rituximab ® (1 g, 2 doses) + PE 4 patients (2 < 18 yr; 2 ≥ 18 yr)…”

Section: Messina M Et Al Focal Segmental Glomerulosclerosis In Renamentioning

confidence: 99%

“…At the present time, very few cases of FSGS have been treated with anti-TNF-a agents. Leroy et al [69] reported a favorable outcome (complete remission) for a 15-year-old patient with recurrent FSGS that was presumably resistant to other treatments (increased immunosuppressant dose, PE, intravenous immunoglobulin, high dose steroids, CyA, and CYC) after administration of an anti-TNF-a blocker (firstly infliximab, then etanercept). Bitzan et al [70] showed that plasmapheresis effluent or fresh plasma (obtained from a child with recurrent FSGS and from two children with primary FSGS) caused cytoskeleton disturbance on podocyte culture.…”

Section: Anti-tumor Necrosis Factor-alpha Agentsmentioning

confidence: 99%

Update on the treatment of focal segmental glomerulosclerosis in renal transplantation

Messina¹,

Gallo²,

Mella³

et al. 2016

WJT

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Focal segmental glomerulosclerosis (FSGS) represents one of the most severe glomerular diseases, with frequent progression to end-stage renal disease and a high rate of recurrence in renal allografts (30%-50%). Recurrent FSGS portends a negative outcome, with the hazard ratio of graft failure being two-fold higher then that of other glomerulonephritis. Two patterns of clinical presentations are observed: Early recurrence, which is characterized by massive proteinuria within hours to days after implantation of the renal graft, and late recurrence, which occurs several months or years after the transplantation. Many clinical conditions have been recognized as risk factors for recurrence, including younger age, rapid progression of the disease to end-stage renal disease on native kidneys, and loss of previous renal allografts due to recurrence. However, much less is known about the incidence and risk factors of the so-called "de novo " type of FSGS, for which sufferers are transplanted patients without disease on native kidneys; but, rapid development of allograft failure is frequently observed. Management of both forms is challenging, and none of the approaches proposed to date have been demonstrated as consistently beneficial or effective. In the present review we report an update on the available therapeutic strategies for FSGS in renal transplantation within the context of a critical overview of the current literature.

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“…He finally went into remission with infliximab. In 2009, Leroy et al [7] reported an interesting case. They encountered a boy with focal segmental glomerular sclerosis who relapsed after renal transplantation.…”

Section: Discussionmentioning

confidence: 97%

Long-term remission of nephrotic syndrome with etanercept for concomitant juvenile idiopathic arthritis

et al. 2010

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Etanercept is a tumor necrosis factor (TNF)-alpha inhibitor that has been applied beneficially for juvenile idiopathic arthritis (JIA). We experienced long-term remission of nephrotic syndrome (NS) in a boy treated with etanercept, which was initially used for concomitant JIA. He developed NS at age 3 years 7 months and had mostly been treated with cyclosporine because of steroid dependency and frequent relapses. Cyclosporine was halted at 10 years 7 months because of nephrotoxicity, and he was subsequently treated with mizoribine. However, he had three relapses in the first year and developed JIA at 11 years 7 months. He was treated with sulfasalazine, methotrexate, and prednisolone, but his arthritis persisted. Etanercept was started at 12 years 3 months. Thereafter, his arthritis went into complete remission. Surprisingly, he has remained relapse-free for both NS and JIA for more than 3 years with etanercept and mizoribine. It is difficult to know whether the NS remission after initiating etanercept was coincidental. However, there are many reports of increased TNF-alpha or soluble TNF-alpha receptor in NS relapse. To date, there are two reports of the efficacy of TNF-alpha inhibitors against NS. It is possible that TNF-alpha inhibitors may have potential as therapeutic agents for NS.

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Successful Anti-TNFα Treatment in a Child with Posttransplant Recurrent Focal Segmental Glomerulosclerosis

Abstract: Posttransplant recurrence of focal and segmental glomulosclerosis (FSGS) occurs in

Cited by 50 publications

References 16 publications

Initial Steroid Sensitivity in Children with Steroid-Resistant Nephrotic Syndrome Predicts Post-Transplant Recurrence

Initial Steroid Sensitivity in Children with Steroid-Resistant Nephrotic Syndrome Predicts Post-Transplant Recurrence

Update on the treatment of focal segmental glomerulosclerosis in renal transplantation

Long-term remission of nephrotic syndrome with etanercept for concomitant juvenile idiopathic arthritis

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