Molecular Adsorbents Recirculating System dialysis in children with cholestatic pruritus

Betti, Silvia; Schaefer, Franz; Wittmer, Dieter; Engelmann, Guido; Wenning, Daniel; Schmitt, Claus Peter

doi:10.1007/s00467-011-2058-8

Cited by 28 publications

(12 citation statements)

References 40 publications

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“…59 Further, it has been shown that bile acids (that increase in content during liver disease) can regulate mast cell histamine release 60 and may also be a source of increased pruritus. 61,62 This suggests that manipulation of mast cell histamine by cromolyn sodium or bile acid therapy might also be an option for patients suffering from cholangiopathies or other liver diseases.…”

Section: Discussionmentioning

confidence: 99%

Inhibition of mast cell-derived histamine secretion by cromolyn sodium treatment decreases biliary hyperplasia in cholestatic rodents

Kennedy

Hargrove

Graf

et al. 2014

Laboratory Investigation

143

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Cholangiopathies are characterized by dysregulation of the balance between biliary growth and loss. We have shown that histamine (HA) stimulates biliary growth via autocrine mechanisms. To evaluate the paracrine effects of mast cell (MC) stabilization on biliary proliferation, sham or BDL rats were treated by IP-implanted osmotic pumps filled with saline or cromolyn sodium (24 mg/kg BW/day (inhibits MC histamine release)) for 1 week. Serum, liver blocks and cholangiocytes were collected. Histidine decarboxylase (HDC) expression was measured using real-time PCR in cholangiocytes. Intrahepatic bile duct mass (IBDM) was evaluated by IHC for CK-19. MC number was determined using toluidine blue staining and correlated to IBDM. Proliferation was evaluated by PCNA expression in liver sections and purified cholangiocytes. We assessed apoptosis using real-time PCR and IHC for BAX. Expression of MC stem factor receptor, c-kit, and the proteases chymase and tryptase were measured by real-time PCR. HA levels were measured in serum by EIA. In vitro, MCs and cholangiocytes were treated with 0.1% BSA (basal) or cromolyn (25 mM) for up to 48 h prior to assessing HDC expression, HA levels and chymase and tryptase expression. Supernatants from MCs treated with or without cromolyn were added to cholangiocytes before measuring (i) proliferation by MTT assays, (ii) HDC gene expression by real-time PCR and (iii) HA release by EIA. In vivo, cromolyn treatment decreased BDL-induced: (i) IBDM, MC number, and biliary proliferation; (ii) HDC and MC marker expression; and (iii) HA levels. Cromolyn treatment increased cholangiocyte apoptosis. In vitro, cromolyn decreased HA release and chymase and tryptase expression in MCs but not in cholangiocytes. Cromolyn-treated MC supernatants decreased biliary proliferation and HA release. These studies provide evidence that MC histamine is key to biliary proliferation and may be a therapeutic target for the treatment of cholangiopathies.

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Section: Discussionmentioning

confidence: 99%

Inhibition of mast cell-derived histamine secretion by cromolyn sodium treatment decreases biliary hyperplasia in cholestatic rodents

Kennedy

Hargrove

Graf

et al. 2014

Laboratory Investigation

143

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“…Previous reports on extracorporeal treatments for the management of refractory pruritus show encouraging results, complications such as bleeding or thrombocytopenia are rare but treatment sessions last longer and are performed more often . We prioritized LA among other extracorporeal treatment options based on the good experience in our center with this technique in small pediatric patients, with very good tolerability, rare complications, small extracorporeal volume, short treatment duration, and efficacy.…”

Section: Laboratory Values Under Pharmacological Treatment Under Trementioning

confidence: 99%

Improvement of refractory pruritus after lipoprotein‐apheresis in arthrogryposis‐renal failure‐cholestasis syndrome

Rosales

Muñoz

Madrid

et al. 2017

J of Clinical Apheresis

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Accumulation of bile acids can lead to invalidating pruritus in cholestatic patients. Few reports exist on the influence of lipoprotein-apheresis (LA) on plasma level of total bile acids (tBA). We report of significant decrease in tBA levels and drastic improvement of pruritus in a 5-year-old girl with arthrogryposis-renal failure-cholestasis syndrome. We present LA as a suitable rescue treatment option in therapy-refractory cholestasis-associated pruritus, at least as bridge until a long-term solution such as entero-biliary anastomosis or transplantation is possible.

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“…Most reports on the use of MARS in children are case reports or small case series. [5][6][7][8][9][10] In this issue of Liver Transplantation, Lexmond et al 11 from the Netherlands describe their experience with MARS in treating 20 children in need of high-urgency liver transplantation over the course of 10 years. The authors conclude that MARS therapy was relatively safe in these sick subjects; that serum bilirubin, creatinine, ammonia, and bile acid levels significantly decreased; and that 80% of these selected children survived long enough to undergo liver transplantation.…”

Section: See Article On Page 369mentioning

confidence: 99%

“…No large, prospective, multicenter randomized controlled trial has been performed with non–cell‐based systems in children. Most reports on the use of MARS in children are case reports or small case series . In this issue of Liver Transplantation, Lexmond et al from the Netherlands describe their experience with MARS in treating 20 children in need of high‐urgency liver transplantation over the course of 10 years.…”

mentioning

confidence: 99%

Is the Molecular Adsorbent Recirculating System the Answer for Children With Acute Liver Failure?

Rosenthal

2015

Liver Transpl

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Molecular Adsorbents Recirculating System dialysis in children with cholestatic pruritus

Abstract: MARS dialysis substantially reduces cholestatic pruritus in children refractory to pharmacological treatment.

Cited by 28 publications

References 40 publications

Inhibition of mast cell-derived histamine secretion by cromolyn sodium treatment decreases biliary hyperplasia in cholestatic rodents

Inhibition of mast cell-derived histamine secretion by cromolyn sodium treatment decreases biliary hyperplasia in cholestatic rodents

Improvement of refractory pruritus after lipoprotein‐apheresis in arthrogryposis‐renal failure‐cholestasis syndrome

Is the Molecular Adsorbent Recirculating System the Answer for Children With Acute Liver Failure?

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