Literature DB >> 30715674

Three months interval therapy of Eculizumab in a patient with atypical hemolytic uremic syndrome with hybrid CFHR1/CFH gene.

Sami Alobaidi1,2, Ammar AlDabbagh3, Amany Alamoudi3, Murad Almowarey3, Ahmed Akl3,4.   

Abstract

Pregnancy-associated atypical hemolytic uremic syndrome (P-aHUS) is a rare condition. It is characterized by very high maternal mortality and morbidity. Most cases of P-aHUS (79%) manifest in the postpartum period; this is probably due to the complement's involvement in aHUS pathogenesis. Eculizumab is approved for aHUS treatment, but its use is limited due to cost, unknown duration of treatment, and vague dose intervals to keep patients in remission. In this case report, we present a 26-year-old female with P-aHUS with hybrid CFHR1/CFH gene. Eculizumab was initiated after 5 weeks of being on hemodialysis and plasmapheresis sessions. Full remission successfully achieved after 6th dose of Eculizumab, within 13 weeks of onset of aHUS. Due to financial issues and inability to financially cover the cost, Eculizumab was set in hold. Within 6 months, she suffered recurrence of the disease and Eculizumab was re-instated. After re-inducing full remission, the patient was switched to Eculizumab every 3 months instead of the recommended manufacture dose interval of every 2 weeks. We followed this patient for 3 years and she continued to be in remission based on clinical and laboratory data. In conclusion, achievement of successful and maintenance of remission of P-aHUS in this patient who had limited access to Eculizumab raise the attention of the efficacy of Eculizumab at longer time intervals. However, it is time to consider conducting a long-term study to learn about the safety and efficacy of this approach, which may have a major financial advantage for patients.

Entities:  

Keywords:  Complement dysregulation nephropathy; Eculizumab; Pregnancy-aHUS; aHUS

Mesh:

Substances:

Year:  2019        PMID: 30715674      PMCID: PMC6450989          DOI: 10.1007/s13730-019-00383-7

Source DB:  PubMed          Journal:  CEN Case Rep        ISSN: 2192-4449


  13 in total

Review 1.  Liver-kidney transplantation to cure atypical hemolytic uremic syndrome.

Authors:  Jeffrey M Saland; Piero Ruggenenti; Giuseppe Remuzzi
Journal:  J Am Soc Nephrol       Date:  2008-12-17       Impact factor: 10.121

2.  Eculizumab in pregnancy-associated atypical hemolytic uremic syndrome: insights for optimizing management.

Authors:  Erika De Sousa Amorim; Miquel Blasco; Luis Quintana; Manel Sole; Santiago Rodríguez de Cordoba; Josep Maria Campistol
Journal:  J Nephrol       Date:  2015-02-25       Impact factor: 3.902

3.  Pathogenic Variants in Complement Genes and Risk of Atypical Hemolytic Uremic Syndrome Relapse after Eculizumab Discontinuation.

Authors:  Fadi Fakhouri; Marc Fila; François Provôt; Yahsou Delmas; Christelle Barbet; Valérie Châtelet; Cédric Rafat; Mathilde Cailliez; Julien Hogan; Aude Servais; Alexandre Karras; Raifah Makdassi; Feriell Louillet; Jean-Philippe Coindre; Eric Rondeau; Chantal Loirat; Véronique Frémeaux-Bacchi
Journal:  Clin J Am Soc Nephrol       Date:  2016-10-31       Impact factor: 8.237

4.  Terminal Complement Inhibitor Eculizumab in Adult Patients With Atypical Hemolytic Uremic Syndrome: A Single-Arm, Open-Label Trial.

Authors:  Fadi Fakhouri; Maryvonne Hourmant; Josep M Campistol; Spero R Cataland; Mario Espinosa; A Osama Gaber; Jan Menne; Enrico E Minetti; François Provôt; Eric Rondeau; Piero Ruggenenti; Laurent E Weekers; Masayo Ogawa; Camille L Bedrosian; Christophe M Legendre
Journal:  Am J Kidney Dis       Date:  2016-03-21       Impact factor: 8.860

5.  Pregnancy-associated hemolytic uremic syndrome revisited in the era of complement gene mutations.

Authors:  Fadi Fakhouri; Lubka Roumenina; François Provot; Marion Sallée; Sophie Caillard; Lionel Couzi; Marie Essig; David Ribes; Marie-Agnès Dragon-Durey; Frank Bridoux; Eric Rondeau; Veronique Frémeaux-Bacchi
Journal:  J Am Soc Nephrol       Date:  2010-03-04       Impact factor: 10.121

Review 6.  Diagnostic criteria for atypical hemolytic uremic syndrome proposed by the Joint Committee of the Japanese Society of Nephrology and the Japan Pediatric Society.

Authors:  Toshihiro Sawai; Masaomi Nangaku; Akira Ashida; Rika Fujimaru; Hiroshi Hataya; Yoshihiko Hidaka; Shinya Kaname; Hirokazu Okada; Waichi Sato; Takashi Yasuda; Yoko Yoshida; Yoshihiro Fujimura; Motoshi Hattori; Shoji Kagami
Journal:  Pediatr Int       Date:  2014-02       Impact factor: 1.524

7.  Atypical hemolytic uremic syndrome: a clinical conundrum.

Authors:  Prabesh Bajracharya; Amrish Jain; Rossana Baracco; Tej K Mattoo; Gaurav Kapur
Journal:  Pediatr Nephrol       Date:  2016-05-02       Impact factor: 3.714

Review 8.  [Atypical hemolytic uremic syndrome (aHUS): new insights into pathogenesis leading to novel therapeutic approaches].

Authors:  Andreas D Kistler
Journal:  Praxis (Bern 1994)       Date:  2016-03-30

9.  Association among Complement Factor H Autoantibodies, Deletions of CFHR, and the Risk of Atypical Hemolytic Uremic Syndrome.

Authors:  Hong Jiang; Meng-Nan Fan; Min Yang; Chao Lu; Ming Zhang; Xiao-Hong Liu; Le Ma
Journal:  Int J Environ Res Public Health       Date:  2016-12-05       Impact factor: 3.390

10.  Improved renal recovery in patients with atypical hemolytic uremic syndrome following rapid initiation of eculizumab treatment.

Authors:  Johan Vande Walle; Yahsou Delmas; Gianluigi Ardissino; Jimmy Wang; John F Kincaid; Herman Haller
Journal:  J Nephrol       Date:  2016-03-19       Impact factor: 3.902

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  1 in total

1.  Atypical hemolytic uremic syndrome after childbirth: a case report.

Authors:  Hong Sang Choi; Jae Won Yun; Hee-Jin Kim; Doyeun Oh; Nah Ihm Kim; Chang Seong Kim; Seong Kwon Ma; Soo Wan Kim; Eun Hui Bae
Journal:  Ann Transl Med       Date:  2021-01
  1 in total

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