Audit of the Use of Regular Haem Arginate Infusions in Patients with Acute Porphyria to Prevent Recurrent Symptoms - PubMed (original) (raw)
Audit of the Use of Regular Haem Arginate Infusions in Patients with Acute Porphyria to Prevent Recurrent Symptoms
Joanne T Marsden et al. JIMD Rep. 2015.
Abstract
The National Acute Porphyria Service (NAPS) provides acute care support and clinical advice for patients in England with active acute porphyria requiring haem arginate treatment and patients with recurrent acute attacks.This audit examined the benefits and complications of regular haem arginate treatment started with prophylactic intent to reduce the frequency of recurrent acute attacks in a group of patients managed through NAPS. We included 22 patients (21 female and 1 male) and returned information on diagnosis, indications for prophylactic infusions, frequency and dose, analgesia, activity and employment and complications including thromboembolic disease and iron overload.The median age at presentation with porphyria was 21 years (range 9-44), with acute abdominal pain as the predominant symptom. Patients had a median of 12 (1-400) attacks before starting prophylaxis and had received a median of 52 (0-1,350) doses of haem arginate. The median age at starting prophylaxis was 28 years (13-58) with a median delay of 4 years (0.5-37) between presentation and prophylaxis. The frequency of prophylactic haem arginate varied from 1 to 8 per month, and 67% patients were documented as having a reduction in pain frequency on prophylaxis. Only one patient developed clinically significant iron overload and required iron chelation, but the number of venous access devices required varied from 1 to 15, with each device lasting a median of 1.2 years before requiring replacement. Six patients stopped haem arginate and in three this was because their symptoms had improved. Prophylactic haem arginate appears to be beneficial in patients with recurrent acute porphyria symptoms, but maintaining central venous access may prove challenging.
Figures
Fig. 1
Graph showing the correlation between the number of haem arginate vials and serum ferritin concentration
Fig. 2
Graph showing total number of doses of haem arginate and indwelling devices used
Similar articles
- Desensitization in patients with hypersensitivity to haem arginate: A case report.
Chapman E, Leal D, Matijasevic E, García E. Chapman E, et al. World Allergy Organ J. 2019 Jan 26;12(1):100002. doi: 10.1016/j.waojou.2018.11.002. eCollection 2019. World Allergy Organ J. 2019. PMID: 30937127 Free PMC article. - Controlled trial of haem arginate in acute hepatic porphyria.
Herrick AL, McColl KE, Moore MR, Cook A, Goldberg A. Herrick AL, et al. Lancet. 1989 Jun 10;1(8650):1295-7. doi: 10.1016/s0140-6736(89)92688-3. Lancet. 1989. PMID: 2566827 Clinical Trial. - Best practice guidelines on clinical management of acute attacks of porphyria and their complications.
Stein P, Badminton M, Barth J, Rees D, Stewart MF; British and Irish Porphyria Network. Stein P, et al. Ann Clin Biochem. 2013 May;50(Pt 3):217-23. doi: 10.1177/0004563212474555. Ann Clin Biochem. 2013. PMID: 23605132 - [Acute intermittent porphyria].
Tollåli G, Nielsen EW, Brekke OL. Tollåli G, et al. Tidsskr Nor Laegeforen. 2002 Apr 30;122(11):1102-5. Tidsskr Nor Laegeforen. 2002. PMID: 12043053 Review. Norwegian. - Update review of the acute porphyrias.
Stein PE, Badminton MN, Rees DC. Stein PE, et al. Br J Haematol. 2017 Feb;176(4):527-538. doi: 10.1111/bjh.14459. Epub 2016 Dec 16. Br J Haematol. 2017. PMID: 27982422 Review.
Cited by
- Long-term follow-up of givosiran treatment in patients with acute intermittent porphyria from a phase 1/2, 48-month open-label extension study.
Sardh E, Balwani M, Rees DC, Anderson KE, Jia G, Sweetser MT, Wang B. Sardh E, et al. Orphanet J Rare Dis. 2024 Oct 3;19(1):365. doi: 10.1186/s13023-024-03284-w. Orphanet J Rare Dis. 2024. PMID: 39363243 Free PMC article. Clinical Trial. - Understanding Hepatic Porphyrias: Symptoms, Treatments, and Unmet Needs.
Balogun O, Nejak-Bowen K. Balogun O, et al. Semin Liver Dis. 2024 May;44(2):209-225. doi: 10.1055/s-0044-1787076. Epub 2024 May 17. Semin Liver Dis. 2024. PMID: 38772406 Free PMC article. Review. - Hematin- and Hemin-Induced Spherization and Hemolysis of Human Erythrocytes Are Independent of Extracellular Calcium Concentration.
Mikhailova DM, Skverchinskaya E, Sudnitsyna J, Butov KR, Koltsova EM, Mindukshev IV, Gambaryan S. Mikhailova DM, et al. Cells. 2024 Mar 21;13(6):554. doi: 10.3390/cells13060554. Cells. 2024. PMID: 38534398 Free PMC article. - Clinical features of acute attacks, chronic symptoms, and long-term complications among patients with acute hepatic porphyria in Japan: a real-world claims database study.
Horie Y, Yasuoka Y, Adachi T. Horie Y, et al. Orphanet J Rare Dis. 2023 Dec 8;18(1):384. doi: 10.1186/s13023-023-02913-0. Orphanet J Rare Dis. 2023. PMID: 38066651 Free PMC article. - The Hepatic Porphyrias: Revealing the Complexities of a Rare Disease.
Balogun O, Nejak-Bowen K. Balogun O, et al. Semin Liver Dis. 2023 Nov;43(4):446-459. doi: 10.1055/s-0043-1776760. Epub 2023 Nov 16. Semin Liver Dis. 2023. PMID: 37973028 Free PMC article. Review.
References
LinkOut - more resources
Full Text Sources
Other Literature Sources