Evidence map›Paper›PMID 36636593›Full record

ArticleJIMD reports2023

Quantifying the impact of symptomatic acute hepatic porphyria on well-being via patient-reported outcomes: Results from the Porphyria Worldwide Patient Experience Research (POWER) study.

Amy Dickey, Kristen Wheeden, Desiree Lyon, Sue Burrell, Sean Hegarty, Rocco Falchetto, Edrin R Williams, Jasmin Barman-Aksözen, Marc DeCongelio, Alison Bulkley and 7 more

Erratum issuedOpen access · goldAbstract read
In one paragraph

Article in JIMD reports, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed
1.5field-weighted citation impact, top 18% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

12 citing papers in PubMed, 18 citations in OpenAlex.

  1. Trial
  2. Trial
  3. Article
  4. Article
  5. Article
  6. Givosiran: a targeted treatment for acute intermittent porphyria.Hematology. American Society of Hematology. Education Program · 2024
    Review
  7. Article
  8. Article
  9. Article
  10. CORRIGENDUM.JIMD reports · 2023
    Article
  11. Review
  12. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

17 authors at 8 institutions in 3 countries.

Amy DickeyDivision of Pulmonary and Critical Care Medicine, Department of Medicine Massachusetts General Hospital Boston Massachusetts USA.ORCID https://orcid.org/0000-0003-2979-9664
Kristen WheedenAmerican Porphyria Foundation Bethesda Maryland USA.
Desiree LyonAmerican Porphyria Foundation Bethesda Maryland USA.
Sue BurrellGlobal Porphyria Advocacy Coalition Durham City UK.
Sean HegartyGlobal Porphyria Advocacy Coalition Durham City UK.
Rocco FalchettoSwiss Society for Porphyria Zürich Switzerland.
Edrin R WilliamsAmerican Porphyria Foundation Bethesda Maryland USA.
Jasmin Barman-AksözenSwiss Society for Porphyria Zürich Switzerland.ORCID https://orcid.org/0000-0001-7272-8824
Marc DeCongelioCerner Enviza (formerly Kantar Health) New York New York USA.
Alison BulkleyCerner Enviza (formerly Kantar Health) New York New York USA.
Joana E MatosCerner Enviza (formerly Kantar Health) Kansas City Missouri USA.
Tarek MnifCerner Enviza (formerly Kantar Health) Paris France.
Jordanna MoraAlnylam Pharmaceuticals Cambridge Massachusetts USA.
John J KoAlnylam Pharmaceuticals Cambridge Massachusetts USA.
Stephen MeningerAlnylam Pharmaceuticals Cambridge Massachusetts USA.
Stephen LombardelliAlnylam Pharmaceuticals Maidenhead UK.
Danielle NanceBanner Health Gilbert Arizona USA.
Alnylam Pharmaceuticals (United States) · USAmerican Porphyria Foundation · USCerner (United States) · USImmunization Action Coalition · USBanner Health · USKantar Health (Brazil) · BRMassachusetts General Hospital · USTriemli Hospital · CH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Acute hepatic porphyria (AHP) is a group of rare genetic diseases of heme biosynthesis resulting in severe neurovisceral attacks and chronic complications that negatively impact patients' well-being. This study evaluated the impacts of AHP on patients' physical and emotional health from a global perspective. Adult patients from the United States, Italy, Spain, Australia, Mexico, and Brazil with AHP with >1 porphyria attack within the past 2 years or receiving intravenous hemin and/or glucose for attack prevention completed an online survey assessing demographics, health characteristics, and patient-reported outcomes. Results were analyzed collectively and by patient subgroups. Ninety-two patients with AHP across the six countries completed the survey. More than 70% of patients reported that their physical, emotional, and financial health was fair or poor. Among patients who reported pain, fatigue, and muscle weakness, 94.3%, 95.6%, and 91.4%, respectively, reported that these symptoms limited daily activities. Moderate to severe depression was present in 58.7% of patients, and moderate to severe anxiety in 48.9% of patients. Of the 47% of patients who were employed, 36.8% reported loss in productivity while at work. Among patients, 85.9% reported that they had to change or modify goals that were important to them because of AHP. Aside from differences in healthcare utilization and pain severity, scores did not significantly vary with attack rate or use of hemin or glucose prophylactic treatments. AHP substantially impacts patients' physical and emotional well-being, regardless of hemin or glucose prophylactic treatment or frequency of attacks. This multinational study demonstrates that there is substantial disease burden for patients with AHP, even among those experiencing sporadic attacks or using prophylactic treatment.

Indexed as

acute hepatic porphyriaacute intermittent porphyriaacute porphyriaAHPpatient‐reported outcomessurvey

Identifiers

PMID36636593
PMCPMC9830021
OpenAlexW4306722613

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.