Evidence map›Paper›PMID 37488949›Full record

ArticleArthritis & rheumatology (Hoboken, N.J.)2024

Anakinra-Associated Systemic Amyloidosis.

Sara Alehashemi, Surendra Dasari, Anvitha Metpally, Kat Uss, Leslie A Castelo-Soccio, Theo Heller, Peter Kellman, Marcus Y Chen, Mark Ahlman, Jeff Kim and 11 more

Registry-linked trialOpen access · bronzeAbstract readCase Reports
In one paragraph

Article in Arthritis & rheumatology (Hoboken, N.J.), 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT06974877 (A Phase 1 Study of Repeat PET/CT Imaging in People With CAPS and Anakinra-Induced Amyloidosis Using Amyloid-Reactive Peptide 124I-AT-01), which is not on this map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
1.5field-weighted citation impact, top 17% 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.

NCT06974877 phase1recruitingnot on this mapstarted 2025, after this paper: background citation

A Phase 1 Study of Repeat PET/CT Imaging in People With CAPS and Anakinra-Induced Amyloidosis Using Amyloid-Reactive Peptide 124I-AT-01 (124I-p5+14, Iodine-124I-Evuzamitide) to Measure Changes in Organ-Specific Amyloid Load

TypeinterventionalSponsorNational Institute of Allergy and Infectious Diseases (NIAID)Ran2025 to 2032Enrolled30ConditionsCryopyrin-Associated Periodic Syndromes, AmyloidosisArms124I AT-01
3 · Its place in the literature

Who cites it

6 citing papers in PubMed, 10 citations in OpenAlex.

  1. Article
  2. How I Treat: Haploinsufficiency of A20.Journal of human immunity · 2026
    Review
  3. Article
  4. Review
  5. Review
  6. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

21 authors at 10 institutions in 1 country.

Sara AlehashemiNational Institute of Allergy and Infectious Diseases, NIH, Bethesda, Maryland.ORCID 0000-0002-6531-6108
Surendra DasariMayo Clinic, Rochester, Minnesota.
Anvitha MetpallyNational Institute of Allergy and Infectious Diseases, NIH, Bethesda, Maryland.
Kat UssNational Institute of Allergy and Infectious Diseases, NIH, Bethesda, Maryland.
Leslie A Castelo-SoccioNational Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda, Maryland.
Theo HellerNational Institute of Diabetes and Digestive and Kidney Diseases, NIH, Bethesda, Maryland.
Peter KellmanNational Heart, Lung, and Blood Institute, NIH, Bethesda, Maryland.
Marcus Y ChenNational Heart, Lung, and Blood Institute, NIH, Bethesda, Maryland.
Mark AhlmanClinical Center, NIH, Bethesda, Maryland.
Jeff KimNational Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland.
Susannah WargoNational Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland.
Douglas B KuhnsFrederick National Laboratory for Cancer Research, Frederick, Maryland.
Danielle FinkFrederick National Laboratory for Cancer Research, Frederick, Maryland.
Adriana de JesusNational Institute of Allergy and Infectious Diseases, NIH, Bethesda, Maryland.
Paul S MartinUniversity of Washington, Seattle.
Richard ChangClinical Center, NIH, Bethesda, Maryland.
Jonathan BolanosNational Institute of Diabetes and Digestive and Kidney Diseases, NIH, Bethesda, Maryland.
Chyi-Chia Richard LeeNational Cancer Institute, NIH, Bethesda, Maryland.
Samih H NasrMayo Clinic, Rochester, Minnesota.
Raphaela Goldbach-ManskyNational Institute of Allergy and Infectious Diseases, NIH, Bethesda, Maryland.ORCID 0000-0001-7865-5769
Ellen McPhailMayo Clinic, Rochester, Minnesota.
National Institute of Allergy and Infectious Diseases · USMayo Clinic · USFrederick National Laboratory for Cancer Research · USNational Heart Lung and Blood Institute · USNational Institute on Deafness and Other Communication Disorders · USNational Institutes of Health · USNational Institutes of Health Clinical Center · USNational Cancer Institute · USNational Institute of Arthritis and Musculoskeletal and Skin Diseases · USUniversity of Washington · US

Funding

Cardiovascular CT Clinical ResearchZIAHL006220 · NHLBI · NATIONAL HEART, LUNG, AND BLOOD INSTITUTE · PI CHEN, MARCUS · 2017 to 2025
$22.5M
Pathogenesis, and Outcome of Autoinflammatory Diseases, NOMID/CAPS, DIRA, CANDLE, SAVI, NLRC4-MAS, Stills-like Diseases, and other Undifferentiated Autoinflammatory DiseasesZIAAI001220 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI GOLDBACH-MANSKY, RAPHAELA · 2017 to 2025
$22.3M
Cardiovascular Clinical Section StudiesZICHL006199 · NHLBI · NATIONAL HEART, LUNG, AND BLOOD INSTITUTE · PI SACK, MICHAEL · 2017 to 2025
$8.8M
Gadgetron Global Network and Intelligence Computing: Clinical Imaging Application Development and Software InfrastructureZIAHL006214 · NHLBI · NATIONAL HEART, LUNG, AND BLOOD INSTITUTE · PI KELLMAN, PETER · 2016 to 2025
$7.3M
Investigation of non-cirrhotic portal hypertension.ZIADK075008 · NIDDK · NATIONAL INSTITUTE OF DIABETES AND DIGESTIVE AND KIDNEY DISEASES · PI HELLER, THEO · 2009 to 2025
$4.5M
Intramural NIH HHS ZIA AI001220
6 · The paper itself

Abstract

objectiveTo describe a 41-year-old woman with a history of neonatal onset multisystem inflammatory disease, on treatment with daily subcutaneous injections of 600 mg of recombinant interleukin-1 receptor antagonist (IL-1Ra) protein, anakinra, since the age of 28, who presented with golf-ball size nodules at the anakinra injection sites, early satiety, new onset nephrotic syndrome in the context of normal markers of systemic inflammation.

methodsClinical history and histologic evaluation of biopsies of skin, gastric mucosa, and kidney with Congo-red staining and proteomic evaluation of microdissected Congo red-positive amyloid deposits by liquid chromatography-tandem mass spectrometry.

resultsThe skin, stomach, and kidney biopsies all showed the presence of Congo red-positive amyloid deposits. Mass spectrometry-based proteomics demonstrated that the amyloid deposits in all sites were of AIL1RAP (IL-1Ra protein)-type. These were characterized by high spectral counts of the amyloid signature proteins (apolipoprotein AIV, apolipoprotein E, and serum amyloid P-component) and the amyloidogenic IL-1Ra protein, which were present in Congo red-positive areas and absent in Congo red-negative areas. The amino acid sequence identified by mass spectrometry confirmed that the amyloid precursor protein was recombinant IL-1Ra (anakinra) and not endogenous wild-type IL-1Ra.

conclusionThis is the first report of iatrogenic systemic amyloidosis due to an injectable protein drug, which was caused by recombinant IL1Ra (anakinra).

Indexed as

AmyloidosisInterleukin 1 Receptor Antagonist ProteinAdultCongo RedFemaleHumansInfant, NewbornPlaque, AmyloidProteomicsCongo RedInterleukin 1 Receptor Antagonist Protein

Identifiers

PMID37488949
PMCPMC10805950
OpenAlexW4385231236

What OpenQuestion holds

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Read underepoch 390

Registered trials

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 OpenQuestion graph.