Evidence map›Paper›PMID 29599423›Full record

ArticleThe American journal of case reports2018

Atypical Presentation of Gelsolin Amyloidosis in a Man of African Descent with a Novel Mutation in the Gelsolin Gene.

Karlos Z Oregel, Geoffrey P Shouse, Cyrus Oster, Freddy Martinez, Jun Wang, Michael Rosenzweig, Jeremy K Deisch, Chien-Shing Chen, Gayathri Nagaraj

Open access · hybridAbstract readCase Reports
In one paragraph

Article in The American journal of case reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing papers in PubMed
1.1field-weighted citation impact, top 23% 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

8 citing papers in PubMed, 16 citations in OpenAlex.

  1. A molecular perspective of gelsolin amyloidosis: An old foe with new faces.Cellular and molecular life sciences : CMLS · 2026
    Review
  2. Plant Hormone Cytokinin as Aggregation Modulator of Gelsolin Amyloidosis.Journal of peptide science : an official publication of the European Peptide Society · 2025
    Article
  3. Article
  4. Article
  5. Article
  6. Analyses Mutations inFrontiers in aging neuroscience · 2020
    Article
  7. Article
  8. The role of gelsolin domain 3 in familial amyloidosis (Finnish type).Proceedings of the National Academy of Sciences of the United States of America · 2019
    Article
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

9 authors at 4 institutions in 1 country.

Karlos Z OregelDivision of Hematology/Oncology, Loma Linda University School of Medicine, Loma Linda, CA, USA.
Geoffrey P ShouseDivision of Hematology/Oncology, Loma Linda University School of Medicine, Loma Linda, CA, USA.
Cyrus OsterDivision of Pathology, Harbor UCLA Medical Center, Los Angeles, CA, USA.
Freddy MartinezUniversity of California, Irvine, CA, USA.
Jun WangDivision of Pathology and Laboratory Medicine, Loma Linda University School of Medicine, Loma Linda, CA, USA.
Michael RosenzweigDepartment of Hematology and Hematopoietic Cell Transplantation, City of Hope Medical Center, Duarte, CA, USA.
Jeremy K DeischDivision of Pathology and Laboratory Medicine, Loma Linda University School of Medicine, Loma Linda, CA, USA.
Chien-Shing ChenDivision of Hematology/Oncology, Loma Linda University School of Medicine, Loma Linda, CA, USA.
Gayathri NagarajDivision of Hematology/Oncology, Loma Linda University School of Medicine, Loma Linda, CA, USA.
Loma Linda University · USCity of Hope · USHarbor–UCLA Medical Center · USUniversity of California, Irvine · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND Gelsolin amyloidosis is a very rare systemic disease presenting with a pathognomonic triad of corneal lattice dystrophy, cutis laxa, and polyneuropathy. The disease is mostly restricted to a Finnish population with known mutations (G654A, G654T) in exon 4 of the gelsolin gene. The mutations lead to errors in protein processing and folding, and ultimately leads to deposition of an amyloidogenic fragment in the extracellular space, causing the symptoms of disease. CASE REPORT We present a case of gelsolin amyloidosis in a male of African descent with an atypical clinical presentation including fevers, skin rash, polyneuropathy, and anemia. Gelsolin amyloidosis was diagnosed based on mass spectrometry of tissue samples. Importantly, a novel mutation in the gelsolin gene (C1375G) in exon 10 was found in this patient. His atypical presentation can possibly be attributed to the presence of a novel mutation in the gelsolin gene as the likely underlying cause of the syndrome. PCR primers were used to amplify the gelsolin gene from genomic DNA. Purified PCR products were then shipped to Eton Biosciences (San Diego, CA) for sequencing. CONCLUSIONS This study carries several important lessons relevant to the practice of medicine. First, the differential diagnosis for multisystem disease presentations should always include amyloidosis. Second, despite what has been uncovered about the molecular biology of disease, there is always more that can be discovered. Finally, further work to verify the link between this mutation and the clinical syndrome is still needed, as are effective treatments for this disease.

Indexed as

MutationAmyloidosisDNADNA Mutational AnalysisGelsolinHumansMaleMiddle AgedDNAGelsolin

Identifiers

PMID29599423
PMCPMC5890616
OpenAlexW2794644152

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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 OpenQuestion graph.