Evidence map›Paper›PMID 32104752›Full record

ArticleJournal of the Endocrine Society2020

Use of PCSK9 Inhibitor in a Mexican Boy with Compound Heterozygous Familial Hypercholesterolemia: A Case Report.

José Juan Ceballos-Macías, Ramón Madriz-Prado, Norma Alejandra Vázquez Cárdenas, Carlos Aguilar-Salinas, Maria Teresa Tusié-Luna, Jorge Alberto Flores-Real, Guillermo Ortega-Gutiérrez, Joel Vargas-Sánchez, Carolina Lara-Sánchez, Alfredo Hernández-Moreno

Open access · goldAbstract readCase Reports
In one paragraph

Article in Journal of the Endocrine Society, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.7field-weighted citation impact, top 27% 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

3 citing papers in PubMed, 4 citations in OpenAlex.

  1. Article
  2. Article
  3. 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

10 authors at 4 institutions in 1 country.

José Juan Ceballos-MacíasServicio de Endocrinología, Unidad de Especialidades Médicas de la Secretaria de Defensa Nacional, Edo MX, Mexico.ORCID 0000-0001-5303-7104
Ramón Madriz-PradoServicio de Endocrinología, Unidad de Especialidades Médicas de la Secretaria de Defensa Nacional, Edo MX, Mexico.
Norma Alejandra Vázquez CárdenasDepartamento de Genética, ICB, Universidad Autónoma de Guadalajara, JLS, Mexico.
Carlos Aguilar-SalinasUnidad de Investigación de Enfermedades Metabólicas, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán.
Maria Teresa Tusié-LunaUnidad de Investigación de Enfermedades Metabólicas, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán.
Jorge Alberto Flores-RealServicio Endocrinología del Hospital Central Militar CDMX, Mexico.
Guillermo Ortega-GutiérrezServicio Endocrinología del Hospital Central Militar CDMX, Mexico.
Joel Vargas-SánchezServicio Endocrinología del Hospital Central Militar CDMX, Mexico.
Carolina Lara-SánchezServicio de Endocrinología, Unidad de Especialidades Médicas de la Secretaria de Defensa Nacional, Edo MX, Mexico.ORCID 0000-0001-7540-7495
Alfredo Hernández-MorenoEscuela Superior de Medicina, CDMX, Mexico.
Hospital Central Militar · MXHospital de Especialidades · MXInstituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán · MXUniversidad Autónoma de Guadalajara · MX

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

We report on the case of an 8-year-old Mexican male, with a 3-year-old clinical diagnosis of familial hypercholesterolemia, and the difficulties encountered in his treatment while in our care. His treatment started with a regimen consisting of ezetimibe/simvastatin, cholestyramine, and a dietary plan of 1600 calories, with a limited intake of 200 mg of cholesterol per day. Problems arose when the patient's low-density lipoprotein cholesterol (LDL) levels did not meet ideal targets, which prompted the use of LDL cholesterol apheresis (not available in Mexico) for 6 months. As a last resort, PCSK9 inhibitors were administered but the LDL levels remained in the 600 mg/dL range. AmbryGenetics conducted a genetic test employing the Sanger method. The results suggested that there were 2 different mutations for each allele of the same LDL receptor gene (c.249delTinsGG and p.(Cys109Arg)), located in exons 3 and 4, respectively. We identified compound heterozygous mutations in our index case, with him having both the p.C109R mutation (from the maternal lineage), as well as a c.249delTinsGG mutation (from the paternal lineage). The p.C109R mutation has been previously reported, not only in Mexico, but in European regions (Germany, Czech Republic, Ireland, Italy) as well. Functional studies indicated a residual enzymatic activity of 15% to 30% for heterozygotes. To date, the variant c.249delTinsGG has not been reported. This case study illustrates the fact that in Mexico there are limited options available for treatment in such a scenario. As medical professionals, we are limited by the tools at our disposal.

Indexed as

familial heterozygous hypercholesterolemiafamilial homozygous hypercholesterolemiafamilial hypercholesterolemiakexin type 9 geneLDL receptor genepremature cardiovascular diseaseprotein convertase subtilisin

Identifiers

PMID32104752
PMCPMC7035209
OpenAlexW2991318791

What OpenQuestion holds

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LicenceCC BY-NC-ND
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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.