Evidence map›Paper›PMID 35628659›Full record

ArticleInternational journal of molecular sciences2022

Hematopoietic Disorders, Renal Impairment and Growth in Mucopolysaccharidosis-Plus Syndrome.

Viktoriia Sofronova, Rina Iwata, Takuya Moriya, Kiunniai Loskutova, Elizaveta Gurinova, Mairanush Chernova, Anastasia Timofeeva, Anna Shvedova, Filipp Vasilev, Saina Novgorodova and 5 more

Open access · goldAbstract read
In one paragraph

Article in International journal of molecular sciences, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed
1.7field-weighted citation impact, top 16% 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, 15 citations in OpenAlex.

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  12. Mucopolysaccharidosis-Plus Syndrome: Report on a Polish Patient with a NovelInternational journal of molecular sciences · 2022
    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

15 authors at 2 institutions in 2 countries.

Viktoriia SofronovaDepartment of Molecular and Genetic Medicine, Kawasaki Medical School, Kurashiki 701-0192, Japan.ORCID 0000-0002-9029-1694
Rina IwataDepartment of Molecular and Genetic Medicine, Kawasaki Medical School, Kurashiki 701-0192, Japan.
Takuya MoriyaDepartment of Pathology, Kawasaki Medical School, Kurashiki 701-0192, Japan.
Kiunniai LoskutovaDepartment of Pathological Anatomy, Republic Hospital No. 1-National Center of Medicine, 677019 Yakutsk, Russia.
Elizaveta GurinovaMedical Genetics Center, Republic Hospital No. 1-National Center of Medicine, 677019 Yakutsk, Russia.
Mairanush ChernovaDepartment of Children's Health and Pathological Anatomy, Republic Hospital No. 1-National Center of Medicine, 677019 Yakutsk, Russia.
Anastasia TimofeevaDepartment of Children's Health and Pathological Anatomy, Republic Hospital No. 1-National Center of Medicine, 677019 Yakutsk, Russia.
Anna ShvedovaDepartment of Children's Health and Pathological Anatomy, Republic Hospital No. 1-National Center of Medicine, 677019 Yakutsk, Russia.
Filipp VasilevLaboratory of Molecular Medicine and Human Genetics, North-Eastern Federal University, 677013 Yakutsk, Russia.ORCID 0000-0002-0315-852X
Saina NovgorodovaLaboratory of Molecular Medicine and Human Genetics, North-Eastern Federal University, 677013 Yakutsk, Russia.
Seigo TerawakiDepartment of Molecular and Genetic Medicine, Kawasaki Medical School, Kurashiki 701-0192, Japan.ORCID 0000-0001-6019-1628
Takahito MoriwakiDepartment of Molecular and Genetic Medicine, Kawasaki Medical School, Kurashiki 701-0192, Japan.
Aitalina SukhomyasovaLaboratory of Molecular Medicine and Human Genetics, North-Eastern Federal University, 677013 Yakutsk, Russia.
Nadezhda MaksimovaLaboratory of Molecular Medicine and Human Genetics, North-Eastern Federal University, 677013 Yakutsk, Russia.
Takanobu OtomoDepartment of Molecular and Genetic Medicine, Kawasaki Medical School, Kurashiki 701-0192, Japan.ORCID 0000-0001-8490-7299
North-Eastern Federal University · RUKawasaki Medical School · JP

Funding

Japan Society for the Promotion of Science JP17K05088Ministry Education and Science of Russian Federation FSRG-2020-0014Ministry of Education, Culture, Sports, Science and Technology Scholarship
6 · The paper itself

Abstract

Mucopolysaccharidoses (MPS) are rare lysosomal storage disorders (LSD) characterized by the excessive accumulation of glycosaminoglycans (GAG). Conventional MPS, caused by inborn deficiencies of lysosomal enzymes involved in GAG degradation, display various multisystemic symptoms-including progressive neurological complications, ophthalmological disorders, hearing loss, gastrointestinal and hepatobiliary issues, cardiorespiratory problems, bone and joint abnormalities, dwarfism, and coarse facial features. Mucopolysaccharidosis-Plus Syndrome (MPSPS), an autosomal recessive disease caused by a mutation in the endo-lysosomal tethering protein VPS33A, shows additional renal and hematopoietic abnormalities ("Plus symptoms") uncommon in conventional MPS. Here, we analyze data from biochemical, histological, and physical examinations-particularly of blood counts and kidney function-to further characterize the clinical phenotype of MPSPS. A series of blood tests indicate hematopoietic symptoms including progressive anemia and thrombocytopenia, which correlate with histological observations of hypoplastic bone marrow. High urinary excretion of protein (caused by impairments in renal filtration), hypoalbuminemia, and elevated levels of creatinine, cholesterol, and uric acid indicate renal dysfunction. Histological analyses of MPSPS kidneys similarly suggest the extensive destruction of glomerular structures by foamy podocytes. Height and weight did not significantly deviate from the average, but in some cases, growth began to decline at around six months or one year of age.

Indexed as

Eye DiseasesHematologic DiseasesMucopolysaccharidosesGlycosaminoglycansHumansMutationGlycosaminoglycansanemiafoamy podocytelysosomal storage diseasemucopolysaccharidosismucopolysaccharidosis-plus syndromeproteinuriarenal failurethrombocytopeniaVPS33A

Identifiers

PMID35628659
PMCPMC9145135
OpenAlexW4281396907

What OpenQuestion holds

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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 OpenQuestion graph.