Evidence map›Paper›PMID 26113602›Full record

ArticleEndocrine-related cancer2015

Development of pheochromocytoma in ceramide synthase 2 null mice.

Woo-Jae Park, Ori Brenner, Aviram Kogot-Levin, Ann Saada, Alfred H Merrill, Yael Pewzner-Jung, Anthony H Futerman

Open access · hybridAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Endocrine-related cancer, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.

0numbers the graph read from it
0cells of the map it votes in
16citing papers in PubMed
1.4field-weighted citation impact, top 19% 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

16 citing papers in PubMed, 34 citations in OpenAlex.

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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

7 authors at 3 institutions in 3 countries.

Woo-Jae ParkDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA Department of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA.
Ori BrennerDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA.
Aviram Kogot-LevinDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA Department of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA.
Ann SaadaDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA.
Alfred H MerrillDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA.
Yael Pewzner-JungDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA yael.pewzner-jung@weizmann.ac.il.
Anthony H FutermanDepartment of Biological ChemistryWeizmann Institute of Science, Rehovot 76100, IsraelDepartment of BiochemistrySchool of Medicine, Gachon University, Incheon 406-799, South KoreaDepartment of Veterinary ResourcesWeizmann Institute of Science, Rehovot 76100, IsraelMonique and Jacques Roboh Department of Genetic ResearchDepartment of Genetics and Metabolic Diseases, Hadassah, Hebrew University Medical Center, Jerusalem, IsraelSchool of Biology and Petit Institute for Bioengineering and BioscienceGeorgia Institute of Technology, Atlanta, Georgia 30332-0230, USA.
Weizmann Institute of Science · ILGeorgia Institute of Technology · USHadassah Medical Center · IL

Funding

Characterization of Mammalian Ceramide SynthasesR01GM076217 · NIGMS · GEORGIA INSTITUTE OF TECHNOLOGY · PI MERRILL, ALFRED HARRISON · 2007 to 2015
$2.4M
NIGMS NIH HHS R01 GM076217
6 · The paper itself

Abstract

Pheochromocytoma (PCC) and paraganglioma are rare neuroendocrine tumors of the adrenal medulla and sympathetic and parasympathetic paraganglia, for which mutations in ∼15 disease-associated genes have been identified. We now document the role of an additional gene in mice, the ceramide synthase 2 (CerS2) gene. CerS2, one of six mammalian CerS, synthesizes ceramides with very-long (C22-C24) chains. The CerS2 null mouse has been well characterized and displays lesions in several organs including the liver, lung and the brain. We now demonstrate that changes in the sphingolipid acyl chain profile of the adrenal gland lead to the generation of adrenal medullary tumors. Histological analyses revealed that about half of the CerS2 null mice developed PCC by ∼13 months, and the rest showed signs of medullary hyperplasia. Norepinephrine and normetanephrine levels in the urine were elevated at 7 months of age consistent with the morphological abnormalities found at later ages. Accumulation of ceroid in the X-zone was observed as early as 2 months of age and as a consequence, older mice displayed elevated levels of lysosomal cathepsins, reduced proteasome activity and reduced activity of mitochondrial complex IV by 6 months of age. Together, these findings implicate an additional pathway that can lead to PCC formation, which involves alterations in the sphingolipid acyl chain length. Analysis of the role of sphingolipids in PCC may lead to further understanding of the mechanism by which PCC develops, and might implicate the sphingolipid pathway as a possible novel therapeutic target for this rare tumor.

Indexed as

Adrenal Gland NeoplasmsAdrenal GlandsAnimalsCatecholaminesCathepsinsFemaleMaleMice, KnockoutMitochondriaPheochromocytomaSphingosine N-AcyltransferaseCatecholaminesCathepsinsCers2 protein, mouseSphingosine N-Acyltransferaseacyl chain lengthceramideceroidlipofuscinoxidative stresspheochromocytomasphingolipid

Identifiers

PMID26113602
PMCPMC5586043
OpenAlexW2171162411

What OpenQuestion holds

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Registered trials

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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.