Evidence map›Paper›PMID 36747680›Full record

ArticlebioRxiv : the preprint server for biology2023

Therapy Development for Microvillus Inclusion Disease using Patient-derived Enteroids.

Meri Kalashyan, Krishnan Raghunathan, Haley Oller, Marie-Bayer Theres, Lissette Jimenez, Joseph T Roland, Elena Kolobova, Susan J Hagen, Jeffrey D Goldsmith, Mitchell D Shub and 3 more

Open access · greenAbstract readPreprint
PubMed Full text at PMC Publisher
In one paragraph

Article in bioRxiv : the preprint server for biology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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

0 citing papers in PubMed, 1 citations in OpenAlex.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

13 authors at 5 institutions in 1 country.

Meri KalashyanDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Krishnan RaghunathanDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Haley OllerDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Marie-Bayer TheresDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Lissette JimenezDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Joseph T RolandSection of Surgical Sciences, Vanderbilt University Medical Center, Nashville, Tennessee.
Elena KolobovaSection of Surgical Sciences, Vanderbilt University Medical Center, Nashville, Tennessee.
Susan J HagenDepartment of Surgery, Division of Surgical Sciences, Beth Israel Deaconess Medical Center and Harvard Medical School, Boston, Massachusetts, USA.
Jeffrey D GoldsmithDepartment of Pathology, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Mitchell D ShubDepartment of Child Health University of Arizona College of Medicine-Phoenix and Division of Gastroenterology, Phoenix Children's.
James R GoldenringSection of Surgical Sciences, Vanderbilt University Medical Center, Nashville, Tennessee.
Izumi KajiSection of Surgical Sciences, Vanderbilt University Medical Center, Nashville, Tennessee.
Jay R ThiagarajahDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.
Boston Children's Hospital · USVanderbilt University Medical Center · USBeth Israel Deaconess Medical Center · USNashville VA Medical Center · USUniversity of Arizona · US

Funding

STRUCTURE-FUNCTION RELATIONSHIPS IN THE ALIMENTARY TRACTP30DK034854 · NIDDK · HARVARD UNIVERSITY (MEDICAL SCHOOL) · PI JONATHAN C KAGAN · 1986 to 2026
$32.4M
COngenital Diarrhea and Enteropathy (PediCODE) Consortium and BioRepositoryRC2DK118640 · NIDDK · UNIVERSITY OF CALIFORNIA LOS ANGELES · PI James Richard Goldenring, Izumi Kaji · 2019 to 2026
$14.8M
SMALL GTP BINDING PROTEINS IN GASTROINTESTINAL MUCOSAR01DK048370 · NIDDK · VANDERBILT UNIVERSITY MEDICAL CENTER · PI GOLDENRING, JAMES RICHARD · 1994 to 2025
$10.1M
Impacting the pathophysiology of malabsorption induced by Myosin Vb inactivating mutationsR01DK128190 · NIDDK · VANDERBILT UNIVERSITY MEDICAL CENTER · PI KAJI, IZUMI · 2021 to 2025
$1.9M
NIDDK NIH HHS P30 DK034854NIDDK NIH HHS R01 DK048370NIDDK NIH HHS R01 DK128190NIDDK NIH HHS RC2 DK118640
6 · The paper itself

Abstract

Microvillus Inclusion Disease (MVID), caused by loss-of-function mutations in the motor protein Myosin Vb (MYO5B), is a severe infantile disease characterized by diarrhea, malabsorption, and acid-base instability, requiring intensive parenteral support for nutritional and fluid management. Human patient-derived enteroids represent a model for investigation of monogenic epithelial disorders but are a rare resource from MVID patients. We developed human enteroids with different loss-of function MYO5B variants and showed that they recapitulated the structural changes found in native MVID enterocytes. Multiplex Immunofluorescence imaging of patient duodenal tissues revealed patient-specific changes in localization of brush border transporters. Functional analysis of electrolyte transport revealed profound loss of Na Conflict-of-interest statement: The authors have declared that no conflict of interest exists.

Identifiers

PMID36747680
PMCPMC9900906
OpenAlexW4318448180

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.