Evidence map›Paper›PMID 39373891›Full record

ArticleOphthalmic genetics2024

Abetalipoproteinemia with angioid streaks, choroidal neovascularization, atrophy, and extracellular deposits revealed by multimodal retinal imaging.

Jacques Bijon, M Mahmood Hussain, Cindy L Bredefeld, Kathleen Boesze-Battaglia, K Bailey Freund, Christine A Curcio

Abstract readCase Reports
In one paragraph

Article in Ophthalmic genetics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. 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

6 authors.

Jacques BijonOphthalmology, Vitreous Retina Macula Consultants of New York, New York, New York, USA.
M Mahmood HussainDepartment of Foundations of Medicine, New York University Grossman Long Island School of Medicine, NYU Langone Hospitals-Long Island, Mineola, New York, USA.
Cindy L BredefeldDepartment of Foundations of Medicine, New York University Grossman Long Island School of Medicine, NYU Langone Hospitals-Long Island, Mineola, New York, USA.
Kathleen Boesze-BattagliaDepartment of Basic and Translational Sciences, School of Dental Medicine, University of Pennsylvania, Philadelphia, USA.
K Bailey FreundOphthalmology, Vitreous Retina Macula Consultants of New York, New York, New York, USA.
Christine A CurcioDepartment of Ophthalmology and Visual Sciences, University of Alabama at Birmingham Heersink School of Medicine, Birmingham, Alabama, USA.

Funding

Novel regulatory mechanisms controlling hepatic apoB-Lp lipid loading and secretionP01HL160470 · NHLBI · NYU LONG ISLAND SCHOOL OF MEDICINE · PI M Mahmood Hussain · 2023 to 2026
$12.3M
QUANTITATIVE ANAYLSIS OF AGING RETINAR01EY006109 · NEI · UNIVERSITY OF WASHINGTON · PI CURCIO, CHRISTINE A · 1986 to 2012
$3.5M
Mapping MTP lipid transfer activities for better therapeuticsR01HL158054 · NHLBI · NYU LONG ISLAND SCHOOL OF MEDICINE · PI FARBER, STEVEN A, HUSSAIN, M MAHMOOD · 2021 to 2024
$2.4M
Microsomal Transfer Protein Modulates Lipoprotein Metabolism and Retinal lipid HomeostasisR21EY032743 · NEI · UNIVERSITY OF PENNSYLVANIA · PI BOESZE-BATTAGLIA, KATHLEEN, HUSSAIN, M MAHMOOD · 2022 to 2023
$454k
NEI NIH HHS R01 EY006109NEI NIH HHS R21 EY032743NHLBI NIH HHS P01 HL160470NHLBI NIH HHS R01 HL158054
6 · The paper itself

Abstract

purposeAbetalipoproteinemia (ABL, MIM 200,100) is a rare autosomal recessive disorder caused by nonfunctional microsomal triglyceride transfer protein leading to absence of apolipoprotein B-containing lipoproteins in plasma and a retinitis pigmentosa-like fundus. The MTTP gene is expressed in retinal pigment epithelium (RPE) and ganglion cells of the human retina. Understanding ABL pathophysiology would benefit from new cellular-level clinical imaging of affected retinas.

methodsWe report multimodal retinal imaging in two patients with ABL. Case 1 (67-year-old woman) exhibited a bilateral decline of vision due to choroidal neovascularization (CNV) associated with angioid streaks and calcified Bruch membrane. Optical coherence tomography were consistent with basal laminar deposits and subretinal drusenoid deposits (SDD).

resultsCase 2 (46-year-old woman) exhibited unusual hyperpigmentation at the right fovea with count-fingers vision and a relatively unremarkable left fundus with 20/30 vision. The left eye exhibited the presence of nodular drusen and SDD and the absence of macular xanthophyll pigments.

conclusionWe propose that mutated MTTP within the retina may contribute to ABL retinopathy in addition to systemic deficiencies of fat-soluble vitamins. This concept is supported by a new mouse model with RPE-specific MTTP deficiency and a retinal degeneration phenotype. The observed range of human pathology, including angioid streaks, underscores the need for continued monitoring in adulthood, especially for CNV, a treatable condition.

Indexed as

AbetalipoproteinemiaAngioid StreaksChoroidal NeovascularizationMultimodal ImagingTomography, Optical CoherenceAgedAtrophyBruch MembraneFemaleFluorescein AngiographyHumansMiddle AgedRetinal DrusenRetinal Pigment Epitheliumabetalipoproteinemiaage-related macular degenerationangioid streaksautofluorescenceoptical coherence tomographyRetina

Identifiers

PMID39373891
PMCPMC11598668

What Socratic 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 Socratic graph.