Evidence map›Paper›PMID 36270406›Full record

ArticleOphthalmology2023

Benign Lobular Inner Nuclear Layer Proliferations of the Retina Associated with Congenital Hypertrophy of the Retinal Pigment Epithelium.

Christian J Sanfilippo, Michael Javaheri, Sheryl Handler, Jesse L Berry, David Cobrinik, Matthew A Deardorff, Miao Sun, Ryan J Schmidt, Andrew J Barkmeier, Aaron Nagiel

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

Article in Ophthalmology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed, 1 pooled it
2.2field-weighted citation impact, top 12% 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

2 citing papers in PubMed, 1 synthesis or guideline pooled it, 11 citations in OpenAlex.

  1. Pooled it
  2. 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

10 authors at 4 institutions in 1 country.

Christian J SanfilippoThe Retina Partners, Los Angeles, California.
Michael JavaheriRoski Eye Institute, Department of Ophthalmology, Keck School of Medicine, University of Southern California, Los Angeles, California.
Sheryl HandlerThe Vision Center, Department of Surgery, Children's Hospital Los Angeles, Los Angeles, California.
Jesse L BerryRoski Eye Institute, Department of Ophthalmology, Keck School of Medicine, University of Southern California, Los Angeles, California; The Vision Center, Department of Surgery, Children's Hospital Los Angeles, Los Angeles, California.
David CobrinikRoski Eye Institute, Department of Ophthalmology, Keck School of Medicine, University of Southern California, Los Angeles, California; The Vision Center, Department of Surgery, Children's Hospital Los Angeles, Los Angeles, California; Department of Biochemistry and Molecular Medicine, Keck School of Medicine, University of Southern California, Los Angeles, California; Norris Comprehensive Cancer Center, Keck School of Medicine, University of Southern California, Los Angeles, California.
Matthew A DeardorffDepartment of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los Angeles, California; Department of Pediatrics, Children's Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los Angeles, California.
Miao SunDepartment of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los Angeles, California.
Ryan J SchmidtDepartment of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los Angeles, California.
Andrew J BarkmeierDepartment of Ophthalmology, Mayo Clinic, Rochester, Minnesota.
Aaron NagielRoski Eye Institute, Department of Ophthalmology, Keck School of Medicine, University of Southern California, Los Angeles, California; The Vision Center, Department of Surgery, Children's Hospital Los Angeles, Los Angeles, California. Electronic address: anagiel@chla.usc.edu.
University of Southern California · USChildren's Hospital of Los Angeles · USMayo Clinic · USRetina Partners · US

Funding

Development of a surrogate liquid biopsy from the aqueous humor in retinoblastoma eyes.K08CA232344 · NCI · CHILDREN'S HOSPITAL OF LOS ANGELES · PI BERRY, JESSE L · 2018 to 2022
$1.1M
Development and Maintenance of the Human Photoreceptor-Bipolar Cell Synapse.K08EY030924 · NEI · CHILDREN'S HOSPITAL OF LOS ANGELES · PI NAGIEL, AARON · 2020 to 2024
$1.1M
NCI NIH HHS K08 CA232344NEI NIH HHS K08 EY030924
6 · The paper itself

Abstract

purposeTo report the clinical and imaging findings of 4 patients with benign intraretinal tumors, 2 of which were associated with retinal pigment epithelium (RPE) hypertrophy. To our knowledge, this condition has not been described previously and should be distinguished from retinoblastoma and other malignant retinal neoplasms.

designRetrospective case series.

participantsFour patients from 3 institutions.

methodsFour patients with intraretinal tumors of the inner nuclear layer (INL) underwent a combination of ophthalmic examination, fundus photography, fluorescein angiography, OCT, OCT angiography, and whole exome sequencing.

main outcome measuresDescription of multimodal imaging findings and systemic findings from 4 patients with benign intraretinal tumors and whole exome studies from 3 patients.

resultsSix eyes of 4 patients 5, 13, 32, and 27 years of age were found to have white intraretinal tumors that remained stable over the follow-up period (range, 9 months-4 years). The tumors were unilateral in 2 patients and bilateral in 2 patients. The tumors were white, centered on the posterior pole, and multifocal, with some consisting of multiple lobules with arching extensions that extended beyond the central tumor mass. OCT demonstrated these lesions to be centered within the INL at the border of the inner plexiform layer. In addition, 2 patients demonstrated congenital hypertrophy of the RPE (CHRPE) lesions. Three of 4 patients underwent whole exome sequencing of the blood that revealed no candidate variants that plausibly could account for the phenotype.

conclusionsWe characterize a novel benign tumor of the INL that, in 2 patients, was associated with separate CHRPE lesions. We propose the term benign lobular inner nuclear layer proliferation to describe these lesions. FINANCIAL DISCLOSURE(S): Proprietary or commercial disclosure may be found after the references.

Indexed as

Retinal DiseasesRetinal NeoplasmsFluorescein AngiographyHumansHypertrophyRetinaRetinal Pigment EpitheliumRetrospective StudiesTomography, Optical CoherenceCongenital hypertrophy of the retinal pigment epitheliumInner nuclear layerRetinal massRetinal tumorRetinoblastoma

Identifiers

PMID36270406
PMCPMC9974858
OpenAlexW4306742354

What OpenQuestion holds

Textmetadata
LicenceTDM
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.