Evidence map›Paper›PMID 28294978›Full record

ReviewInternational journal of molecular sciences2017

Discriminative Features in Three Autosomal Recessive Cutis Laxa Syndromes: Cutis Laxa IIA, Cutis Laxa IIB, and Geroderma Osteoplastica.

Ariana Kariminejad, Fariba Afroozan, Bita Bozorgmehr, Alireza Ghanadan, Susan Akbaroghli, Hamid Reza Khorram Khorshid, Faezeh Mojahedi, Aria Setoodeh, Abigail Loh, Yu Xuan Tan and 5 more

Open access · goldAbstract readCase ReportsReview
In one paragraph

Review in International journal of molecular sciences, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed
2.4field-weighted citation impact, top 11% 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

11 citing papers in PubMed, 27 citations in OpenAlex.

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  7. The role of the Golgi apparatus in disease (Review).International journal of molecular medicine · 2021
    Review
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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

15 authors at 8 institutions in 5 countries.

Ariana KariminejadKariminejad-Najmabadi Pathology & Genetics Center, #2, 4th Street, Hasan Seyf Street, Sanat Square, Tehran 14667-13713, Iran. arianakariminejad@yahoo.com.
Fariba AfroozanKariminejad-Najmabadi Pathology & Genetics Center, #2, 4th Street, Hasan Seyf Street, Sanat Square, Tehran 14667-13713, Iran. afroozan@yahoo.com.
Bita BozorgmehrKariminejad-Najmabadi Pathology & Genetics Center, #2, 4th Street, Hasan Seyf Street, Sanat Square, Tehran 14667-13713, Iran. b_bzwr@yahoo.com.
Alireza GhanadanDepartment of Dermatopathology, Razi Dermatology Hospital, Tehran University of Medical Sciences, Tehran 14167-53955, Iran. dermpath101@gmail.com.
Susan AkbaroghliClinical Genetics Division, Mofid Children's Hospital, Faculty of Medicine, Shahid Beheshti University of Medical Sciences, Tehran 15514-15468, Iran. susanakbaroghli@yahoo.com.
Hamid Reza Khorram KhorshidGenetic Research Centre, University of Social Welfare and Rehabilitation Sciences, Tehran 19857-13834, Iran. hrkhkh@yahoo.com.
Faezeh MojahediMashhad Medical Genetic Counseling Center, Social Welfare and Rehabilitation Organization, Mashhad 91767-61999, Iran. mojahedi1381@yahoo.com.
Aria SetoodehDivision of Pediatric Endocrinology and Inherited Metabolic Disorders, Department of Pediatrics, Tehran University of Medical Sciences, Tehran 14197-33141, Iran. arset59@yahoo.com.
Abigail LohInstitute of Medical Biology, A*STAR, Singapore 138648, Singapore. abigail.loh@reversade.com.
Yu Xuan TanInstitute of Medical Biology, A*STAR, Singapore 138648, Singapore. t_yuxuan@yahoo.com.sg.
Nathalie Escande-BeillardInstitute of Medical Biology, A*STAR, Singapore 138648, Singapore. nathalie.escande@reversade.com.
Fransiska MalfaitCenter for Metabolic Diseases, Department of Pediatrics, University Hospitals Leuven, Leuven 3000, Belgium. Fransiska.Malfait@UGent.be.
Bruno ReversadeInstitute of Medical Biology, A*STAR, Singapore 138648, Singapore. bruno@reversade.com.
Thatjana GardeitchikDepartment of Pediatrics, Radboud University Nijmegen Medical Center, Nijmegen, Gelderland 9102-6500, The Netherlands. Thatjana.Gardeitchik@radboudumc.nl.
Eva MoravaDepartment of Pediatrics, Radboud University Nijmegen Medical Center, Nijmegen, Gelderland 9102-6500, The Netherlands. emoravakozicz@tulane.edu.
Agency for Science, Technology and Research · SGImam Khomeini Hospital · IRKU Leuven · BERadboud University Nijmegen · NLShahid Beheshti University of Medical Sciences · IRTehran University of Medical Sciences · IRTulane University · USUniversity of Social Welfare and Rehabilitation Sciences · IR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Cutis laxa is a heterogeneous condition characterized by redundant, sagging, inelastic, and wrinkled skin. The inherited forms of this disease are rare and can have autosomal dominant, autosomal recessive, or X-linked inheritance. Three of the autosomal recessive cutis laxa syndromes, namely cutis laxa IIA (ARCL2A), cutis laxa IIB (ARCL2B), and geroderma osteodysplastica (GO), have very similar clinical features, complicating accurate diagnosis. Individuals with these conditions often present with cutis laxa, progeroid features, and hyperextensible joints. These conditions also share additional features, such as short stature, hypotonia, and congenital hip dislocation, but the severity and frequency of these findings are variable in each of these cutis laxa syndromes. The characteristic features for ARCL2A are abnormal isoelectric focusing and facial features, including downslanting palpebral fissures and a long philtrum. Rather, the clinical phenotype of ARCL2B includes severe wrinkling of the dorsum of the hands and feet, wormian bones, athetoid movements, lipodystrophy, cataract and corneal clouding, a thin triangular face, and a pinched nose. Normal cognition and osteopenia leading to pathological fractures, maxillary hypoplasia, and oblique furrowing from the outer canthus to the lateral border of the supraorbital ridge are discriminative features for GO. Here we present 10 Iranian patients who were initially diagnosed clinically using the respective features of each cutis laxa syndrome. Each patient's clinical diagnosis was then confirmed with molecular investigation of the responsible gene. Review of the clinical features from the cases reported from the literature also supports our conclusions.

Indexed as

PhenotypeAdultBone DiseasesChildChild, PreschoolCutis LaxaDiagnosis, DifferentialDwarfismFemaleHumansInfantMaleSkin Diseases, GeneticSyndromeATPase, H+ transporting lysosomal V0 subunit A2 (ATP6V0A2)autosomal recessive cutis laxa 2Aautosomal recessive cutis laxa 2Bgeroderma osteodysplasticaGOLGIN, RAB6-INTERACTING (GORAB)Pyrroline-5-carboxylate reductase 1 (PYCR1)

Identifiers

PMID28294978
PMCPMC5372648
OpenAlexW2595948873

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.