Evidence map›Paper›PMID 36185913›Full record

ArticleCureus2022

Radiological Findings of Woodhouse-Sakati Syndrome: Cases Reported From Saudi Arabia.

Arwa M Alzahrani, Lamis O Alsuwailem, Rinad M Alghoraiby, Fahad B Albadr, Yahya M Alaseri

Abstract readCase Reports
In one paragraph

Article in Cureus, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

3 citing papers in PubMed.

  1. Article
  2. Article
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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

5 authors.

Arwa M AlzahraniDepartment of Radiology and Medical Imaging, King Saud University Medical City/College of Medicine, King Saud University, Riyadh, SAU.
Lamis O AlsuwailemDepartment of Radiology and Medical Imaging, King Saud University Medical City/College of Medicine, King Saud University, Riyadh, SAU.
Rinad M AlghoraibyDepartment of Radiology and Medical Imaging, King Saud University Medical City/College of Medicine, King Saud University, Riyadh, SAU.
Fahad B AlbadrDepartment of Radiology and Medical Imaging, King Saud University Medical City/College of Medicine, King Saud University, Riyadh, SAU.
Yahya M AlaseriDepartment of Radiology, King Abdulaziz Medical City, Riyadh, SAU.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Woodhouse-Sakati syndrome (WSS) is a rare autosomal recessive neurodegenerative genetic disorder caused by mutations in the DCAF17 gene. It primarily manifests with endocrinological symptoms such as hypogonadism, failure to develop secondary sexual characteristics, diabetes, and hypotrichosis. Neurological manifestations include intellectual disabilities, dystonia, dysarthria, and hearing loss. This paper describes the cases of two Saudi Arabian sisters, aged 37 and 36, who were born to first-degree consanguineous parents. They had normal growth and development except for certain intellectual disabilities. However, they were presented with primary amenorrhea and no secondary sexual characteristics at puberty, and they were subsequently diagnosed with WSS. The first patient presented with dysmorphic features, dysarthria, tremors, and dystonia. The second patient presented with hypotrichosis, predominantly affecting the temporo-occipital regions, and cerebellar signs on physical exam. Both patients had hair thinning and bilateral sensorineural hearing loss. Brain MRI of both patients showed increased iron deposition in the basal ganglia and multiple faint T2-FLAIR (fluid-attenuated inversion recovery) hyperintensity foci involving the centrum semiovale, corona radiata, and peritrigonal white matter bilaterally. MRI abdomen of the second patient revealed early hepatic fibrosis, with diffuse moderate to severe hepatic steatosis reaching a fat fraction of 19%, and increased intensity of the splenic vein with multiple collaterals. Further research is needed to achieve a better understanding of this syndrome to improve patient care and outcomes.

Indexed as

geneticsmrineurological disordersneuroradiologywoodhouse-sakati syndrome

Identifiers

PMID36185913
PMCPMC9522504

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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.