Evidence map›Paper›PMID 35221874›Full record

ArticleMolecular syndromology2022

What Can We Learn from the Parents of Children Affected with Mucopolysaccharidosis Type III-A in Israel?

Shiri Liber, Orna Staretz-Chacham, Mor Kishon, Ben Pode-Shakked, Odelia Chorin, Katya Kneller, Yair Anikster, Geto Mangisto, Ann Saada, Annick Raas-Rothschild

Open access · bronzeAbstract read
In one paragraph

Article in Molecular syndromology, 2022. 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, top 79% 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

0 citing papers in PubMed, 1 citations in OpenAlex.

No citing paper in PubMed yet.

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 6 institutions in 1 country.

Shiri LiberThe Arrow Project, Sheba Medical Center, Tel-Hashomer, Israel.
Orna Staretz-ChachamMetabolic Clinic, Pediatric Division, Soroka Medical Center, Ben-Gurion University, Beer Sheva, Israel.
Mor KishonThe Institute for Rare Diseases, Edmond and Lily Safra Children Hospital, Sheba Medical Center, Tel-Hashomer, Israel.
Ben Pode-ShakkedThe Institute for Rare Diseases, Edmond and Lily Safra Children Hospital, Sheba Medical Center, Tel-Hashomer, Israel.
Odelia ChorinThe Institute for Rare Diseases, Edmond and Lily Safra Children Hospital, Sheba Medical Center, Tel-Hashomer, Israel.
Katya KnellerSackler Faculty of Medicine, Tel-Aviv University, Tel-Aviv, Israel.
Yair AniksterSackler Faculty of Medicine, Tel-Aviv University, Tel-Aviv, Israel.
Geto MangistoDepartment of Genetics, Hadassah Medical Center, Jerusalem, Israel.
Ann SaadaDepartment of Genetics, Hadassah Medical Center, Jerusalem, Israel.
Annick Raas-RothschildThe Institute for Rare Diseases, Edmond and Lily Safra Children Hospital, Sheba Medical Center, Tel-Hashomer, Israel.
Sheba Medical Center · ILEdmond and Lily Safra Children's Hospital · ILHadassah Medical Center · ILHebrew University of Jerusalem · ILSoroka Medical Center · ILTel Aviv University · IL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Sanfilippo Syndrome, or mucopolysaccharidosis type III (MPS III), is a group of autosomal-recessive lysosomal storage disorders leading to tissue accumulation of heparan sulfate. MPS III is caused by deficiency in one of 4 enzymes involved in lysosomal degradation of heparan sulfate. Based on the relevant enzyme deficiency, 4 types have been recognized. MPS III constitutes a progressive neurodegenerative and systemic disorder. Parents of children diagnosed with MPS III were interviewed using a retrospective questionnaire based on the known clinical manifestations of MPS III. Eight patients from 4 unrelated families of varied ethnic origin were included. All children were diagnosed with MPS type III-A. Average age at diagnosis was 6.1 years. The most common early clinical manifestations leading to parental suspicion of illness were speech delay and coarse facial features. All children were reported to have global developmental delay, sleep disorders, recurrent infections, hyperactivity, and decreased hearing. The time from first medical inquiry until diagnosis was over 2 years on average, consistent with the delay in diagnosis described in the literature. MPS III children frequently undergo early and repeated ear, nose and throat surgeries, thus we suggest that a high index of suspicion is warranted in relevant clinical circumstances.

Indexed as

Lysosomal storage diseaseMucopolysaccharidosis type IIINatural historyParental experienceSanfilippo syndrome

Identifiers

PMID35221874
PMCPMC8832230
OpenAlexW4200201926

What OpenQuestion holds

Textmetadata
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.