Evidence map›Paper›PMID 39397911›Full record

ArticleHeliyon2024

Intracranial tumor in a patient with mucopolysaccharidosis type 1 (Scheie syndrome): An extremely rare combination.

Sandhaya Kukreja, Atiqa Imtiaz Soomro, Sapna Lohana, Asifa Kalwar, Sidhant Ochani, Rachna, Md Al Hasibuzzaman

Abstract readCase Reports
In one paragraph

Article in Heliyon, 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. Review
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

7 authors.

Sandhaya KukrejaDepartment of Medicine, Dow University of Health and Sciences, Karachi, Pakistan.
Atiqa Imtiaz SoomroDepartment of Paediatrics, Dr. Ruth K. M. Pfau Civil Hospital Karachi, Pakistan.
Sapna LohanaDepartment of Medicine, Dow University of Health and Sciences, Karachi, Pakistan.
Asifa KalwarDepartment of Medicine, Dow University of Health and Sciences, Karachi, Pakistan.
Sidhant OchaniDepartment of Medicine, Khairpur Medical College, Khairpur Mir's, Pakistan.
RachnaDepartment of Medicine, Ghulam Muhammad Mahar Medical College, Sukkur, Pakistan.
Md Al HasibuzzamanDepartment of Medicine, Niramoy Hospital, Panchagarh, Bangladesh.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Scheie syndrome is a mild variant of mucopolysaccharidosis type I (MPS I), a rare group of lysosomal storage diseases that affect multiple organ systems. It is rarely associated with neoplasia. To the best of our knowledge, only a single case of mucopolysaccharidosis associated with a brain tumor has been reported, and it was nearly three decades ago. We present the case of a 10-year-old female with Scheie syndrome associated with a brain tumor. Physical and laboratory findings were suggestive of Scheie syndrome. A skeletal survey also revealed a spectrum of dysostosis multiplex supporting MPS. Children with MPS can have rapidly enlarging head sizes due to hydrocephalus, but our patient had several red flags that demanded further evaluation. A brain MRI revealed a mass in the fourth ventricle and a biopsy of the mass revealed pilocytic astrocytoma grade 1. Intraventricular pilocytic astrocytoma itself is a rare occurrence, accounting for only 4%-15.6 % of all pilocytic astrocytomas. Altered mucopolysaccharide metabolism can be involved in tumor pathogenesis, but the exact mechanism is unknown. Mucopolysaccharidoses, being a group of complicated disorders, are difficult to manage, and many symptoms can be missed in children due to intellectual disability. This case highlights the importance of suspecting brain tumors in children with mucopolysaccharidoses who present with signs and symptoms of increased intracranial pressure. Prompt diagnosis and management can save the child from dire neurological consequences.

Indexed as

Copper-beaten skullDysostosis multiplexMadelung deformityMucopolysaccharidosisOar/paddle-shaped ribsPilocytic astrocytomaSchiei syndromeTumor

Identifiers

PMID39397911
PMCPMC11471181

What OpenQuestion holds

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LicenceCC BY-NC-ND
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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.