Evidence map›Paper›PMID 27699153›Full record

ArticleMolecular genetics and metabolism reports2016

Bone mineral density in mucopolysaccharidosis IVB.

Francyne Kubaski, Heidi H Kecskemethy, H Theodore Harcke, Shunji Tomatsu

Open access · goldAbstract read
In one paragraph

Article in Molecular genetics and metabolism reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
0.8field-weighted citation impact, top 25% 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

6 citing papers in PubMed, 12 citations in OpenAlex.

  1. Article
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  4. Article
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  6. 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

4 authors at 1 institution in 1 country.

Francyne KubaskiDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, 1600 Rockland Road, Wilmington, DE 19803, USA; Department of Biological Sciences, University of Delaware, 118 Wolf Hall, Newark, DE 19716, USA.
Heidi H KecskemethyDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, 1600 Rockland Road, Wilmington, DE 19803, USA; Department of Medical Imaging, Nemours/Alfred I. duPont Hospital for Children, 1600 Rockland Road, Wilmington, DE 19803, USA.
H Theodore HarckeDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, 1600 Rockland Road, Wilmington, DE 19803, USA; Department of Radiology and Pediatrics, Jefferson Medical College, Thomas Jefferson University, 901 Walnut St, Philadelphia, PA 19107, USA.
Shunji TomatsuDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, 1600 Rockland Road, Wilmington, DE 19803, USA; Department of Biological Sciences, University of Delaware, 118 Wolf Hall, Newark, DE 19716, USA.
Alfred I. duPont Hospital for Children · US

Funding

Newborn Screening and Biomarkers for MucopolysaccharidosesR01HD065767 · NICHD · SAINT LOUIS UNIVERSITY · PI MONTANO, ADRIANA MARIA, TOMATSU, SHUNJI · 2011 to 2015
$2.4M
Oral tolerance in enzyme replacement therapy of Morquio A diseaseR03HD064749 · NICHD · SAINT LOUIS UNIVERSITY · PI MONTANO, ADRIANA MARIA, TOMATSU, SHUNJI · 2010 to 2011
$145k
NICHD NIH HHS R01 HD065767NICHD NIH HHS R03 HD064749
6 · The paper itself

Abstract

To date, the only published reports of bone mineral density (BMD) in MPS IV involve patients with MPS IVA; no reports exist describing BMD for MPS IVB. In this prospective study of BMD in three patients with MPS IVB, BMD was acquired by dual-energy X-ray absorptiometry (DXA) at whole body (WB), lumbar spine (LS), and lateral distal femur (LDF). Functional abilities, ambulatory status, medical history, and height z-score were evaluated. Three patients with MPS IVB (two females), aged 17.7, 31.4 and 31.7 years, were evaluated. Every patient was ambulatory and one sustained two fractures caused by trauma. Whole body and hip DXA scans were technically invalid in every patient due to the presence of prosthetic hip hardware. Lumbar spine was valid in only 1 patient due skeletal abnormalities, and was normal (Z-score of - 0.8). The LDF was valid in every patient and was low at all three regions of interest: average LDF z-scores were - 3.1 (range, - 2.9 to - 3.6), - 2.3 (range, - 2.0 to - 2.5), and - 2.1 (range, - 2.0 to - 2.3) for region 1-region 3, respectively. Patients with MPS IVB have low BMD of the lower extremities even with full-time ambulation. Routine body sites to measure by DXA were problematic; hip and WB were invalid due to artifact, and LS had limited utility. The LDF was the only body site consistently available on all patients. Patients did not experience low-energy fractures despite low BMD.

Indexed as

BMD, bone mineral densityBone mineral densityDual-energy X-ray absorptiometryDXA, dual energy X-ray absorptiometryGAGs, glycosaminoglycansGLB1, betagalactosideaseHAZ, height-adjusted Z-scoreHGMD, The Human Gene Mutation DatabaseKS, keratan sulfateLateral distal femur dual-energy X-ray absorptiometryLDF, lateral distal femurLS, lumbar spineMPS IVB, mucopolysaccharidosis type IV BMucopolysaccharidosis type BNHANES, National Health and Nutrition SurveyWB, whole body

Identifiers

PMID27699153
PMCPMC5036531
OpenAlexW2480895532

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.