Evidence map›Paper›PMID 27687687›Full record

ArticleScientific reports2016

Bisphosphonates in multicentric osteolysis, nodulosis and arthropathy (MONA) spectrum disorder - an alternative therapeutic approach.

Karin Pichler, Daniela Karall, Dieter Kotzot, Elisabeth Steichen-Gersdorf, Alexandra Rümmele-Waibel, Laureane Mittaz-Crettol, Julia Wanschitz, Luisa Bonafé, Kathrin Maurer, Andrea Superti-Furga and 1 more

Open access · goldAbstract read
In one paragraph

Article in Scientific reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
18citing papers in PubMed, 1 pooled it
2.1field-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

18 citing papers in PubMed, 1 synthesis or guideline pooled it, 20 citations in OpenAlex.

  1. Pooled it
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  12. Multicentric Carpo-Tarsal Osteolysis.Journal of the Belgian Society of Radiology · 2023
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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

11 authors at 5 institutions in 2 countries.

Karin PichlerClinic for Pediatrics I, Inherited Metabolic Disorders, Medical University of Innsbruck, Anichstrasse 35, A-6020 Innsbruck, Austria.
Daniela KarallClinic for Pediatrics I, Inherited Metabolic Disorders, Medical University of Innsbruck, Anichstrasse 35, A-6020 Innsbruck, Austria.
Dieter KotzotDepartment of Human Genetics, Medical University of Innsbruck, Peter-Mayr-Strasse 1/1, A-6020 Innsbruck, Austria.
Elisabeth Steichen-GersdorfClinic for Pediatrics I, Inherited Metabolic Disorders, Medical University of Innsbruck, Anichstrasse 35, A-6020 Innsbruck, Austria.
Alexandra Rümmele-WaibelDepartment of Pediatrics, Dornbirn General Hospital, Lustenauerstrasse 4, A-6850 Dornbirn, Austria.
Laureane Mittaz-CrettolCenter for Molecular Diseases, Lausanne University Hospital, Av. P. Decker 2, CH-1011 Lausanne, Switzerland.
Julia WanschitzDepartment of Neurology, Medical University of Innsbruck, Anichstrasse 35, A-6020 Innsbruck, Austria.
Luisa BonaféCenter for Molecular Diseases, Lausanne University Hospital, Av. P. Decker 2, CH-1011 Lausanne, Switzerland.
Kathrin MaurerDepartment of Radiology, Medical University of Innsbruck, Anichstrasse 35, A-6020 Innsbruck, Austria.
Andrea Superti-FurgaDepartment of Pediatrics, University of Lausanne, Rue du Bugnon 46, CH-1011 Lausanne, Switzerland.
Sabine Scholl-BürgiClinic for Pediatrics I, Inherited Metabolic Disorders, Medical University of Innsbruck, Anichstrasse 35, A-6020 Innsbruck, Austria.
Universität Innsbruck · ATUniversity of Lausanne · CHInnsbruck Medical University · ATKrankenhaus Oberndorf · ATMedical University of Vienna · AT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Multicentric osteolysis, nodulosis and arthropathy (MONA) spectrum disorder is a rare inherited progressive skeletal disorder caused by mutations in the matrix metalloproteinase 2 (MMP2) gene. Treatment options are limited. Herein we present successful bisphosphonate therapy in three affected patients. Patients were treated with bisphosphonates (either pamidronate or zoledronate) for different time periods. The following outcome variables were assessed: skeletal pain, range of motion, bone densitometry, internal medical problems as well as neurocognitive function. Skeletal pain was dramatically reduced in all patients soon after initiation of therapy and bone mineral density increased. Range of motion did not significantly improve. One patient is still able to walk with aids at the age of 14 years. Neurocognitive development was normal in all patients. Bisphosphonate therapy was effective especially in controlling skeletal pain in MONA spectrum disorder. Early initiation of treatment seems to be particularly important in order to achieve the best possible outcome.

Identifiers

PMID27687687
PMCPMC5043187
OpenAlexW2526151079

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.