Evidence map›Paper›PMID 42643995›Full record

ArticleIntractable & rare diseases research2026

Skeletal involvement in Joubert syndrome: From low bone mineral density to severe secondary osteoporosis.

Tatsiana Babina, Bogumiła Alicja Górczewska, Elżbieta Jakubowska-Pietkiewicz

Abstract readLetter
In one paragraph

Article in Intractable & rare diseases research, 2026. 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
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.

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

3 authors.

Tatsiana BabinaDepartment of Pediatrics, Neonatal Pathology and Metabolic Bone Diseases, Medical University of Lodz, Lodz, Poland.
Bogumiła Alicja GórczewskaDepartment of Pediatrics, Neonatal Pathology and Metabolic Bone Diseases, Medical University of Lodz, Lodz, Poland.
Elżbieta Jakubowska-PietkiewiczDepartment of Pediatrics, Neonatal Pathology and Metabolic Bone Diseases, Medical University of Lodz, Lodz, Poland.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Joubert syndrome (JS) is a rare ciliopathy characterized by neurological and multisystem manifestations; however, skeletal involvement remains poorly recognized. We report two genetically confirmed pediatric patients with JS illustrating different degrees of bone impairment. The first patient, a 2.5-year-old boy with a TMEM67 mutation, presented with severe hypotonia, profound immobility, multiple fractures, and markedly reduced bone mineral density (DXA Z-score -5.875), fulfilling the criteria for secondary osteoporosis and requiring bisphosphonate therapy. The second patient, a 5-year-old girl with an OFD1 mosaic mutation, showed delayed motor development and reduced bone mineral density (DXA Z-score -2.2) without fractures and was managed conservatively. These cases demonstrate the broad spectrum of skeletal manifestations in JS, ranging from low bone mineral density to severe secondary osteoporosis. Reduced mobility and chronic hypotonia may contribute to impaired bone mineralization. Increased awareness and early bone health surveillance, including fracture assessment and densitometric evaluation, may facilitate timely diagnosis and improve management of bone complications in patients with Joubert syndrome.

Indexed as

fractureshypotoniaJoubert syndromelow bone mineral densitypediatric osteoporosissecondary osteoporosis

Identifiers

PMID42643995
PMCPMC13504545

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.