Evidence map›Paper›PMID 36544157›Full record

ArticleOrphanet journal of rare diseases2022

Growth hormone treatment improves final height in children with X-linked hypophosphatemia.

Julia André, Volha V Zhukouskaya, Anne-Sophie Lambert, Jean-Pierre Salles, Brigitte Mignot, Claire Bardet, Catherine Chaussain, Anya Rothenbuhler, Agnès Linglart

Open access · goldAbstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed, 1 pooled it
2.9field-weighted citation impact, top 8% 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

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

  1. Guideline
  2. Article
  3. Article
  4. Article
  5. Article
  6. Review
  7. Article
  8. Review
  9. The Diagnosis and Therapy of XLH.Calcified tissue international · 2025
    Review
  10. Article
  11. Article
  12. X-linked hypophosphatemic rickets: from diagnosis to management.Clinical and experimental pediatrics · 2024
    Article
  13. Observational
  14. 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

9 authors at 5 institutions in 1 country.

Julia André *AP-HP, Endocrinology and Diabetes for Children, Reference Center for Rare Diseases of Calcium and Phosphate Metabolism, DMU SEA, OSCAR Filière, EndoRare and BOND ERN, Bicêtre Paris Saclay Hospital, 78 Rue du Général Leclerc, 94270, Le Kremlin Bicêtre, France.
Volha V Zhukouskaya *AP-HP, Endocrinology and Diabetes for Children, Reference Center for Rare Diseases of Calcium and Phosphate Metabolism, DMU SEA, OSCAR Filière, EndoRare and BOND ERN, Bicêtre Paris Saclay Hospital, 78 Rue du Général Leclerc, 94270, Le Kremlin Bicêtre, France. volha.zhukouskaya@aphp.fr.ORCID 0000-0002-2435-1855
Anne-Sophie LambertAP-HP, Endocrinology and Diabetes for Children, Reference Center for Rare Diseases of Calcium and Phosphate Metabolism, DMU SEA, OSCAR Filière, EndoRare and BOND ERN, Bicêtre Paris Saclay Hospital, 78 Rue du Général Leclerc, 94270, Le Kremlin Bicêtre, France.
Jean-Pierre SallesUnit of Endocrinology and Bone Diseases, Children Hospital, Toulouse University Hospital, CHU de Toulouse, Université de Toulouse, ERN BOND, INSERM UMR 1291/CNRS 5051, INFINITY Center, Toulouse, France.
Brigitte MignotDepartment of Pediatrics, CHU of Besancon, Besançon, France.
Claire BardetLaboratory Orofacial Pathologies, Imaging and Biotherapies URP2496 and FHU-DDS-Net, Dental School, and Plateforme d'Imagerie du Vivant (PIV), Université Paris Cité, Montrouge, France.
Catherine ChaussainAP-HP, Endocrinology and Diabetes for Children, Reference Center for Rare Diseases of Calcium and Phosphate Metabolism, DMU SEA, OSCAR Filière, EndoRare and BOND ERN, Bicêtre Paris Saclay Hospital, 78 Rue du Général Leclerc, 94270, Le Kremlin Bicêtre, France.
Anya RothenbuhlerAP-HP, Endocrinology and Diabetes for Children, Reference Center for Rare Diseases of Calcium and Phosphate Metabolism, DMU SEA, OSCAR Filière, EndoRare and BOND ERN, Bicêtre Paris Saclay Hospital, 78 Rue du Général Leclerc, 94270, Le Kremlin Bicêtre, France.
Agnès LinglartAP-HP, Endocrinology and Diabetes for Children, Reference Center for Rare Diseases of Calcium and Phosphate Metabolism, DMU SEA, OSCAR Filière, EndoRare and BOND ERN, Bicêtre Paris Saclay Hospital, 78 Rue du Général Leclerc, 94270, Le Kremlin Bicêtre, France.
Assistance Publique – Hôpitaux de Paris · FRUniversité Paris Cité · FRCentre Hospitalier Universitaire de Besançon · FRCentre National de la Recherche Scientifique · FRInserm · FR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND/

aimDespite optimal conventional treatment (oral phosphate supplements and active vitamin D analogs), about 40-50% of children with well-controlled X-linked hypophosphatemia (XLH) show linear growth failure, making them less likely to achieve an acceptable final height. Here, we studied the hypothesis that rhGH treatment improves final height in children with XLH and growth failure.

methodsTwo cohorts of children with XLH were included in this retrospective longitudinal analysis: (1) a cohort treated with rhGH for short stature (n = 34) and (2) a cohort not treated with rhGH (n = 29). The mean duration of rhGH treatment was 4.4 ± 2.9 years. We collected the auxological parameters at various time points during follow-up until final height.

resultsIn rhGH-treated children, 2 years of rhGH therapy was associated with a significant increase in height from - 2.4 ± 0.9 to - 1.5 ± 0.7 SDS (p < 0.001). Their mean height at rhGH discontinuation was - 1.2 ± 0.9 SDS and at final height was - 1.3 ± 0.9 SDS corresponding to 165.5 ± 6.4 cm in boys and 155.5 ± 6.3 cm in girls. Notably, the two groups had similar final heights; i.e., the final height in children not treated with rhGH being - 1.2 ± 1.1 SDS (165.4 ± 6.8 cm in boys and 153.7 ± 7.8 cm in girls), p = 0.7.

conclusionTreatment with rhGH permits to improve final height in children with XLH and growth failure, despite optimal conventional treatment. We propose therefore that rhGH therapy could be considered as an option for short stature in the context of XLH.

Indexed as

DwarfismFamilial Hypophosphatemic RicketsHuman Growth HormoneBody HeightChildFemaleGrowth DisordersGrowth HormoneHumansMaleRetrospective StudiesGrowth HormoneHuman Growth HormoneFibroblast growth factor 23Final heightRecombinant human growth hormoneRicketsX-linked hypophosphatemia

Identifiers

PMID36544157
PMCPMC9768884
OpenAlexW4312069956

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

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.