{
  "abstract": "Introduction: Osteogenesis imperfecta (OI) is a rare inherited disorder characterized by skeletal fragility and recurrent fractures. Intravenous pamidronate is widely used in pediatric OI, but long-term real-world data from Eastern Europe remain limited. We compared fracture incidence before and after pamidronate initiation in Romanian children with OI.\n\nMethods: We conducted a 20-year retrospective multicenter before-and-after cohort study in three tertiary pediatric centers in Bucharest, Romania. Fracture counts and person-years before and after treatment initiation were analyzed using negative binomial generalized estimating equations (GEE), accounting for repeated observations within patients and including log person-years as an offset. The model included treatment period, Sillence type, sex, and age at pamidronate initiation.\n\nResults: Forty-nine children with OI receiving intravenous pamidronate were included. The cohort included 28 females (57.1%) and 21 males (42.9%). Median age at diagnosis was 24.5 months (IQR, 0.25–81.25), and pamidronate was initiated at a median age of 45 months (IQR, 15.5–95). Before initiation, 198 fractures occurred over 250.3 person-years, compared with 208 fractures over 359.3 person-years afterward, corresponding to incidence rates of 0.79 and 0.58 fractures per person-year, respectively. In the adjusted GEE model, fracture incidence was lower during the post-pamidronate period (IRR 0.49, 95% CI 0.35–0.68; p < 0.001). Compared with type I OI, fracture incidence was higher in type III (IRR 2.09, 95% CI 1.49–2.92; p < 0.001) and type IV (IRR 1.72, 95% CI 1.01–2.94; p = 0.048). Sex was not associated with fracture incidence. Older age at pamidronate initiation was associated with lower fracture incidence (IRR 0.992 per month, 95% CI 0.989–0.996; p < 0.001). Adjustment for corrective osteotomies with telescopic intramedullary rodding procedures did not materially change the findings.\n\nConclusions: Fracture incidence was substantially lower after pamidronate initiation, including after accounting for repeated within-patient observations and clinical characteristics. Although the observational design prevents causal attribution to pamidronate, these findings provide long-term real-world data from an Eastern European pediatric OI cohort and support prospective studies incorporating age-dependent fracture risk and time-varying exposures.",
  "abstract_shown": true,
  "affiliations": [
    "Department of Pediatrics, “Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania",
    "Department of Pediatrics, Emergency Hospital for Children “Grigore Alexandrescu”, Bucharest, Romania",
    "Department of Pediatric Orthopedics, “Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania",
    "Department of Pediatric Orthopedics, Emergency Hospital for Children “Grigore Alexandrescu”, Bucharest, Romania"
  ],
  "article_type": "research article",
  "arxiv": null,
  "author_total": 6,
  "authors": [
    {
      "affs": [
        0,
        1
      ],
      "name": "Alexandru Dinulescu"
    },
    {
      "affs": [
        0,
        1
      ],
      "name": "Mara-Elena Stăiculescu"
    },
    {
      "affs": [
        0,
        1
      ],
      "name": "Irina Dijmărescu"
    },
    {
      "affs": [
        0,
        1
      ],
      "name": "Mirela-Luminița Pavelescu"
    },
    {
      "affs": [
        0,
        1
      ],
      "name": "Daniela Păcurar"
    },
    {
      "affs": [
        2,
        3
      ],
      "name": "Alexandru Ulici"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/1st93k2rmac93.bib",
  "cited_by": 0,
  "doi": "10.3389/fped.2026.1973795",
  "fulltext_url": "https://pmc.ncbi.nlm.nih.gov/articles/PMC13731108/",
  "id": "1st93k2rmac93",
  "issns": [
    "2296-2360"
  ],
  "landing_url": "https://doi.org/10.3389/fped.2026.1973795",
  "language": "en",
  "licence": "CC-BY-4.0",
  "licence_label": "CC BY 4.0",
  "licence_source": "article",
  "licence_url": "https://creativecommons.org/licenses/by/4.0/",
  "metadata_licence": null,
  "native_subjects": [
    [
      "heading",
      "Original Research"
    ],
    [
      "openalex",
      "1311|Connective tissue disorders research"
    ]
  ],
  "openalex": "W7223158125",
  "pdf_url": "https://www.frontiersin.org/journals/pediatrics/articles/10.3389/fped.2026.1973795/pdf",
  "pmcid": "PMC13731108",
  "pmid": "42984663",
  "published": "2026-10-21",
  "publisher": "Frontiers Media SA",
  "ris": "https://research.draconitic.com/paper/1st93k2rmac93.ris",
  "sources": [
    {
      "first_seen": "2026-11-08T12:21:21Z",
      "native_id": "PMC13731108",
      "source": "pmc",
      "url": "https://doi.org/10.3389/fped.2026.1973795"
    }
  ],
  "subjects": [
    {
      "field": "biology",
      "origin": "openalex:1311|Connective tissue disorders research",
      "subfield": "genetics-genomics"
    }
  ],
  "title": "Fracture outcomes following intravenous pamidronate initiation in children with osteogenesis imperfecta: a multicenter retrospective cohort study from Bucharest, Romania",
  "title_text": "Fracture outcomes following intravenous pamidronate initiation in children with osteogenesis imperfecta: a multicenter retrospective cohort study from Bucharest, Romania",
  "type": "peer-reviewed",
  "updated_at": "2026-11-08T12:21:21Z",
  "url": "https://research.draconitic.com/paper/1st93k2rmac93",
  "venue": "Frontiers in Pediatrics",
  "versions": []
}