{
  "abstract": "Background: Osteoporosis in young adults warrants evaluation for secondary causes. SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, osteitis) is rare. Severe osteoporosis with fragility fractures as a presenting manifestation of chronic recurrent multifocal osteomyelitis/SAPHO syndrome is exceedingly rare.\n\nCase Report: A 26-year-old man with normal body mass index, no tobacco or alcohol use, and regular physical activity presented with severe osteoporosis after low-trauma fractures of the left arm and L2 and L4 vertebrae following 5 years of back, hip, anterior chest wall pain, and axial stiffness. Initial DXA showed bone mineral density below the expected range for age (lumbar spine Z-score −3.0). Imaging demonstrated sternoclavicular marrow edema, multifocal radiotracer uptake, and bilateral sacroiliitis. Laboratory evaluation showed elevated ESR, CRP, IgA, and HLA-B27 positivity; available metabolic bone evaluation was otherwise unrevealing. Genetic testing identified an ALPK1 variant of uncertain significance. Isolated axial spondyloarthritis, infection, and malignancy were less likely because of multifocal sterile osteitis, anterior chest wall involvement, chronic course, and absence of systemic infectious or malignant features. He received adalimumab and completed 3 pamidronate infusion cycles. Through 2026 follow-up, ESR/CRP normalized on 3 checks, back pain resolved, no interim falls or fractures occurred, lumbar fractures showed radiographic healing, and repeat DXA was clinically stable.\n\nDiscussion: Chronic recurrent multifocal osteomyelitis/SAPHO syndrome may cause secondary osteoporosis through inflammation-mediated osteoclast activation and impaired bone formation.\n\nConclusion: Consider autoinflammatory bone disease in young adults with unexplained osteoporosis, fragility fractures, axial symptoms, anterior chest wall pain, or sacroiliitis.",
  "abstract_shown": true,
  "affiliations": [
    "Department of Internal Medicine, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania",
    "Division of Endocrinology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania"
  ],
  "article_type": "case report",
  "arxiv": null,
  "author_total": 6,
  "authors": [
    {
      "affs": [
        0
      ],
      "name": "Sani Yasnain"
    },
    {
      "affs": [
        0
      ],
      "name": "Sandhya Shanthosh Kumar"
    },
    {
      "affs": [
        0
      ],
      "name": "Dur E Shahwar"
    },
    {
      "affs": [
        0
      ],
      "name": "Saira Tanveer"
    },
    {
      "affs": [
        0
      ],
      "name": "Ezza Wasim"
    },
    {
      "affs": [
        1
      ],
      "name": "Niveditha Manivannan"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/edvw6tf4bpqw8.bib",
  "cited_by": 0,
  "doi": "10.1016/j.aed.2026.07.009",
  "fulltext_url": "https://pmc.ncbi.nlm.nih.gov/articles/PMC13728276/",
  "id": "edvw6tf4bpqw8",
  "issns": [
    "3050-9157"
  ],
  "landing_url": "https://doi.org/10.1016/j.aed.2026.07.009",
  "language": "en",
  "licence": "CC-BY-NC-ND-4.0",
  "licence_label": "CC BY-NC-ND 4.0",
  "licence_source": "article",
  "licence_url": "https://creativecommons.org/licenses/by-nc-nd/4.0/",
  "metadata_licence": null,
  "native_subjects": [
    [
      "heading",
      "Case Report"
    ],
    [
      "openalex",
      "2745|Osteomyelitis and Bone Disorders Research"
    ]
  ],
  "openalex": "W7171618720",
  "pdf_url": "https://www.endocrinologydiabetes.org/article/S3050-9157(26)00156-6/pdf",
  "pmcid": "PMC13728276",
  "pmid": "42979057",
  "published": "2026-07-29",
  "publisher": "American Association of Clinical Endocrinology",
  "ris": "https://research.draconitic.com/paper/edvw6tf4bpqw8.ris",
  "sources": [
    {
      "first_seen": "2026-11-06T12:23:59Z",
      "native_id": "PMC13728276",
      "source": "pmc",
      "url": "https://doi.org/10.1016/j.aed.2026.07.009"
    }
  ],
  "subjects": [
    {
      "field": "medicine",
      "origin": "journal-openalex:2712|Thyroid Disorders and Treatments",
      "subfield": "endocrinology-metabolism"
    }
  ],
  "title": "Severe Early-Onset Osteoporosis and Fragility Fractures in a Young Male With Chronic Recurrent Multifocal Osteomyelitis Within the SAPHO Spectrum",
  "title_text": "Severe Early-Onset Osteoporosis and Fragility Fractures in a Young Male With Chronic Recurrent Multifocal Osteomyelitis Within the SAPHO Spectrum",
  "type": "peer-reviewed",
  "updated_at": "2026-11-07T12:21:18Z",
  "url": "https://research.draconitic.com/paper/edvw6tf4bpqw8",
  "venue": "AACE Endocrinology and Diabetes",
  "versions": []
}