{
  "abstract": "Plasminogen deficiency is an ultra-rare disorder of fibrinolysis that most commonly presents as ligneous conjunctivitis. Ophthalmic manifestations often precede systemic diagnosis, making ophthalmologists essential to early detection and management. We report two contrasting cases illustrating the spectrum of disease and therapeutic response. A 65-year-old woman with untreated type 1 plasminogen deficiency developed recurrent ligneous conjunctivitis leading to enucleation and multisystem morbidity, while a 4-year-old girl with autosomal dominant type 1 disease achieved sustained remission following systemic plasminogen replacement therapy (Ryplazim). These cases highlight the evolution of management from supportive care to disease-modifying therapy and the critical importance of early recognition. Long-term outcomes and treatment durability remain poorly defined, underscoring the need for registry-based collaboration to better characterize disease progression, therapeutic response, and quality-of-life impact in plasminogen deficiency.",
  "abstract_shown": true,
  "affiliations": [
    "Department of Ophthalmology, Albany Medical College, Albany, NY 12208, USA",
    "Department of Ophthalmology, Dean McGee Eye Institute, Oklahoma City, OK 73104, USA",
    "The Jimmy Everest Section of Pediatric Hematology-Oncology, Department of Pediatrics, University of Oklahoma Health Sciences Center, Oklahoma City, OK 73104, USA",
    "Hematology-Oncology Section, Department of Internal Medicine, University of Oklahoma Health Sciences Center, Oklahoma City, OK 73104, USA"
  ],
  "article_type": null,
  "arxiv": null,
  "author_total": 5,
  "authors": [
    {
      "affs": [
        0
      ],
      "name": "Sayeed Khan",
      "orcid": "0009-0001-0565-2118"
    },
    {
      "affs": [
        1
      ],
      "name": "Ashtyn Vogt",
      "orcid": "0000-0001-7065-9079"
    },
    {
      "affs": [
        2
      ],
      "name": "Osman Khan",
      "orcid": "0000-0001-5955-8568"
    },
    {
      "affs": [
        1
      ],
      "name": "Jeremy Tan"
    },
    {
      "affs": [
        3
      ],
      "name": "Sanober Nusrat"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/c7nfnzw3ejkv2.bib",
  "cited_by": 0,
  "doi": "10.3390/ijms27209373",
  "fulltext_url": "https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13705256/?tool=EBI",
  "id": "c7nfnzw3ejkv2",
  "issns": [
    "1422-0067"
  ],
  "landing_url": "https://www.mdpi.com/1422-0067/27/20/9373",
  "language": "eng",
  "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": [
    [
      "openalex",
      "2720|Blood Coagulation and Thrombosis Mechanisms"
    ]
  ],
  "openalex": "W7223272758",
  "pdf_url": "https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13705256/pdf/?tool=EBI",
  "pmcid": "PMC13705256",
  "pmid": null,
  "published": "2026-10-01",
  "publisher": "MDPI AG",
  "ris": "https://research.draconitic.com/paper/c7nfnzw3ejkv2.ris",
  "sources": [
    {
      "first_seen": "2026-10-30T12:18:57Z",
      "native_id": "10.3390/ijms27209373",
      "source": "crossref",
      "url": "https://www.mdpi.com/1422-0067/27/20/9373"
    },
    {
      "first_seen": "2026-10-30T12:21:45Z",
      "native_id": "PMC:PMC13705256",
      "source": "europepmc",
      "url": "https://europepmc.org/article/PMC/PMC13705256"
    }
  ],
  "subjects": [
    {
      "field": "medicine",
      "origin": "openalex:2720|Blood Coagulation and Thrombosis Mechanisms",
      "subfield": "general"
    }
  ],
  "title": "Natural History and Therapeutic Response in Ophthalmic Plasminogen Deficiency: Two Cases and Call for Registry-Based Data",
  "title_text": "Natural History and Therapeutic Response in Ophthalmic Plasminogen Deficiency: Two Cases and Call for Registry-Based Data",
  "type": "peer-reviewed",
  "updated_at": "2026-10-30T12:21:45Z",
  "url": "https://research.draconitic.com/paper/c7nfnzw3ejkv2",
  "venue": "International Journal of Molecular Sciences",
  "versions": []
}