{
  "abstract": "Acquired generalized lipodystrophy (AGL) is a rare disorder characterized by progressive loss of subcutaneous and visceral adipose tissue and severe metabolic disturbances, including diabetes mellitus (DM). Although AGL is typically associated with type 2 DM, clinical experience with patients affected by both AGL and Fulminant type 1 DM (FT1DM) remains limited. We report a case of a 50-year-old man with long-standing (4-year) FT1DM who developed AGL, with histopathological and immunological evaluation and long-term follow-up after metreleptin therapy. The patient presented with rapid deterioration of glycemic control and extreme insulin resistance, requiring a marked increase in insulin dose. Imaging studies revealed generalized loss of subcutaneous and visceral adipose tissue, and skin biopsy demonstrated marked adipose tissue atrophy with infiltration of CD3+ (cluster of differentiation 3) and CD8+ T lymphocytes. Notably, soluble interleukin-2 receptor (sIL-2R) levels remained persistently elevated for more than 12 months, suggesting sustained systemic T-cell activation. Although metreleptin therapy resulted in partial improvement in glycemic control, hemoglobin A1c (HbA1c) did not return to the pre-AGL level during follow-up. This case raises the possibility that persistent immune activation may contribute to insulin resistance beyond leptin deficiency in some patients with AGL.",
  "abstract_shown": true,
  "affiliations": [],
  "article_type": null,
  "arxiv": null,
  "author_total": 6,
  "authors": [
    {
      "name": "Toshitaka Sawamura"
    },
    {
      "name": "Shigehiro Karashima"
    },
    {
      "name": "Yuya Nishimoto"
    },
    {
      "name": "Ai Ohmori"
    },
    {
      "name": "Mitsuhiro Kometani"
    },
    {
      "name": "Takashi Yoneda"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/4ezfhena2cd54.bib",
  "cited_by": 0,
  "doi": "10.1507/endocrj.ej26-0240",
  "fulltext_url": "https://www.jstage.jst.go.jp/article/endocrj/advpub/0/advpub_EJ26-0240/_article",
  "id": "4ezfhena2cd54",
  "issns": [
    "1348-4540",
    "0918-8959"
  ],
  "landing_url": "https://www.jstage.jst.go.jp/article/endocrj/advpub/0/advpub_EJ26-0240/_article",
  "language": null,
  "licence": "CC-BY-NC-ND",
  "licence_label": "CC BY-NC-ND",
  "licence_source": "journal",
  "licence_url": "https://creativecommons.org/licenses/by-nc-nd/4.0/",
  "metadata_licence": null,
  "native_subjects": [
    [
      "jstage-journal",
      "medicine/endocrinology-metabolism"
    ],
    [
      "openalex",
      "2713|Adipokines, Inflammation, and Metabolic Diseases"
    ]
  ],
  "openalex": "W7214398815",
  "pdf_url": "https://www.jstage.jst.go.jp/article/endocrj/advpub/0/advpub_EJ26-0240/_pdf",
  "pmcid": null,
  "pmid": null,
  "published": "2026-09-26",
  "publisher": null,
  "ris": "https://research.draconitic.com/paper/4ezfhena2cd54.ris",
  "sources": [
    {
      "first_seen": "2026-10-30T12:17:20Z",
      "native_id": "10.1507/endocrj.ej26-0240",
      "source": "jstage",
      "url": "https://www.jstage.jst.go.jp/article/endocrj/advpub/0/advpub_EJ26-0240/_article"
    }
  ],
  "subjects": [
    {
      "field": "medicine",
      "origin": "openalex:2713|Adipokines, Inflammation, and Metabolic Diseases",
      "subfield": "epidemiology-public-health"
    }
  ],
  "title": "Acquired generalized lipodystrophy complicated by fulminant type 1 diabetes mellitus: a case report with review of literature and immunological insights",
  "title_text": "Acquired generalized lipodystrophy complicated by fulminant type 1 diabetes mellitus: a case report with review of literature and immunological insights",
  "type": "peer-reviewed",
  "updated_at": "2026-10-30T12:17:20Z",
  "url": "https://research.draconitic.com/paper/4ezfhena2cd54",
  "venue": "Endocrine Journal",
  "versions": []
}