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Multiorgan Autoimmune Manifestations Associated with Thymoma

Multiorgan Autoimmune Manifestations Associated with Thymoma CASE REPORT Multiorgan Autoimmune Manifestations  Associated with Thymoma Anish Thomas, MD,* Arun Rajan, MD,* Arlene Berman, RN,* and Giuseppe Giaccone, MD, PhD† 38-year-old woman presented with progressive metastatic TABLE 1.  Results of Pertinent Diagnostic Tests at  A World Health Organization (WHO) subtype B1 thymoma Presentation after previous cisplatin-based chemotherapy, mediastinal radi- Variable Laboratory Value (Normal) ation, and pemetrexed. She enrolled on a phase II trial of cixu- tumumab, an insulin-like growth factor-1 receptor antibody White blood cell count 14.3 (3.9–10K/ul) (20 mg/kg, intravenously q 3 weeks). CD4+ T cells 3324 (359–1565/uL) After two doses, a 29% reduction in the sum of the tar- CD8+ T cells 3389 (178–853/uL) get lesions was observed. Three weeks after the third dose, she CD4−/CD8− T cells 375 (18–185/uL) presented with fatigue, dyspnea, dysphagia, and dysphonia. Mature naive (CD45RA+/CD3+) 2466 (82–755/uL) She had grade 4 proximal muscle weakness of extremities and CD4+ T cells neck with generalized hyporeflexia. Investigations (Table 1) Mature naive (CD45RA+/CD3+) 3093 (51–572/uL) CD8+ T cells revealed severe multi-organ dysfunction with polymyositis, CD4/CD8 ratio 0.98 fascitis, myocarditis, hepatitis, hypogammaglobulinemia, XXX and possible bulbar myasthenic involvement. A day later, she Eosinophils 15 (0.7–5.8%) developed worsening of the bulbar symptoms and respira- CPK 4927 (38–252 U/L) tory failure necessitating noninvasive ventilation. She http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Thoracic Oncology Wolters Kluwer Health

Multiorgan Autoimmune Manifestations Associated with Thymoma

Giaccone, Giuseppe
Journal of Thoracic Oncology , Volume 10 (2) – Feb 1, 2015
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Copyright
Copyright © 2014 by the International Association for the Study of Lung Cancer
ISSN
1556-0864
DOI
10.1097/JTO.0000000000000324
pmid
25611232
Publisher site
See Article on Publisher Site

Abstract

CASE REPORT Multiorgan Autoimmune Manifestations  Associated with Thymoma Anish Thomas, MD,* Arun Rajan, MD,* Arlene Berman, RN,* and Giuseppe Giaccone, MD, PhD† 38-year-old woman presented with progressive metastatic TABLE 1.  Results of Pertinent Diagnostic Tests at  A World Health Organization (WHO) subtype B1 thymoma Presentation after previous cisplatin-based chemotherapy, mediastinal radi- Variable Laboratory Value (Normal) ation, and pemetrexed. She enrolled on a phase II trial of cixu- tumumab, an insulin-like growth factor-1 receptor antibody White blood cell count 14.3 (3.9–10K/ul) (20 mg/kg, intravenously q 3 weeks). CD4+ T cells 3324 (359–1565/uL) After two doses, a 29% reduction in the sum of the tar- CD8+ T cells 3389 (178–853/uL) get lesions was observed. Three weeks after the third dose, she CD4−/CD8− T cells 375 (18–185/uL) presented with fatigue, dyspnea, dysphagia, and dysphonia. Mature naive (CD45RA+/CD3+) 2466 (82–755/uL) She had grade 4 proximal muscle weakness of extremities and CD4+ T cells neck with generalized hyporeflexia. Investigations (Table 1) Mature naive (CD45RA+/CD3+) 3093 (51–572/uL) CD8+ T cells revealed severe multi-organ dysfunction with polymyositis, CD4/CD8 ratio 0.98 fascitis, myocarditis, hepatitis, hypogammaglobulinemia, XXX and possible bulbar myasthenic involvement. A day later, she Eosinophils 15 (0.7–5.8%) developed worsening of the bulbar symptoms and respira- CPK 4927 (38–252 U/L) tory failure necessitating noninvasive ventilation. She

Journal

Journal of Thoracic Oncology – Wolters Kluwer Health

Published: Feb 1, 2015

References

  • Cixutumumab for patients with recurrent or refractory advanced thymic epithelial tumours: a multicentre, open-label, phase 2 trial
    Rajan
  • Thymoma and autoimmunity
    Shelly
  • Expression of autoimmune regulator gene (AIRE) and T regulatory cells in human thymomas
    Scarpino
  • Transcriptional regulation by AIRE: molecular mechanisms of central tolerance
    Peterson
  • Chronic mucocutaneous candidiasis in APECED or thymoma patients correlates with autoimmunity to Th17-associated cytokines
    Kisand