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Lack of efficacy of Cyclosporine A in
a patient with episodic angioedema
and eosinophilia
(Gleich’s syndrome)
Immunoallerg...
A 29yo man refers to our Unit for persistent
eosinophilia, up to >18k/mm3
No target organ
involvement
(skin, airways, card...
Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E
Clinica...
After a few days in hospital…
Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F...
Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E
Diagnos...
Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E
Therape...
Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E
Therape...
Thank you!
fil.fassio@tin.itImmunoallergology Unit, Dept. of Biomedicine
University of Florence
June 9th, 2009
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Fassio warsaw

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Fassio warsaw

  1. 1. Lack of efficacy of Cyclosporine A in a patient with episodic angioedema and eosinophilia (Gleich’s syndrome) Immunoallergology Unit, Dept. of Biomedicine University of Florence June 9th, 2009 Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E
  2. 2. A 29yo man refers to our Unit for persistent eosinophilia, up to >18k/mm3 No target organ involvement (skin, airways, cardiovascular, G-I, neurologic) Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E Clinical presentation: Eosinophilia No genetic abnormalities (FIP1L1, Bcr/Abl, JAK) 0 500 1000 1500 2000 2500 3000 1 2 3 4 5 6 7 8 9 10 cells/mm3 Days Eosinophils
  3. 3. Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E Clinical presentation: Angioedema Recurrent Angioedema, in the last 6 years No evident trigger: food intake, drugs, insect stings… No C1-INH deficiency, either quantitative or functional Diagnostic workup: negative
  4. 4. After a few days in hospital… Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E Clinical evolution: EAE Episodic Angioedema with Eosinophilia: Gleich’s Syndrome CCS AE
  5. 5. Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E Diagnosis: Gleich’s Syndrome Recurrent Angioedema + Eosinophilia Described by Gleich in 1984 Unclear etiology Very good prognosis (no target organ involvement) Angioedema attacks can be treated with corticosteroids… …but no prophylactic therapy!
  6. 6. Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E Therapeutic Approach: Gleich’s Syndrome Demonstration of an aberrant T Lymphocyte population (CD3-CD4+CD5+) Course of CyA as a steroid-sparing agent… Failure!
  7. 7. Fassio F, Matucci A, Vultaggio A, Rossi O, Almerigogna F, Agostini C, Liotta F, Annunziato F, Romagnani S, Maggi E Therapeutic Approach: Gleich’s Syndrome Procedure for compassionate use of Mepolizumab currently ongoing… Increased production of Th2 cytokines, ↑↑↑ IL-5
  8. 8. Thank you! fil.fassio@tin.itImmunoallergology Unit, Dept. of Biomedicine University of Florence June 9th, 2009

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