Mesothelioma in Familial Mediterranean Fever With Colchicine Intolerance: A Case Report and Literature Review
| Autor: | Manuel Soldato, Elena Verrecchia, Rosa Talerico, Raffaele Manna, Francesca Schinzari, Carmine Cardillo, Maria Cristina Giustiniani, Francesco De Vito |
|---|---|
| Jazyk: | angličtina |
| Rok vydání: | 2020 |
| Předmět: |
Male
Mesothelioma 0301 basic medicine lcsh:Immunologic diseases. Allergy medicine.medical_specialty Abdominal pain Immunology Familial Mediterranean fever Peritonitis Case Report Gastroenterology colchicine canakinumab 03 medical and health sciences 0302 clinical medicine Internal medicine Humans interleukin (IL)-1b inhibition Medicine Immunology and Allergy Peritoneal Fibrosis Aged Familial Mediterranean fever (FMF) Inflammation Anakinra Polymorphism Genetic Leukopenia business.industry Settore MED/09 - MEDICINA INTERNA Homozygote Pyrin malignant mesothelioma (MST) medicine.disease Familial Mediterranean Fever Interleukin 1 Receptor Antagonist Protein 030104 developmental biology Familial Mediterranean fever (FMF) malignant mesothelioma (MST) colchicine peritoneal recurrent inflammation canakinumab anakinra interleukin (IL)-1b inhibition Peritoneal mesothelioma Peritoneum medicine.symptom Tomography X-Ray Computed business lcsh:RC581-607 peritoneal recurrent inflammation 030215 immunology medicine.drug anakinra |
| Zdroj: | Frontiers in Immunology, Vol 11 (2020) Frontiers in Immunology |
| ISSN: | 1664-3224 |
| DOI: | 10.3389/fimmu.2020.00889/full |
| Popis: | A 65-year-old Italian physician affected by Familial Mediterranean fever (FMF) was hospitalized due to progressive abdominal enlargement, which had begun 6 months before admission. Physical examination revealed ascites and bilateral leg edema. Abdominal CT scan showed ascitic fluid and extensive multiple peritoneal implants; peritoneal CT-guided biopsy revealed an epithelial-type malignant mesothelioma. The patient's past medical history revealed recurrent episodes of abdominal pain and fever from the age of 2. Clinical diagnosis of FMF was suspected at the age of 25, while genetic analysis, performed at the age of 50, confirmed homozygosity for the M694I mutation in the MEFV gene. Treatment with the first line FMF drug colchicine was started and stopped several times because of worsened leukopenia. The patient in fact had a history of asymptomatic leukopenia/lymphopenia from an early age; the intake of colchicine aggravated his pre-existing problem until the definitive suspension of the drug. As for second-line drugs, canakinumab was first prescribed, but due to prescription issues, it was not possible to be administered. When he was given anakinra, there was a worsening of leukopenia leading to septic fever. Systematic literature review indicates that, in most cases, recurrent peritoneal inflammation results in benign peritoneal fibrosis or less commonly in encapsulating peritonitis. There are only a few reported cases of recurrent peritoneal inflammation progressing from FMF to peritoneal mesothelioma (MST). In such cases, intolerance to colchicine or its erratic intake may lead to long-term recurrent inflammation, which usually precedes the development of the tumor, while pre-existing leukopenia, as in our patient, could also be a factor promoting or accelerating the tumor progression. In conclusion, we suggest that in the presence of intolerance or resistance to colchicine, interleukin (IL)-1 inhibition could suppress peritoneal inflammation and prevent MSTs. |
| Databáze: | OpenAIRE |
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