Michal Landes1, Yasmin Maor2, Diego Mercer3, Zohar Habot-Wilner4, Efraim Bilavsky5, Bibiana Chazan6, Regev Cohen7, Daniel Glikman8, Jacob Strahilevitz9, Michal Katzir10, Vladislav Litachevsky11, Rimma Melamed12, Alex Guri13, Hila Shaked14, Odelya Perets15, Yonit Wiener-Well16, Anat Stren17, Michal Paul17, Oren Zimhony18, Isaac Srugo19, Galia Rahav20, Jihad Bishara14, Amir A Kuperman21, Ronen Ben-Ami1, Moshe Ephros22, Michael Giladi1. 1. Infectious Disease Unit, Tel Aviv Sourasky Medical Center and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 2. Infectious Disease Unit, Wolfson Medical Center, Holon, Israel, and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 3. Department of Radiology, Tel Aviv Sourasky Medical Center and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 4. Division of Ophthalmology, Tel Aviv Sourasky Medical Center and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 5. Schneider Children's Medical Center, Petah Tiqva, Israel, and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 6. Infectious Diseases Unit, Emek Medical Center, Afula, Israel, and Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel. 7. Infectious Diseases Unit, Sanz Medical Center, Laniado Hospital, Netanya, Israel, and Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel. 8. Azrieli Faculty of Medicine in the Galilee, Bar-Ilan University, Safed, Israel. 9. Department of Clinical Microbiology and Infectious Diseases, Hadassah-Hebrew University, Jerusalem, Israel. 10. Infectious Disease Unit, Meir Medical Center, Kfar Saba, Israel. 11. Infectious Disease Unit, Sheba Medical Center, Tel-Hashomer, Ramat Gan, Israel. 12. Pediatric Infectious Disease Unit, Soroka University Medical Center, Beer-Sheva, Israel. 13. Department of Pediatrics, Kaplan Medical Center, Rehovot, Israel, and School of Medicine, Hebrew University and Hadassah Medical Center, Jerusalem, Israel. 14. Infectious Diseases Unit, Rabin Medical Center, Beilinson Hospital, Petah-Tikva, Israel, and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 15. Infectious Disease Unit, Soroka University Medical Center, Beer Sheva, Israel. 16. Infectious Disease Unit, Shaare Zedek Medical Center, Hebrew University, Jerusalem, Israel. 17. Infectious Diseases Institute, Rambam Health Care Campus, Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel. 18. Infectious Diseases Unit, Kaplan Medical Center, Rehovot, Israel, and School of Medicine, Hebrew University and Hadassah, Jerusalem, Israel. 19. Department of Pediatrics, Bnai Zion Medical Center, Haifa, Israel, and Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel. 20. Infectious Disease Unit, Sheba Medical Center, Tel-Hashomer, Ramat Gan, Israel, and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel. 21. Blood Coagulation Service and Pediatric Hematology Clinic, Galilee Medical Center, Nahariya, Israel, and Azrieli Faculty of Medicine, Bar-Ilan University, Safed, Israel. 22. Pediatric Infectious Disease Unit, Carmel Medical Center and Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel.
Abstract
BACKGROUND: Fever of unknown origin (FUO) is a rare manifestation of cat scratch disease (CSD). Data regarding CSD-associated FUO (CSD-FUO), particularly in adults, are limited. We aimed to study disease manifestations and long-term clinical outcome. METHODS: A national CSD surveillance study has been conducted in Israel since 1991. Data are obtained using questionnaires, review of medical records, and telephone interviews. FUO was defined as fever of ≥14 days without an identifiable cause. CSD-FUO patients were identified in the 2004-2017 CSD national registry. Follow-up included outpatient clinic visits and telephone/e-mail surveys. RESULTS: The study included 66 CSD-FUO patients. Median age was 35.5 years (range, 3-88). Median fever duration was 4 weeks (range, 2-9). Relapsing fever pattern was reported in 52% of patients, weight loss in 57%, and night sweats in 48%. Involvement of ≥1 organs occurred in 59% of patients; hepatosplenic space-occupying lesions (35%), abdominal/mediastinal lymphadenopathy (20%), ocular disease (18%), and multifocal osteomyelitis (6%) were the most common. Malignancy, particularly lymphoma, was the initial radiological interpretation in 21% of patients; 32% underwent invasive diagnostic procedures. Of the 59 patients available for follow-up (median duration, 31 weeks; range, 4-445), 95% had complete recovery; 3 patients remained with ocular sequelae. CONCLUSION: This is the first attempt to characterize CSD-FUO as a unique syndrome that may be severe and debilitating and often mimics malignancy. Relapsing fever is a common clinical phenotype. Multiorgan involvement is common. Recovery was complete in all patients except in those with ocular disease.
BACKGROUND:Fever of unknown origin (FUO) is a rare manifestation of cat scratch disease (CSD). Data regarding CSD-associated FUO (CSD-FUO), particularly in adults, are limited. We aimed to study disease manifestations and long-term clinical outcome. METHODS: A national CSD surveillance study has been conducted in Israel since 1991. Data are obtained using questionnaires, review of medical records, and telephone interviews. FUO was defined as fever of ≥14 days without an identifiable cause. CSD-FUO patients were identified in the 2004-2017 CSD national registry. Follow-up included outpatient clinic visits and telephone/e-mail surveys. RESULTS: The study included 66 CSD-FUO patients. Median age was 35.5 years (range, 3-88). Median fever duration was 4 weeks (range, 2-9). Relapsing fever pattern was reported in 52% of patients, weight loss in 57%, and night sweats in 48%. Involvement of ≥1 organs occurred in 59% of patients; hepatosplenic space-occupying lesions (35%), abdominal/mediastinal lymphadenopathy (20%), ocular disease (18%), and multifocal osteomyelitis (6%) were the most common. Malignancy, particularly lymphoma, was the initial radiological interpretation in 21% of patients; 32% underwent invasive diagnostic procedures. Of the 59 patients available for follow-up (median duration, 31 weeks; range, 4-445), 95% had complete recovery; 3 patients remained with ocular sequelae. CONCLUSION: This is the first attempt to characterize CSD-FUO as a unique syndrome that may be severe and debilitating and often mimics malignancy. Relapsing fever is a common clinical phenotype. Multiorgan involvement is common. Recovery was complete in all patients except in those with ocular disease.
Authors: Helga Kaiser Sanches de Maria; Emily Gazzoli; Marina Rovani Drummond; Amanda Roberta de Almeida; Luciene Silva Dos Santos; Ricardo Mendes Pereira; Antonia Terezinha Tresoldi; Paulo Eduardo Neves Ferreira Velho Journal: Rev Inst Med Trop Sao Paulo Date: 2022-02-16 Impact factor: 1.846