Infections in the immunocompromised: lessons from a fatal case report of tricuspid endocarditis in multiple sclerosis

Abstract

Background: Multiple sclerosis (MS), a chronic neurological disorder, is characterized by damage to the central nervous system, autoimmune processes, and deterioration of the myelin sheath, which lead to a variety of physical, cognitive, and emotional symptoms. Currently, a cure for MS does not exist, and treatments aim only to manage symptoms and enhance quality of life. Betaferon (interferon beta-1b), a disease-modifying therapy, is used to minimize the frequency of relapses and the progression of disability by altering immune responses. However, this therapeutic strategy also increases the risk of infectious diseases, such as tricuspid endocarditis, which is a severe infection of the heart valve that is often associated with chronic intravenous drug use and carries potential life-threatening consequences.

Case presentation: We present a case of a 48-year-old male who was undergoing Betaferon therapy for MS. He was admitted to the emergency room owing to fever, shortness of breath, and altered mental state. Physical examination revealed clinical signs of endocarditis, and laboratory results indicated severe thrombocytopenia, anemia, and elevated inflammatory markers. Imaging confirmed lung abnormalities and vegetations on the tricuspid valve. Blood cultures identified methicillin-sensitive Staphylococcus aureus. Intravenous antibiotics were initiated, and surgical intervention was planned. Despite initial antibiotic therapy improving his clinical and laboratory conditions, the patient experienced multiple pulmonary embolic events that led to death after 16 days of hospitalization.

Conclusion: This case underscores the challenges of identifying and managing endocarditis in immunocompromised patients and highlights the complexities of treating infections in individuals with chronic diseases.

Keywords

Betaferon (interferon beta-1b) / Case report / Immunocompromised patients / Multiple sclerosis / Tricuspid endocarditis

Conflict of interest statement

The authors declare no conflict of interest.

Author contributions

Cacciapuoti Fulvio and Cacciapuoti Federico conceived of and drafted the manuscript, Cacciapuoti Fulvio and Gottilla R performed the echocardiographic evaluations, Maniscalco G performed the neurological evaluations, Dell’Aversano Orabona G performed the radiological evaluations, Cacciapuoti Fulvio, Costabile F, and Cacciapuoti Federico verified the analytical methods. All authors have discussed the results, read and approved the final version of the manuscript, and agreed to be held accountable for all aspects of the work.

Funding

None.

Ethical approval of studies and informed consent

The study followed the principles of the Declaration of Helsinki as revised in 2013. The publication of individual case reports is exempt from ethical approval according to the Antonio Cardarelli Hospital guidelines. Written informed consent was obtained from the patient.

Acknowledgments

None.

References

[1] Dobson R, Giovannoni G.Multiple sclerosis—a review. Eur J Neurol. 2019, 26(1):27-40. doi:10.1111/ene.13819

[2] Yong HYF, Yong VW. Mechanism-based criteria to improve therapeutic outcomes in progressive multiple sclerosis. Nat Rev Neurol. 2022, 18(1):40-55. doi:10.1038/s41582-021-00581-x

[3] Etemadifar M, Janghorbani M, Shaygannejad V. Comparison of Betaferon, Avonex, and Rebif in treatment of relapsing-remitting multiple sclerosis. Acta Neurol Scand. 2006, 113(5):283-287. doi:10.1111/j.1600-0404.2006.00585.x

[4] Suljic E, Sinanovic O, Kurtovic-Alajbegovic A, Klupka-Saric I. Betaferon efficiency indicators in the treatment of relapsing remitting form of multiple sclerosis. Med Arch. 2013, 67(3):178-180. doi:10.5455/medarh.2013.67.178-180

[5] Hartung HP, Graf J, Kremer D. Long-term follow-up of multiple sclerosis studies and outcomes from early treatment of clinically isolated syndrome in the BENEFIT 11 study. J Neurol. 2020, 267(2):308-316. doi:10.1007/s00415-018-09169-w

[6] Barbieri MA, Sorbara EE, Battaglia A, et al. Adverse drug reactions with drugs used in multiple sclerosis: an analysis from the Italian pharmacovigilance database. Front Pharmacol. 2022, 13:808370. doi:10.3389/fphar.2022.808370

[7] Manova MG, Kostadinova II. Adverse drug reactions after 24-month treatment with two-dosage regimens of Betaferon in patients with multiple sclerosis. Folia Med (Plovdiv) 2009, 51(3):31-36.

[8] Shmueli H, Thomas F, Flint N, Setia G, Janjic A, Siegel RJ. Right-sided infective endocarditis 2020: challenges and updates in diagnosis and treatment. J Am Heart Assoc. 2020, 9(15):e017293. doi:10.1161/JAHA.120.017293

[9] Raut N, Potdar A, Sharma S. Tricuspid valve endocarditis in non-drug abusers: a case series from India. Indian Heart J. 2018, 70(4):476-481. doi:10.1016/j.ihj.2017.09.011

[10] Nandakumar R, Raju G. Isolated tricuspid valve endocarditis in nonaddicted patients: a diagnostic challenge. Am J Med Sci. 1997, 314(3):207-212. doi:10.1097/00000441-199709000-00011

[11] Travers BS, Tsang BK, Barton JL. Multiple sclerosis: diagnosis, disease-modifying therapy and prognosis. Aust J Gen Pract. 2022, 51(4):199-206. doi:10.31128/AJGP-07-21-6103

[12] Sauerborn M, van Beers MM, Jiskoot W, et al. Antibody response against Betaferon® in immune tolerant mice: involvement of marginal zone B-cells and CD4+ T-cells and apparent lack of immunological memory. J Clin Immunol. 2013, 33(1):255-263. doi:10.1007/s10875-012-9783-z

[13] Li H, Hu F, Zhang Y, Li K. Comparative efficacy and acceptability of disease-modifying therapies in patients with relapsing-remitting multiple sclerosis: a systematic review and network meta-analysis. J Neurol. 2020, 267(12):3489-3498. doi:10.1007/s00415-019-09395-w

[14] Revilla A, López J, Villacorta E, et al. Isolated right-sided valvular endocarditis in non-intravenous drug users. Rev Esp Cardiol. 2008, 61(12):1253-1259. doi:10.1016/s1885-5857(09)60052-9

[15] Dhib-Jalbut S, Marks S. Interferon-beta mechanisms of action in multiple sclerosis. Neurology. 2010, 74(Suppl 1):S17-S24. doi:10.1212/WNL.0b013e3181c97d99

[16] Willekens R, Puig-Asensio M, Suanzes P, et al. Empirical use of β-lactam/β-lactamase inhibitor combinations does not increase mortality compared with cloxacillin and cefazolin in methicillin-susceptible staphylococcus aureus bacteraemia: a propensity-weighted cohort study. J Antimicrob Chemother. 2022, 77(8):2288-2295. doi:10.1093/jac/dkac152

[17] Cuervo G, Escrihuela-Vidal F, Gudiol C, Carratalà J. Current challenges in the management of infective endocarditis. Front Med (Lausanne). 2021, 8:641243. doi:10.3389/fmed.2021.641243

[18] Dobreva-Yatseva B, Nikolov F, Raycheva R, Tokmakova M. Infective endocarditis-characteristics and prognosis according to the affected valves. Microorganisms. 2024, 12(5):987. doi:10.3390/microorganisms12050987