Case Presentation: An 18-year-old African American male presented to the hospital with complaints of fever, vomiting, weakness in his upper and lower extremities, unsteady gait and joint pain. A few days prior to the onset of symptoms he had a diarrheal illness. He denies any recent history of travel, sick contacts, animal bites, scratches or insect bites. Physical examination revealed scattered non-confluent macules less than 1cm on upper chest and arms bilaterally. Bilateral tenderness and swelling was present in shoulder, knee and ankle joints. Motor strength was noted to be 2/5 in the upper and lower extremities. Investigations revealed pancytopenia, creatinine kinase of 22,000 U/L, transaminitis and impaired renal function secondary to rhabdomyolysis. Myositis specific antibodies were negative along with connective tissue disease panel. Bacteriological studies yielded negative results and viral studies showed, apart from a past parvovirus B19 infection, positive Coxsackie B4 titers at 1:16 and B6 titers at 1:8. Initially there was suspicion of hemophagocytic syndrome and possibly dermatomyositis; however, patient’s rash was not typical and scant factors in support. An echocardiogram demonstrated a low normal ejection fraction of 45-50% with mild global hypokinesis. He was given aggressive intravenous hydration which resulted in shortness of breath; with diuresis his respiratory symptoms improved. Intravenous infusion of methylprednisolone 125mg every eight hours was begun and within a 24 hour period his muscle enzymes trended downwards and his inflammatory myopathy and arthropathy gradually improved. He was discharged 3 days later in stable condition on oral prednisone 80mg daily. He was seen one week later as an outpatient with complete resolution of symptoms and prednisone was tapered. Four weeks later Coxsackie B titers were rechecked and found to be negative at B4 < 1:8 and B6 <1:8.
Discussion: Coxsackie B infections have been known to present with varied clinical manifestations including myopericarditis, meningoencephalitis and polymyositis, however arthropathy has not been widely recognized with only 11 cases reported. Initially when considering a rash in association with proximal muscle weakness and arthritis our first thought was polymyositis or dermatomyositis, however the patient did not have the typical rash features and the myositis antibody panel was negative. Adult onset stills disease and macrophage activation syndrome or hemophagocytic syndrome were in the differential due to the presence of cytopenias, however our patient did not have the characteristic fever pattern or a highly elevated triglyceride level. Infectious etiologies were extensively saught after including viral hepatitis and parvoviruses. Lastly, the hint of myocarditis lead us towards the consideration of the coxsackie infection. Coxsackie B viruses are both highly resistant to chemical treatment and highly contagious primarily through the fecal-oral route, so a high index of suspicion for unusual presentations of this infection may aid in the timely diagnosis and treatment of this potentially fatal disease.
Conclusions: This case represents features strikingly outside the realm of a usual Coxsackie B infection and therefore of great significance to a wide range of physicians to consider when encountering patients with similar presentations.
To cite this abstract:Haider BA, Ajmal M. An Outlier of Inflammatory Arthropathy in a Coxsackie B Infection. Abstract published at Hospital Medicine 2016, March 6-9, San Diego, Calif. Abstract 542. Journal of Hospital Medicine. 2016; 11 (suppl 1). https://www.shmabstracts.com/abstract/an-outlier-of-inflammatory-arthropathy-in-a-coxsackie-b-infection/. Accessed September 22, 2019.