ABSTRACT
CASE: A 22-year-old woman with h/o asthma initially presented to the hospital with lip swelling and sore throat. She tested positive for COVID-19 and received a casirivimab-imdevimab (monoclonal antibody) infusion. She returned a week later with worsening lip swelling, dysphagia and conjunctivitis. Physical exam revealed edematous lips with vesicular lesions, no tongue swelling, tonsillar exudate, 4+ conjunctival injection bilaterally with purulent discharge, and shallow clean based clitoral ulceration. She reports no history of allergic reactions, angioedema or exposure to new medications. Nasopharyngolaryngoscopy showed no laryngeal edema but visualized exudates throughout the supraglottis and glottis. C4, ANA, CMV, EBV, throat and blood cultures were negative. STI testing was trichomonas positive and gonorrhea/chlamydia negative. Respiratory virus panel remained positive for COVID-19. HSV swab of lip lesion, HSV 1/2 IgG and IgM were negative. Mycoplasma pneumoniae IgG was elevated (0.60, negative is ≤0.09), IgM equivocal (0.85, negative is ≤0.76), and nasopharyngeal PCR negative. Conjunctival culture showed rare bacteria (S. Aureus) and no leukocytes. She initially received methylprednisolone IV due to concern for angioedema, acyclovir for empiric HSV treatment and empiric antibacterial moxifloxacin eye drops. Given lack of infectious trigger, her presentation was concerning for reactive infectious mucocutaneous eruption (RIME) associated with SARSCoV-2 or Mycoplasma. Prednisone 1mg/kg daily was initiated followed by improvement in oral mucositis and conjunctivitis within days. IMPACT/DISCUSSION: A broad differential is important when evaluating oral swelling and mucositis. Her lack of cutaneous involvement, medication exposure or family history and negative infectious, autoimmune and inflammatory workup make other causes including Stevens-Johnson syndrome, erythema multiforme, angioedema, and HSV less likely. Our final diagnosis of RIME describes mucocutaneous eruptions likely due to an immune response triggered by bacterial or viral infection. Our patient's RIME may be due to COVID-19 or Mycoplasma given her equivocal Mycoplasma IgM. Eruptions generally involve two or more mucosal sites and occur mostly in children and adolescents. Common presentations include oral erosions and ulcers, purulent bilateral conjunctivitis, or urogenital lesions, which were all seen in our patient. As this is a relatively rare and new condition, no standard of care treatment exists for RIME but systemic steroids have been effective in case reports for initial treatment and subsequent flares. CONCLUSION: RIME is a rare, newly described condition in young patients who develop postinfectious mucocutaneous eruptions of two or more mucosal sites. It has been recently reported in association with COVID-19 and its association with Mycoplasma infection is important to evaluate. This condition is important to recognize and treat given the requirement for higher dose steroids than that used for angioedema.
ABSTRACT
CASE: A 64 year old female with a history of rheumatoid arthritis (RA) and hypertension presented to the hospital with shortness of breath. Her RA had been well controlled with prednisone 5mg daily and rituximab infusions every 6 months, last given three months prior. SARS-CoV-2 PCR was positive and she was treated with dexamethasone. Two months later, she presented to the emergency department with recurrent shortness of breath. At this time, her cycle threshold was 29.82 and CT chest demonstrated bilateral air bronchograms. No improvement was noted with antibiotics, BAL showed 500 WBCs, 70% lymphocytes and biopsy of the left lung showed chronic interstitial inflammation, consistent with patchy organizing pneumonia. She improved with a course of prednisone 30mg for five weeks. Four months later, she was admitted for worsening shortness of breath. At this time, she was noted to be SARS-CoV-2 PCR negative and bronchoscopy showed no growth on cultures. One month later (7 months from initial hospitalization) she was readmitted with hypoxia. Cycle time was 19 indicating high viral load, SARS-CoV-2 PCR positive, nucleocapsid antibody positive and spike IgG antibody negative thus suggesting reinfection with lack of immune response to vaccination. She was treated with remdesivir with minimal improvement. Due to concern for reinfection, she was treated with baricitinib and casirivimab-imdevimab monoclonal antibody treatment with improvement. CD19 count was <10 with <0.5%, consistent with B cell depletion. IMPACT/DISCUSSION: Rituximab, an anti-CD20 monoclonal antibody that results in B-cell depletion, is commonly used to treat rheumatoid arthritis. While it is essential for disease control, it also risks compromising antiviral immunity, re-infection to SARS-CoV-2, and impaired vaccine efficacy (1). Our case describes a patient with rheumatoid arthritis treated with rituximab infusion, and her subsequent prolonged COVID-19 re-infection with multiple hospitalizations. B-Cell depletion from rituximab infusion leading to a prolonged COVID-19 course is a rare and relatively new phenomenon that is being reported over the course of the pandemic. Given her intermittent negative testing, high viral load, and lack of response to prior vaccination, it is likely that her presentation represented true re-infection in the setting of insufficient antibody production due to rituximab, further supported by improvement with monoclonal antibody treatment. As demonstrated by this patient, monoclonal antibodies were particularly effective in clinical improvement given her own lack of antibody response. CONCLUSION: While rituximab may be safe for many patients, it may result in significant, prolonged B-cell depletion. This depletion may increase the risk of reinfection, affect vaccine response and antibody formation. Administration of monoclonal antibody treatment should be considered in immunosuppressed and B-cell depleted patients with COVID-19 infection.