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
Introduction: A 67 year old man presented dysphagia after a right partial epiglottectomy for a cT1N2bM0 supraglottic squamous cell carcinoma. Immunohistological assessment showed a mixed cellularity: a neuroendocrine (CK-20 +, S100 +, Ki67 90%) and squamous cell carcinoma. Margin ampliation to the right glossoepiglottic fold and a bilateral cervical lymphadenectomy was performed. Tracheostomy was needed after surgery because of laryngeal edema. Materials and Methods: Nasolaryngoscopy showed no glottic closure, saliva retention with aspiration and an absent answer to stimuli in the right supraglottic space. After a 5 ml pudding texture was given, methylene blue remained in the right pyriform sinus, aspiration and an absence of cough reflex. No oxygen desaturation or oral residuewas evidenced during exploration. Fractioned swallowing was needed. Results: A multidisciplinary evaluation of the patient in our Dysphagia Department took place. Logopedic restitutive techniques were a pillar in the rehabilitation of the patient. Sensory stimulation with heat foam, increase in muscular tone with direct stimulation to the tongue/pharynx and compensatory swallowing techniques were taught to the patient. After a 6 month follow up the same test with 5 ml pudding was performed. Glottic closure returned, with no evidence of aspiration to 5 ml pudding texture. Aspiration to nectar texture, fractioned swallowing and a right absence to stimuli remained. Conclusions: Dysphagia after head and neck cancer surgery is a common occurrence, given the most common location of appearance for squamous cell carcinoma is the supraglottis. Multidisciplinary approach to the patient that develops dysphagia remains key in the improvement of their quality of life and reduces the number of comorbidities after treatment. In this case the patient needed a tracheostomy after surgery, increasing their chances of dysphagia. After a closely followed multidisciplinary rehabilitation the patient was able to improve glottic closure and never had an episode of aspiration pneumonia until their death this past March 2021 from COVID-19.