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Paediatric patients comprise a small proportion of the SARS-CoV-2 infected population. They usually present with mild symptoms, however a small proportion of them may require intensive care due to shock and multi-organ failure related to Paediatric Inflammatory Multisystem Syndrome temporally associated with SARS-CoV-2 (PIMS-TS). This review article summarises the oral mucosal lesions in children with COVID-19 and PIMS-TS. The most common sites affected are the tongue and lips. Commonly reported lesions include cheilitis, dry and red lips, and tongue swelling. This article is of importance to all healthcare professionals involved in the multidisciplinary care for this group of patients.Copyright © 2021 The Authors
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SESSION TITLE: Poster Cases in Asthma and Allergy SESSION TYPE: Case Report Posters PRESENTED ON: 10/17/2022 12:15 pm - 01:15 pm INTRODUCTION: Anaphylaxis is an acute, potentially life-threatening, systemic allergic reaction that presents as a multitude of manifestations after exposure to an allergen. It is rare to have an anaphylactic reaction to the SARS-Cov-2 vaccine, however, we present a patient who had a prolonged anaphylactic reaction to the SARS-CoV-2 virus itself. CASE PRESENTATION: A 23-year-old female with past medical history of chronic idiopathic urticaria and dermatographism presented with fatigue, rash, rhinitis, and lip swelling in the setting of being COVID-19 positive. She had received two doses of the Pfizer-BioNTech COVID-19 vaccine nine months prior without any adverse affects. During the SARS-CoV-2 Omicron variant surge, the patient was exposed to a family member who had contracted COVID-19 infection. Two days prior to presentation, she started having a diffuse rash throughout her body. In the Emergency Department, she was given IM epinephrine x2, IV Solu-Medrol and IV Benadryl with temporary improvement in her symptoms. She developed hypotension that required initiation of a continuous epinephrine infusion and was admitted to the Medical Intensive Care Unit. Each time the epinephrine was held, generalized urticaria recurred and she would become hypotensive with her systolic blood pressure decreasing below 80 mmHg. The urticaria completely resolved and her blood pressure improved when the epinephrine infusion was restarted. The patient was treated with remdesivir, Solu-Medrol, loratadine, and famotidine. She was able to be weaned off the epinephrine infusion over the course of four days. Immunological workup was unremarkable apart from elevation of IL-10 on her cytokine panel. DISCUSSION: The symptoms and syndromes associated with COVID-19 infection remain diverse and while urticaria has been a documented manifestation of the virus, it is uncommon (1). There is a single case report of an anaphylactic reaction in response to COVID-19 (2). Given our patient's symptoms were temporally related to the COVID-19 infection, we believe the SARS-COV-2 virus was the trigger. She did not have any other exposure during this time. This case is unique in that the virus not only triggered the urticaria, but that her symptoms persisted for four days. After receiving antiviral therapy plus steroids and antihistamines, her symptoms finally resolved. The SARS-COV-2 virus itself is primarily attacked by immune cells, including mast cells, which release an array of proinflammatory cytokines (3). We postulate that in our patient, her history of chronic urticaria predisposed her to an exaggerated inflammatory response. CONCLUSIONS: SARS-CoV-2 has clinically presented itself in a multitude of ways across many disciplines. In a patient with a history of urticaria, prolonged anaphylactic symptoms can be triggered by the virus itself. Reference #1: Adeliño R., Andrés-Cordón J.F., De la Cruz Martínez C.A. Acute urticaria with angioedema in the setting of coronavirus disease 2019. J Allergy Clin Immunol Pract. 2020;8(7):2386–2387. Reference #2: Alvarez-Perea A, Baeza ML. Anaphylactic shock following the diagnosis of coronavirus disease 2019. Ann Allergy Asthma Immunol. 2020;125(4):477-478. Reference #3: Kritas SK, Ronconi G, Caraffa A, Gallenga CE, Ross R, Conti P. Mast cells contribute to coronavirus-induced inflammation: new anti-inflammatory strategy. J Biol Regul Homeost Agents. 2020 January-February,;34(1):9-14. DISCLOSURES: No relevant relationships by Brianne Navetta-Modrov No relevant relationships by Azzam Paroya Speaker/Speaker's Bureau relationship with Bayer Please note: current Added 03/30/2022 by Paul Strachan, value=Honoraria Speaker/Speaker's Bureau relationship with United Therapeutics Please note: more than 5 years Added 03/30/2022 by Paul Strachan, value=Honoraria Speaker/Speaker's Bureau relationship with Gilead Please note: $1001 - $5000 by Paul Strachan, value=Honoraria Removed 03/30/2022 by Pau Strachan Speaker/Speaker's Bureau relationship with Genentech Please note: $5001 - $20000 by Paul Strachan, value=Honoraria Removed 03/30/2022 by Paul Strachan Speaker/Speaker's Bureau relationship with Boehringer Ingelhein Please note: More than 5 years Added 03/30/2022 by Paul Strachan, value=Honoraria Stock Ownership relationship with Pfizer Please note: Purchased in 2000-2002 Added 03/30/2022 by Paul Strachan, value=nothing, I purchased stock Speaker/Speaker's Bureau relationship with Portola Please note: $1001 - $5000 by Paul Strachan, value=Honoraria Removed 03/30/2022 by Paul Strachan Stock Ownership relationship with Portola Please note: $5001 - $20000 by Paul Strachan, value=nothing, I purchased stock Removed 03/30/2022 by Paul Strachan Stock Ownership relationship with La Jolla Pharmaceuticals Please note: Purchased a few years back Added 03/30/2022 by Paul Strachan, value=nothing, I purchased stock Stock Ownership relationship with Seatle Genetics Please note: Purchased more than 5 years ago Added 03/30/2022 by Paul Strachan, value=nothing, I purchased stock PI relationship with United Therapeutics Please note: Current Added 03/30/2022 by Paul Strachan, value=Grant/Research Support PI relationship with Actelion/Janssen Please note: Current Added 03/30/2022 by Paul Strachan, value=Grant/Research Support PI for clinicial trial relationship with Roche Please note: Current Added 03/30/2022 by Paul Strachan, value=Grant/Research PI for clinicial trial relationship with Bellerophon Please note: Current Added 03/30/2022 by Paul Strachan, value=Grant/Research Support Speaker/Speaker's Bureau relationship with Actelion/Janssen Please note: Current Added 03/30/2022 by Paul Strachan, value=Honoraria
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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.
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CASE: Our patient is a 52-year-old female with a history of gastroesophageal reflux and hypertension. 36 hours after receiving the second Pfizer COVID-19 vaccine, she developed lip and tongue swelling, mucosal ulcerations, and respiratory distress. There was no conjunctivitis or genital involvement. She denied taking any new medications, supplements, or food that might have led to the reactions. She initially presented to an outside hospital and required intubation prior to transfer to our facility. A bedside esophagogastroduodenoscopy (EGD) was performed noting extensive Grade D erosive esophagitis and gastric ulcerations with friability. When the endoscope was removed a 34cm section of necrotic esophageal tissue was removed from the airway. Despite intravenous steroid treatment, she continued to have esophageal scarring and was unable to tolerate food by mouth. Therefore, a gastrostomy tube was placed. Since that time, she has required several recurrent EGDs for esophageal dilation due to scarring. It has now been six months from her initial injury, and unfortunately, the patient is still unable to take PO and is dependent on tube feedings. IMPACT/DISCUSSION: The coronavirus pandemic began in December 2019. At the time of this report, SARS-CoV-2 infection has been the cause of 5.48 million deaths worldwide and 836,000 deaths in the United States alone. In addition, this global pandemic has had severe economic and social implications. There are currently three vaccines authorized by the United States Food and Drug Administration for emergency use. I report an extremely uncommon complication of the Pfizer COVID-19 vaccine: a case of Eryethema Multiforme Major that occurred after the second dose vaccine without exposure to any other drug. Eryethema Multiforme is divided into major and minor forms and is regarded as distinct from Stevens- Johnson syndrome and toxic epidermal necrolysis. It is related to infections, usually Herpes Simplex Virus, or less commonly, to medications. In Erythema Multiforme, mucous membrane involvement is absent or mild. Erythema Multiforme Major is an immune mediated skin reaction involving the oral cavity and mucosa that is serious and occasionally life threatening. There have been several reported cases of Erythema Multiforme following COVID-19 vaccination but only one other cases of Erythema Multiforme Major associated with the mRnA-1273 SARS-CoV-2 vaccine (Moderna.) CONCLUSION: This case highlights an extremely rare vaccine consequence. The benefits still greatly outweigh the risks of vaccination, and this case does not diminish the importance of COVID-19 vaccination to effectively control this pandemic.
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Introduction: Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is well described as an etiology to severe acute respiratory distress syndrome (ARDS). However, rare immunologic and allergic manifestations may also occur from this infection. We report a novel case of angioedema occurring in the setting of COVID-19 infection in a fully vaccinated patient. Case Report: A 61-yearold COVID-19 vaccinated female with hypertension presented to the emergency department with tongue and lip swelling, odynophagia, dysphonia, and difficulty breathing. She denied personal or family history of allergies, anaphylaxis, or angioedema. Her home medications included Aspirin, methadone, Seroquel, and Klonopin, with no recent changes reported. Physical exam was notable for significant lip and tongue edema, audible dysphonia, and bilateral end-inspiratory wheezing. She was hypoxemic and placed on nasal cannula. Laboratory findings revealed lymphopenia, elevated inflammatory proteins, including C-reactive protein (57), Lactate dehydrogenase (LDH) (238), and D-dimer (11.52). Functional C1 esterase inhibitor levels (>91) were normal. Nasal PCR swab returned positive for SARS-CoV-2. Ear, nose, and throat specialist was consulted given concern for angioedema, and flexible nasolaryngoscopy was performed revealing uvular, epiglottic, and bilateral arytenoid edema concerning for impending airway compromise. The patient was initiated on intravenous methylprednisolone, epinephrine, antihistamines, tranexamic acid and admitted to the medical intensive care unit (ICU). She was monitored closely in the ICU with subsequent improvement of the angioedema and resolution of the hypoxemia. She was discharged with an oral steroid regimen and scheduled for a follow-up appointment with an allergist. Discussion: There exists only a handful of case reports describing angioedema in patients with COVID-19 infection. In those reports, patients also had normal C1 esterase inhibitor levels and no personal or family history of inherited angioedema. Interestingly, our patient was vaccinated six months prior to her presentation. The association between SARS-CoV-2 and angiotensinconverting enzyme 2 (ACE-2), the primary receptor for viral entry into the epithelial cells of the lungs, could be a potential explanation for the occurrence of angioedema. ACE-2 plays a pivotal role in inhibiting a potent ligand of bradykinin receptor 1, Arginine bradykinin. It has been postulated that SARS-CoV-2 downregulation of ACE-2 leads to elevated angiotensin II levels and subsequent activation of the bradykinin pathway. Excessive bradykinin production generates high levels of nitric oxide and prostaglandins, resulting in vasodilation, increased vascular permeability, and angioedema. This case highlights the importance of recognizing atypical and rare presentations of COVID-19 infection, especially angioedema, given its sudden onset and life-threatening complications.
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Introduction: Circadian rhythms have critical roles in human health. We quantified the effect of time-of-day of COVID-19 vaccination and other covariates on self-reported side effects post vaccination. Methods: The dataset was created from MassGeneralBrigham (MGB) electronic health records and REDCap survey that collected self-reported symptoms for 1-3 days after each immunization. Variables are demographics (age, sex, race, and ethnicity), vaccine manufacturer, clock time of vaccine administration/appointment, any COVID-19 diagnosis/positive test prior to vaccination, any history of allergy, and any note of epinephrine self-injection (e.g., EpiPen) medication. Time of day groupings were morning (6 am10 am), midday (10 am2 pm), late afternoon (2 pm6 pm) or evening (6 pm10 pm). Side effects were classified as Allergic (Rash;Hives;Swollen lips, tongue, eyes, or face;Wheezing) and Non-Allergic (New Headache, New Fatigue, Arthralgias, Myalgias, Fever) symptoms. The study was approved by the MGB IRB.Machine learning (ML) techniques (e.g., extreme gradient boosting) were applied to the variables to predict the occurrence of side effects. Stratified k-fold cross validation was used to validate the performance of the ML models. Shapley Additive Explanation values were computed to explain the contribution of each of the variables to the prediction of the occurrence of side effects. Results: Data were from 54,844 individuals. On day 1 after the first vaccination, (i) females, people who received the Moderna vaccine, and those with any allergy history were more likely to report Allergic side effects;and (ii) females, people who received the Janssen vaccine, those who had prior COVID-19 diagnosis ,and those who received their vaccine in the morning or midday and were more likely to report Non-Allergic symptoms. Older persons had fewer side effects of any type. Conclusion: ML techniques identified demographic and time-ofday- of-vaccination effects on side effects reported on the first day after the first dose of a COVID-19 vaccination. We will use these techniques to test for changes on days 2 and 3 after the first dose, and the first 3 days after the second dose and for the influence of recent night or shiftwork. Future work should target underlying physiological reasons.
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Background: Systemic lupus erythematosus (SLE) is an autoimmune disease with a multiple organs-involvement. SLE pathogenesis is sustained by innate and adaptive immune system disregulation, complement activation, immune complexes, and tissue inflammation. The pattern of clinical manifestations is heterogenous and changes over time. Hydroxychloroquine (HCQ)is a primary drug in the treatment of SLE. It modulates the immune response by inhibiting B cell receptor and TLR signaling and activation. Clinical Case: This is a case report of a 61-years-old female affected by SLE with recurrent oral aftosis, Lupus anti-coagulant antibodies and previous pleuritis in remission. The patient had a long course treatment with low-dose oral corticosteroids (5 mg prednisone) and HCQ for three weeks. After 20 days of treatment with HCQ she developed an abrupt cutaneous manifestation consisting in painful and infiltrating papules which appeared over the upper trunk and limbs, redness, scales lesions on palms and soles, lip swelling, painful conjunctivitis and mild disphagia. Slowly, progressive improvement of cutaneous and mucosal signs was observed after drug discontinuation, two days after appearance of clinical manifestations, and introducing medium-high dose of oral corticosteroids therapy. Results: Clinical exams, performed in the acute phase highlighted a marked lymphocyto penia and raise of Reactive C Protein (RCP). Nicolsky sign was slightly positive. After discontinuation of HCQ the patient did not show cutaneous or infective sequelae due to prompt identification of Steven-Johnson Syndrome (SJS). The patient replaced HCQ with the increase of the mainteinance dose of oral corticosteroids. Skin tests were not perfomed for the risk of SJS exacerbation and concomitant corticosteroids therapy. Conclusions: Like all medications, HCQ has side effects which may occur in autoimmune disease patients. The main undesirable effects observed are digestive disorders and skin manifestations. Although skin diseases are common side effects of HCQ severe drug hypersensitivity reaction is a rare condition described in few case reports of DRESS and SJS. Despite the rare occurrence of these reactions, we observed other cases of drug reactions during the pandemic breakthrough in Italy when HCQ was widely used for the treatment of COVID-19.