Coronavirus disease 2019–related Kawasaki-like disease in an adult: A case report

In late 2019, a new viral infection emerged from China, spreading worldwide to cause a pandemic. This coronavirus disease 2019 (COVID-19) is caused by the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). Different clinical patterns have occurred, including a Kawasaki-like disease in children.1Royal College of Paediatrics and Child Health Guidance—Paediatric multisystem inflammatory syndrome temporally associated with COVID-19.https://www.rcpch.ac.uk/resources/guidance-paediatricmultisystem-inflammatory-syndrome-temporally-associated-covid-19Date: 2020Date accessed: May 5, 2020Google Scholar This report describes a similar case in an adult. In late April, 2020, a previously healthy 35-year-old woman was admitted to the infectious diseases department for a 4-day history of fever, myalgia, dyspnea, dry cough, hypogeusia, vomiting, diarrhea, and a pruritic rash. On physical examination, she was weak, irritable, and febrile (38.4°C). She had chapped lips with ulceration above the upper lip (Fig 1), lingual enanthem characterized by a reddish and swollen tongue, bilateral erythematous conjunctivitis, edema of hands and feet, and hepatomegaly. She also presented with a rash on the legs and forearms characterized by erythemato-violaceous macules with irregular borders (Fig 2). She had no cervical lymph node enlargement. Chest auscultation found regular tachycardia and fine crackles on the lower lobes.Fig 2Macular rash with some postinflammatory hyperpigmentation on the right proximal forearm.View Large Image Figure ViewerDownload Hi-res image Download (PPT) Full blood count showed anemia, thrombocytopenia, neutrophilia, lymphopenia, and slight eosinophilia. Laboratory values were C-reactive protein, 367 mg/L (normal <5.0 mg/L); ferritin, 5384 μg/L (67.5-449 μg/L); lactate dehydrogenase, 4.57 μkat/L (2.25-3.57 μkat/L); and triglycerides, 3.42 mmol/L (<1.70 mmol/L). Liver function tests were slightly abnormal (alanine aminotransferase, 1.03 μkat/L [<0.55 μkat/L] and aspartate aminotransferase, 1.07 μkat/L [<0.53 μkat/L]). A bone marrow aspiration was not performed. She had acute renal impairment, and urinalysis results showed hematuria, leukocyturia, and nonnephrotic proteinuria. There was no serologic evidence of connective tissue disease. Serologic assay for HIV was negative. Antibodies against Epstein-Barr virus, cytomegalovirus, and parvovirus B19 were indicative of past infection. Peripheral blood immunophenotyping was normal. Blood and urinary cultures remained negative. Diagnosis of COVID-19 was based on reverse transcription polymerase chain reaction for SARS-CoV-2 from nasopharyngeal swab and thoracoabdominal computed tomography, which showed peripheral interstitial infiltrates of the lower pulmonary lobes and hepatosplenomegaly. Initially, serum cardiac markers such as n-terminal pro–brain natriuretic peptide and troponins were normal but increased on the third day of hospitalization. Electrocardiogram showed sinus tachycardia. Echocardiography was unremarkable, whereas cardiac magnetic resonance imaging, obtained after 1 week, showed global hypokinetic myocardia. She did not undergo coronary angiography. Hydroxychloroquine, azithromycin, and cefuroxime were empirically given for a total of 5 days without any complication. During the second (and last) week of hospitalization, evolution of inflammatory parameters (ferritin, leucocytosis, and C-reactive protein) were favorable together with cardiac (n-terminal pro–brain natriuretic peptide and troponins) and liver markers (aspartate aminotransferase and alanine aminotransferase) and urinary findings (hematuria, leukocyturia, and proteinuria). The lingual enanthem and acral edema resolved within 10 days, whereas the conjunctival injection and chapped lips remained. Four weeks after the onset of the symptoms, desquamation of the hands and feet (Fig 3) occurred. The diagnosis of Kawasaki-like disease was retrospectively suggested, and the patient was subsequently requested to undergo coronary investigation. Kawasaki disease (KD), described by Tomisaku Kawasaki in 1967, is a systemic vasculitis of unknown origin that occurs mainly in children and seldom in adults.2Burns J.C. Glode M.P. Kawasaki syndrome.Lancet. 2004; 364: 533-544Abstract Full Text Full Text PDF PubMed Scopus (774) Google Scholar Infectious triggers may lead to an exaggerated inflammatory response explaining the clinical findings in KD.2Burns J.C. Glode M.P. Kawasaki syndrome.Lancet. 2004; 364: 533-544Abstract Full Text Full Text PDF PubMed Scopus (774) Google Scholar Although incomplete forms of KD exists, diagnosis of KD requires the presence of fever for at least 5 days, 4 of the 5 Kawasaki established criteria (bilateral conjunctival infiltrate, changes of the mucous membranes of the upper respiratory tract, polymorphous rash, changes of the extremities, and cervical adenopathy) and the lack of alternative explanation.2Burns J.C. Glode M.P. Kawasaki syndrome.Lancet. 2004; 364: 533-544Abstract Full Text Full Text PDF PubMed Scopus (774) Google Scholar Our patient fulfilled all the criteria for KD except cervical lymphadenopathy.2Burns J.C. Glode M.P. Kawasaki syndrome.Lancet. 2004; 364: 533-544Abstract Full Text Full Text PDF PubMed Scopus (774) Google Scholar Accordingly, this patient had a Kawasaki-like disease associated with COVID-19 infection. In the time of the COVID-19 pandemic, several recent clinical reports underline the observation of Kawasaki-like disease in children.1Royal College of Paediatrics and Child Health Guidance—Paediatric multisystem inflammatory syndrome temporally associated with COVID-19.https://www.rcpch.ac.uk/resources/guidance-paediatricmultisystem-inflammatory-syndrome-temporally-associated-covid-19Date: 2020Date accessed: May 5, 2020Google Scholar The diagnosis may be challenging and retrospective, as no specific tests exist. Coronary involvement is the main complication of KD but remains less frequent in adults than in children.3Fraison J.B. Sève P. Dauphin C. et al.Kawasaki disease in adults: observations in France and literature review.Autoimmun Rev. 2016; 15: 242-249Crossref PubMed Scopus (44) Google Scholar Early diagnosis allows treatment with intravenous immunoglobulins and aspirin. Intravenous immunoglobulins should be given within the first 10 days but its administration may also be beneficial for delayed diagnosis in some cases.4McCrindle B.W. Rowley A.H. Newburger J.W. et al.Diagnosis, treatment, and long-term management of Kawasaki disease: a scientific statement for health professionals from the American Heart Association.Circulation. 2017; 135: e927-e999Crossref PubMed Scopus (1737) Google Scholar Health professionals should be aware of novel and rare presentations of COVID-19, including pediatric and adult-onset Kawasaki-like disease, especially given the potential complications and therapeutic implications.