Clinical and Laboratory ObservationResistant Kawasaki Disease Treated with Anti-CD20
Section snippets
Case Report
A 6-year-old child with a fever was admitted for a marked cervical lymphadenopathy, stiff neck, and trismus evolving in a 4-day period, despite being given antibiotics. A discreet perineal eruption was found. Blood analysis results showed leukocytosis (13.4 G/L), with predominance of neutrophils (10.92 G/L) and elevated acute phase reactants with C-reactive protein level of 53 mg/L and fibrinogen level of 7.91 g/L.
On the next day, a non-purulent conjunctivitis with uveitis and cheilitis
Discussion
Because of the resistance to the IVIG treatment, as proposed by the 2004 guidelines,2 we decided to start steroid therapy.3 In case of failure, like in our case report, immunomodulatory treatments would also have been discussed, on the basis of the supposed physiopathology of KD, with production of proinflammatory cytokines (interleukin [IL] 1), TNF-α, and interferon gamma), and endothelial cell activation.4 Methotrexate inhibits the production of leukotrienes and leads to a diminution of the
References (10)
- et al.
A single intravenous infusion of gamma globulin as compared with four infusions in the treatment of acute Kawasaki syndrome
N Engl J Med
(1991) - et al.
Diagnosis, treatment and long-term management of Kawasaki disease: a statement for health professionals from the committee on rheumatic fever, endocarditis, and Kawasaki disease, council on cardivascular diseases in the young, American Heart Association
Pediatrics
(2004) - et al.
Re-treatment for immune globulin-resistant Kawasaki disease: a comparative study of additional immune globulin and steroid pulse therapy
Pediatr Int
(2001) - et al.
Low-dose methotrexate therapy for intravenous immunoglobulin-resistant Kawasaki disease
Yonsei Med J
(2008) - et al.
Infliximab treatment of intravenous immunoglobulin-resistant Kawasaki disease
J Pediatr
(2008)
Cited by (23)
Monoclonal antibody and anti-cytokine biologics for Kawasaki disease: A systematic review and meta-analysis
2021, Seminars in Arthritis and RheumatismCitation Excerpt :However, clinical trials of anti-TNF-α agents such as infliximab and etanercept as an initial or additional therapy for refractory KD patients have demonstrated contradictory results [10–12]. Furthermore, research on other monoclonal antibodies, including anakinra, rituximab, and tocilizumab, is limited to case reports or case series [13–15]. Taken together, the body of evidence regarding the effectiveness of monoclonal antibody and other anti-cytokine therapeutic agents for KD has not been fully investigated.
Developments in Large and Midsize Vasculitis
2013, Rheumatic Disease Clinics of North AmericaCitation Excerpt :Eighteen of 20 patients were effectively treated with regression of the dilated coronary artery to normal size in the convalescent phase.39 Rituximab was described as effective in one case and there are no cases reported with abatacept and tocilizumab treatment.40 A periodic follow-up of patients is recommended regardless of cardiovascular compromise, because these patients can develop complications during adulthood.
Kawasaki disease: Part II. Complications and treatment
2013, Journal of the American Academy of DermatologyCitation Excerpt :Plasma exchange, reported in 176 cases, is typically provided for 3 consecutive days; while the speed of the response is unclear, all cases improved except for 6 that had residual sequelae (level IIB).86,108-111 Rituximab and anakinra are currently controversial drugs that lack sufficient supportive evidence to be recommended as therapeutic options (level IV).112,113 The complications of untreated KD can be devastating and even life-threatening.
Kawasaki disease: Part I. Diagnosis, clinical features, and pathogenesis
2013, Journal of the American Academy of DermatologyCitation Excerpt :No reactivity occurs when KD patients are tested with other intradermal antigens.59 Findings of the oropharyngeal mucosa in KD can take many forms, including dryness and fissuring of the lips, erythema of the lips (Figs 4 and 7), oral cavity, buccal mucosa, and pharyngeal mucosa, nonexudative inflammation of the throat, and strawberry tongue.3,10,45-47,62 Just 1 of these findings is sufficient to meet this diagnostic criterion.
Intravenous immunoglobulin for the treatment of Kawasaki disease
2023, Cochrane Database of Systematic ReviewsEpidemiology of Kawasaki Disease in children in Switzerland: a national prospective cohort study
2022, Swiss Medical Weekly
The authors declare no conflicts of interest.