TY - JOUR
T1 - Resolution of Giant Coronary Aneurisms in a Child With Refractory Kawasaki Disease Treated With Anakinra
AU - Gambacorta, Alessandro
AU - Buonsenso, Danilo
AU - De Rosa, Gabriella
AU - Lazzareschi, Ilaria
AU - Gatto, Antonio
AU - Brancato, Federica
AU - Pata, Davide
AU - Valentini, Piero
PY - 2020
Y1 - 2020
N2 - Kawasaki disease (KD) is an acute, febrile illness of unknown etiology that mainly
affects children under 5 years of age. intravenous immunoglobulin (IVIG), the standard
treatment, has reduced coronary involvement to <5%. Patients who do not improve
after an initial IVIG have a higher risk of developing coronary arteries aneurysms, and
its optimal treatment remains controversial. We present a case of IVIG, steroids, and
infliximab-resistant KD in a 9-month-old child, which developed giant aneurysms and
was successfully treated with anakinra, a recombinant antagonist of the IL-1 receptor. In
our case, the introduction of IL-1 receptor antagonist therapy seems to have blocked the
disease from both a clinical and a laboratory point of view. We also noted a very rapid
regression of coronary aneurysms passed from giant aneurysms to small ones, or, as in
the case of the anterior descending artery, the complete disappearance of the aneurysm
formation. We think that our case adds more evidences to the potential role of IL-1RA as
therapy in some selected cases of refractory KD, in particular with severe involvement of
coronary arteries, although new efficacy trials are needed to better understand the role
of Anakinra in these patients.
AB - Kawasaki disease (KD) is an acute, febrile illness of unknown etiology that mainly
affects children under 5 years of age. intravenous immunoglobulin (IVIG), the standard
treatment, has reduced coronary involvement to <5%. Patients who do not improve
after an initial IVIG have a higher risk of developing coronary arteries aneurysms, and
its optimal treatment remains controversial. We present a case of IVIG, steroids, and
infliximab-resistant KD in a 9-month-old child, which developed giant aneurysms and
was successfully treated with anakinra, a recombinant antagonist of the IL-1 receptor. In
our case, the introduction of IL-1 receptor antagonist therapy seems to have blocked the
disease from both a clinical and a laboratory point of view. We also noted a very rapid
regression of coronary aneurysms passed from giant aneurysms to small ones, or, as in
the case of the anterior descending artery, the complete disappearance of the aneurysm
formation. We think that our case adds more evidences to the potential role of IL-1RA as
therapy in some selected cases of refractory KD, in particular with severe involvement of
coronary arteries, although new efficacy trials are needed to better understand the role
of Anakinra in these patients.
KW - kawasaki disease, anakinra, IL-1 receptor antagonist, Coronary aneurysms, personalized medicine, precision medicine, paediatrics 2
KW - kawasaki disease, anakinra, IL-1 receptor antagonist, Coronary aneurysms, personalized medicine, precision medicine, paediatrics 2
UR - http://hdl.handle.net/10807/154034
U2 - 10.3389/fped.2020.00195
DO - 10.3389/fped.2020.00195
M3 - Article
SN - 2296-2360
VL - 8
SP - N/A-N/A
JO - Frontiers in Pediatrics
JF - Frontiers in Pediatrics
ER -