Diffuse coronary artery dilatation in a neonate: A case report

Parashar, Rachna; Lysecki, Paul; Mondal, Tapas

doi:10.3233/npm-1365612

Cited by 7 publications

(3 citation statements)

References 4 publications

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“…Krapf et al described a neonate who presented with fatal myocardial infarction without any clinical features of KD, who at autopsy, was found to have an acute/subacute necrotizing coronary vasculitis typical of KD [ 15 ]. In a more recent case report by Parashar et al, bilateral coronary artery dilatations were detected on the first day of life in the absence of other KD features [ 14 ]. Bolz et al described a baby who presented during his first week of life in congestive heart failure who was found to have dilatation of his left coronary arteries without other KD features [ 13 ].…”

Section: Discussionmentioning

confidence: 99%

Kawasaki Disease in the neonate: case report and literature review

Altammar

Lang

2018

Pediatr Rheumatol

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BackgroundKawasaki Disease (KD), the leading cause of acquired heart disease in children in the developed world, is extremely rare in neonates. We present a case of incomplete KD in a neonate and a review of the literature on neonatal KD.Case presentationA previously healthy full term 15 day old Caucasian male with an unremarkable antenatal and perinatal history, presented on Day 2 of illness with fever, rash, irritability, and poor feeding. Examination revealed fever (39.6C), tachycardia, tachypnea, extreme irritability, and a generalized maculopapular rash, but was otherwise normal. His complete blood count, CRP and ESR were normal. Empiric intravenous antibiotics and acyclovir resulted in no improvement. On day 4, he had ongoing fever and developed recurrent apnea, required supplemental oxygen, and was transferred to the pediatric intensive care unit. On day 6, he developed bilateral non-purulent conjunctivitis, palmar erythema, bilateral non-pitting edema and erythema of his feet, and arthritis. His full septic work-up and viral studies were negative. On Day 7 he was treated with intravenous immunoglobulin, and over the next 48 h his symptoms including extremity edema resolved, he no longer required supplemental oxygen, and fever did not recur. On day 9 of illness he had marked thrombocytosis. Subsequently, he developed distal extremity desquamation. Repeated echocardiograms excluded the presence of coronary artery aneurysms (CAA).ConclusionsWe believe this to be a rare case of incomplete KD in a neonate, in which timely IVIG administration led to resolution of the acute illness and may have prevented CAA. A comprehensive English-language medical literature review of KD presenting in the neonatal period revealed only fifteen case reports. Cases often presented with incomplete KD, and a number had atypical laboratory features including a normal CRP in the acute phase, similar to what was seen in our patient. This case and our literature review should increase awareness that KD can rarely occur in neonates, often presenting atypically. Recognizing KD in a neonate enables appropriate treatment that can result in resolution of symptoms and may decrease the risk of cardiac complications.

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Section: Discussionmentioning

confidence: 99%

Kawasaki Disease in the neonate: case report and literature review

Altammar

Lang

2018

Pediatr Rheumatol

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“…There were four patients with no fever among the 19 neonatal KD cases in our literature review. Only one afebrile patient was diagnosed as IKD purely based on CALs [ 11 ]. The other three afebrile patients [ 9 – 10 ], including the present case, had the same clinical manifestations, such as rash and periungual desquamation.…”

Section: Discussionmentioning

confidence: 99%

Kawasaki disease in neonates: a case report and literature review

Shen,

Liu,

et al. 2024

Pediatr Rheumatol

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Background Kawasaki disease (KD) is an acute systemic vasculitis of unknown etiology that affects infants and young children but is extremely rare in neonates, especially afebrile KD. We present a case of KD without fever in a neonate and review the literature on KD in neonates. Case presentation A newborn female was hospitalized because her peripheral blood leukocytes increased for half a day. The admission diagnosis was considered neonatal sepsis and bacterial meningitis. She had no fever since the admission, but a rash appeared on her face by the 7th day. On day 11 after admission, there was a desquamation on the distal extremities. On day 15 after admission, ultrasound showed non-suppurative cervical lymphadenopathy. Echocardiogram revealed coronary artery aneurysms in both sides. Finally, the patient was diagnosed with incomplete KD (IKD). The follow-up echocardiogram showed that the internal diameter of both coronary arteries returned to normal three months after birth. Conclusions Fever, rash, and distal extremity desquamation during the recovery phase are the most common symptoms of IKD. When newborns present with clinical manifestations such as rash, distal extremity desquamation and cervical lymph adenitis and with an increased peripheral blood leukocyte count and progressive increase in platelets simultaneously, the medical staff should be highly alert to the possibility of KD even without fever. The echocardiogram needs to be performed promptly. The incidence of coronary artery lesions is significantly higher if neonatal KD patients miss timely diagnosis and treatment.

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“…Our literature search revealed only a few reported cases of suspected NKD worldwide (18 cases, including 14 English literature and 4 Chinese literature) (Table 1) [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20], together with this case, 19 cases in total. In all the presented cases including ours, 12 were male and 7 were female, the age of onset was 1-26 days.…”

Section: Discussionmentioning

confidence: 99%

Neonatal Kawasaki Disease: Case Report and Literature Review

Wang

et al. 2020

Preprint

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Background: Kawasaki Disease (KD) is a self-limiting and acute systemic vasculitis of childhood that leads to coronary artery abnormality in about 25% of untreated cases. KD is extremely rare in neonates. The purpose of this paper is to explore the clinical features and diagnosis and treatment of Neonatal Kawasaki Disease (NKD) for early identification. Case presentation: A boy was a 24-day-old full-term infant with no special birth history. He had fever, irritability and rash at the time of admission. After 4 days of treatment with empirical antibiotics, the rash and fever were still present, and the rash was progressively increased. It is considered that the infection was serious, and antibiotics were changed and given intravenous immunoglobulin. The temperature and rash were better than before. On day 5, he had cracking of lips, on day 9, he had lymph node enlargement in the neck, and molted fingers appeared on the 10th day. Thus the complete Kawasaki Disease (CKD) was diagnosed, and the patient was given intravenous immunoglobulin (IVIG) and aspirin. Within 48h after the start of this therapy, the fever declined, the chapped lips got better, the rash resolved, and distal extremity desquamation appeared. On day 5, 13, and 18, there was no abnormality in echocardiography and no coronary dilatation. On day 20, the chapped lips and the distal extremity desquamation got better. After discharge, the patient continued to take aspirin orally. The patient was followed up to 1 year old, with good prognosis and no carditis or coronary artery abnormalities. Our literature search revealed only a few reported cases of suspected NKD worldwide (18 cases, including 14 English literature and 4 Chinese literature) (Table 1), together with this case, 19 cases in total. Among these 19 cases, 12 were male and 7 were female, the age of onset was 1-26 days. Six patients had complete KD (31.5%), 13 patients were IKD. Clinical manifestations: there were 17 cases of fever (89.5%, 13cases of fever >5days), 13cases (68.4%) of early limb edema/swelling or peeling, 16 cases (84.2%)of skin rash, 14cases (73.7%)of oral mucosal changes (lips red/cleft or strawberry tongue), 10 cases (53%) of coronary artery changes, 7cases (36.8%) of conjunctival congestion, 3cases（15.8%）of cervical lymph node enlargement. Laboratory investigations: There were 5 cases (35.7%) with elevated ESR, 9 cases normal, 5cases unknown. CRP was normal in 6 cases (33%). Thrombocytopenia occurred in 11cases (64.7%). Among the 10 patients with coronary artery changes, 1 had an unknown prognosis due to loss of follow-up (patient 15). One patient died 2 days after discharge due to multiple organ failure (patient 17). 4 cases returned to normal (patient 3.6.7.14). The coronary artery changes in 4 cases still did not return to normal and were not followed up for a long time. Of the 19 NKD patients, 16 were sensitive to IVIG, 1did not use IVIG, and 1 was not sensitive to IVIG，and due to multiple organ failure, the parents gave up treatment and died two days after discharge.Conclusion: NKD is extremely rare, and its clinical manifestation is not typical and easy to be missed. If not treated early, it will potentially give rise to coronary artery aneurysms or expansion, ischemic heart disease, and sudden death. Early diagnosis and treatment are very important.

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Diffuse coronary artery dilatation in a neonate: A case report

Cited by 7 publications

References 4 publications

Kawasaki Disease in the neonate: case report and literature review

Kawasaki Disease in the neonate: case report and literature review

Kawasaki disease in neonates: a case report and literature review

Neonatal Kawasaki Disease: Case Report and Literature Review

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