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Erythema and Induration of Bacillus Calmette-Guérin Scar Associated With Multisystem Inflammatory Syndrome in
Naoki Tsuboya1, Hirotoshi Makino1, Yoshihide Mitani1
1Department of Pediatrics, Mie University Graduate School of Medicine, Tsu, Japan.
Insights
Multisystem Inflammatory Syndrome in Children (MIS-C) can present with Bacillus Calmette-Guérin (BCG) scar reactions, mimicking Kawasaki disease (KD). This case highlights a potential diagnostic clue for MIS-C in BCG-vaccinated populations.
Area of Science:
- Pediatrics
- Infectious Diseases
- Immunology
Background:
- Multisystem Inflammatory Syndrome in Children (MIS-C) is a rare post-COVID-19 condition with overlapping features with Kawasaki disease (KD).
- Bacillus Calmette-Guérin (BCG) scar reactions are characteristic of KD but have not been reported in MIS-C.
- The relationship between MIS-C and KD remains incompletely understood.
Observation:
- A 3-year-old boy with a history of BCG vaccination presented with MIS-C symptoms, including fever, gastrointestinal issues, and complete KD features.
- The patient exhibited erythema and induration at the BCG scar from the early disease stage.
- Congestive heart failure developed post-initial treatment, with the patient ultimately responding to a third IVIG dose combined with prednisolone.
Findings:
- This is the first reported case of BCG scar changes associated with MIS-C.
- The patient's presentation included MIS-C criteria, KD features, and BCG scar inflammation.
- No coronary artery abnormalities were observed despite KD-like symptoms.
Implications:
- BCG scar reactions may serve as a diagnostic indicator for MIS-C in populations with mandatory BCG vaccination.
- This finding could offer insights into the pathogenesis of MIS-C and its relationship to KD.
- Further research is needed to explore the full spectrum of MIS-C and its overlap with KD, particularly in BCG-vaccinated children.
Abstract:
Multisystem Inflammatory Syndrome in Children (MIS-C) is a rare febrile disorder with multisystem organ involvement temporally associated with coronavirus 2019 infection (COVID-19) and frequently exhibits features mimicking Kawasaki disease (KD), another febrile disorder in children. The pathogenesis and the full clinical spectrum of MIS-C is poorly understood: It is still unclear whether MIS-C and KD are different syndromes or represent a common spectrum. The erythema and induration of Bacillus Calmette-Guérin (BCG) scar is one of the characteristic findings of KD, and is useful for the diagnosis in countries where BCG vaccination is mandated in infancy. Furthermore, such findings in BCG scar were also reported after SARS-CoV-2 vaccination, which may be related to molecular mimicry. However, there are no reports of changes at the BCG scar in MIS-C cases. Here, we report a case of MIS-C in a 3-year-old Hispanic boy in Japan, with erythema and induration at the BCG scar. The patient received BCG vaccination at 16 months of age in Japan. Four weeks before the onset, he had positive polymerase chain reaction (PCR) results for SARS-CoV-2 following household outbreak, although he was asymptomatic. He presented with fever and gastrointestinal symptoms, followed by the appearance of all six principal findings of complete KD. He exhibited congestive heart failure, following intravenous immunoglobulin (IVIG) therapy. He was diagnosed with MIS-C based on characteristic mucocutaneous and gastrointestinal symptoms, decreased cardiac function, and coagulopathy, in addition to laboratory data consistent with MIS-C. The BCG finding was present from the early stage of the disease. The patient was refractory to two doses of IVIGs, and the third IVIG plus prednisolone resulted in defervescence and improvement in heart failure. No coronary involvement was observed. This is the first case of erythema and induration at the BCG scar associated with MIS-C accompanied by KD features, which may give clinical and mechanistic insights in the understanding of the disease. Since the full spectrum of MIS-C is still evolving and both of them are syndromes with overlapped clinical features, further studies are warranted for deep phenotyping of MIS-C with KD features relative to KD in countries with mandatory BCG programs in infancy.
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