Yonsei Med J.  2019 Mar;60(3):312-314. 10.3349/ymj.2019.60.3.312.

Erythema Nodosum Masking Kawasaki Disease with an Initial Manifestation of Skin Lesions

• Okada S
• Ishikawa Y
• Shimomura
• Sunagawa S
• Hirano R
• Fukunaga S
• Miyake A
• Okada Y
• Maki T

• KMID: 2441310
• DOI: http://doi.org/10.3349/ymj.2019.60.3.312

Abstract

We report the first case demonstrating an association between Kawasaki disease (KD) and erythema nodosum (EN). A 3-year-old girl presented with EN as an initial manifestation of KD. At the initial visit, she showed high fever of 40℃, injection of the oropharynx, cervical lymphadenopathy, and red-purple cutaneous nodules, particularly on the lower limbs. She complained of severe pain in the neck and cutaneous lesions. Initially, the development of EN was attributed to Salmonella spp infection, which was detected in stool culture. However, the patient did not respond to high-dose ampicillin/sulbactam to which the Salmonella spp is sensitive. Echocardiography performed as screening for fever of unknown origin revealed medium-sized aneurysms of the left anterior descending artery. EN masked the diagnosis of KD, and the patient developed a coronary artery lesion. KD should be considered in the differential diagnosis of refractory EN in pediatric patients.

Keyword

Bacterial infections; exanthema; inflammatory disorders; subcutaneous tissue

MeSH Terms

Aneurysm
Arteries
Bacterial Infections
Child, Preschool
Coronary Vessels
Diagnosis
Diagnosis, Differential
Echocardiography
Erythema Nodosum*
Erythema*
Exanthema
Female
Fever
Fever of Unknown Origin
Humans
Lower Extremity
Lymphatic Diseases
Masks*
Mass Screening
Mucocutaneous Lymph Node Syndrome*
Neck
Oropharynx
Salmonella
Skin*
Subcutaneous Tissue

Figure

• 

  Fig. 1 Patient's cutaneous manifestations upon admission and findings of the coronary artery lesion. (A) Image shows the patient's presentation upon admission (on the 3rd day of illness). Painful, red-purple cutaneous nodules can be observed (arrows), particularly on the lower limbs. (B) A two-dimensional echocardiography scan obtained on the 10th day of illness shows medium-sized aneurysms of the left anterior descending artery, measuring 4.6 mm (Z: +6.6, indicated by the asterisk). (C) Selective angiographic image of the left coronary artery obtained during the 2nd month of illness shows that the luminal dimension of the left anterior descending artery has regressed to its normal diameter. Ao, aorta.

Reference

1. Ozbagcivan O, Akarsu S, Avci C, Inci BB, Fetil E. Examination of the microbial spectrum in the etiology of erythema nodosum: a retrospective descriptive study. J Immunol Res. 2017; 2017:8139591.

2. Aydın-Teke T, Tanır G, Bayhan GI, Metin O, Oz N. Erythema nodosum in children: evaluation of 39 patients. Turk J Pediatr. 2014; 56:144–149.

3. Kakourou T, Drosatou P, Psychou F, Aroni K, Nicolaidou P. Erythema nodosum in children: a prospective study. J Am Acad Dermatol. 2001; 44:17–21.

4. Cribier B, Caille A, Heid E, Grosshans E. Erythema nodosum and associated diseases. A study of 129 cases. Int J Dermatol. 1998; 37:667–672.

5. Psychos DN, Voulgari PV, Skopouli FN, Drosos AA, Moutsopoulos HM. Erythema nodosum: the underlying conditions. Clin Rheumatol. 2000; 19:212–216.

6. Hara T, Nakashima Y, Sakai Y, Nishio H, Motomura Y, Yamasaki S. Kawasaki disease: a matter of innate immunity. Clin Exp Immunol. 2016; 186:134–143.

7. McCrindle BW, Rowley AH, Newburger JW, Burns JC, Bolger AF, Gewitz M, et al. American Heart Association Rheumatic Fever, Endocarditis, and Kawasaki Disease Committee of the Council on Cardiovascular Disease in the Young. Council on Cardiovascular and Stroke Nursing. Council on Cardiovascular Surgery and Anesthesia. Council on Epidemiology and Prevention. Diagnosis, treatment, and long-term management of Kawasaki disease: a scientific statement for health professionals from the American Heart Association. Circulation. 2017; 135:e927–e999.

8. Kim KY, Kim DS. Recent advances in Kawasaki disease. Yonsei Med J. 2016; 57:15–21.

9. Ming A, Wargon O. Annular lesions in Kawasaki disease: a cause of confusion. Australas J Dermatol. 2008; 49:207–212.

10. Okada S, Kobayashi-Fujiwara Y, Oga A, Furuta T, Ikemoto K, Fujii H, et al. Distinct distribution of immunocytes in a retropharyngeal lymphadenopathy associated with Kawasaki disease: a case study compared with tonsillitis. Cardiology. 2017; 137:237–243.

11. Cai Z, Zuo R, Liu Y. Characteristics of Kawasaki disease in older children. Clin Pediatr (Phila). 2011; 50:952–956.