Pleural Effusion as an Atypical Presentation of Kawasaki Disease

A Case Report and Review of the Literature

Elif Arslanoglu Aydin; Selcan Demir; Orkun Aydin; Yelda Bilginer; Seza Ozen

J Med Case Reports. 2019;13(344)

Abstract and Introduction

Abstract

Background: Kawasaki disease is an acute, febrile vasculitis of childhood that affects medium-sized arteries, predominantly the coronary arteries. It is a multisystem disease; therefore, it may present with non-cardiac findings of disease.

Case presentation: Here, we report the case of 7-year-old Turkish girl who presented with symptoms of fever, chest pain, and vomiting, who was diagnosed as having Kawasaki disease. We also present a literature review on pulmonary involvement due to Kawasaki disease.

Conclusion: Pediatricians should consider the diagnosis of Kawasaki disease in the presence of pneumonia and pleural effusion that is nonresponsive to antibiotic therapy. This will prevent delay in diagnosis and the adverse consequences of the disease.

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Table 1. Clinical symptoms and laboratory parameters of patients who had pulmonary involvement associated with Kawasaki disease
Authors, year, reference number	Rash	Oral changes	Extremity changes	Red eyes	Adenitis	Other clinical symptoms	Hb (g/dL)	WBC (/mm³)	Plt (/mm³)	CRP (mg/dL)	ESR (mm/hour)
Singh et al., 2018, [6]	5^a	1 ^a	8^a	1 ^a	0 ^a	Perianal desquamation, 3 ^a; irritability, 1 ^a	NA	25,009^b	886,545^b	14.05^c	53.75 ^d
Alhammadi and Hendaus, 2013, [12]	NA	NA	NA	NA	NA	NA	NA	24,000	600,000	10	65
Lee et al., 2011, [11]	NA	NA	NA	NA	NA	NA	NA	NA	NA	NA	NA
Lee et al., 2010, [14]	Yes	Yes	Yes	Yes	Yes	Perianal desquamation	NA	5500	178,000	2.8	21
Falcini et al., 2009, [15]	No	No	No	Yes	No	Irritability	9.5	21,800	710,000	27.8	99
Elizabeth et al., 2007, [16]	No	Yes	Yes	No	Yes	Irritability	10.2	12,800	550,000	7.7	86
Yavuz et al., 2007, [17]	Yes	Yes	Yes	Yes	No	Non-pigmented keratic precipitates in both of the patient's eyes, sterile pyuria	10.4	32,800	734,000	21.8	90
Sittiwangkul and Pongprot, 2004, [13]	Yes	No	Yes	Yes	No	Irritability	NA	21,200	231,000	4.77	66
de Magalhães et al., 2012, [21]	Yes	Yes	Yes	Yes	No	Induration at the BCG site, perianal desquamation	6.5	25,000	905,000	34	120
Hamada et al., 2005, [18]	Yes	Yes	Yes	No	Yes	Hepatomegaly	NA	17,800	NA	13.9	NA
D'Souza et al., 2006, [20]	No	No	Yes	No	Yes	Sterile pyuria	9.8	56,800	690,000	NA	138
de Maddi et al., 2009, [22]
Case 1	No	Yes	No	Yes	Yes	No	8.7	11,200	561,000	10.4	70
Case 2	No	No	No	No	No	No	9	26,960	142,000	40.8	107
Case 3	Yes	Yes	No	Yes	No	Irritability, sterile pyuria	11	18,500	1,087,000	2.95	50
Freeman et al., 2003, [23]
Case 1	Yes	Yes	No	Yes	No	Irritability	NA	NA	1,120,000	NA	NA
Case 2	No	Yes	No	Yes	Yes	Torticollis	NA	NA	1,102,000	NA	114
Case 3	Yes	No	No	No	Yes	Anorexia	NA	NA	450,000	10.5	NA
Kobayashi et al., 2006, [24]
Case 1	Yes	Yes	No	Yes	Yes		9.6	13,000	321,000	12.6	102
Case 2	Yes	Yes	Yes	Yes	No	Induration at the BCG site	11.6	18,800	314,000	7.6	NA
Vaidya et al., 2017, [8]	Yes	Yes	Yes	No	No		8.9	15,600	567,000	5.6	40
Akagi et al., 2017, [19]
Case 1	Yes	Yes	Yes	No	No		NA	NA	NA	4.26	NA
Case 2	Yes	Yes	No	No	No		NA	NA	NA	4.32	NA

^aNumber of the patients who had the symptom
^b,c,dValues are expressed as mean for 11, 8, and 10 patients, respectively
BCG Bacille Calmette–Guérin, CRP C-reactive protein, ESR erythrocyte sedimentation rate, Hb hemoglobin, NA not available, Plt platelet, WBC white blood cell

Table 2. Demographic parameters and clinical presentations of patients who had pulmonary involvement associated with Kawasaki disease
Authors, year, reference number	Patients (n)	Sex	Age at onset of disease (months)	Initial symptoms	Fever duration (days)	Chest X-ray findings
Singh et al., 2018, [6]	11	F, 6 ^a;M, 5 ^a	30 ^b	Fever, cough, tachypnea	14.1 ^b	Consolidation, 11 ^a; pleural effusion, 6 ^a;empyema, 3 ^a; pneumothorax, 2 ^a
Alhammadi and Hendaus, 2013, [12]	1	F	36	Fever, cough, sore throat	18	Consolidation, pleural effusion
Lee et al., 2011, [11]	54	NA	NA	NA	NA	Reticulonodular, 17 ^a; opacification, 34 ^a; consolidation, 12 ^a; pleural effusion, 5 ^adiffuse interstitial, 5 ^a; atelectasis, 2 ^a;
Lee et al., 2010, [14]	1	M	22	Fever, cough, rhinorrhea	6	Infiltration, pleural effusion
Falcini et al., 2009, [15]	1	F	30	Fever, cough,	12	Pleural effusion
Elizabeth et al., 2007, [16]	1	F	36	Fever, gum bleeding	21	Pleural effusion
Yavuz et al., 2007, [17]	1	M	11	Fever, pharyngeal erythema, dyspnea	> 5	Pleural effusion
Sittiwangkul and Pongprot, 2004, [13]	1	F	11	Fever, jaundice, diarrhea, dyspnea	15	Pleural effusion
de Magalhães et al., 2012, [21]	1	F	3	Fever	10	Infiltration
Hamada et al., 2005, [18]	1	F	60	Fever, abdominal pain, knee joint pain	15	Pleural effusion
D'Souza et al., 2006, [20]	1	M	5	Fever, diarrhea, dyspnea	7	Pleural effusion
Case 1	1	M	8	Fever, sore throat	8	Consolidation
Case 2	1	F	11	Febrile seizure, sore throat, cough	14	Consolidation
Case 3	1	F	23	Fever, cough	10	Consolidation
Freeman et al., 2003, [23]
Case 1	1	M	4	Fever, cough, rash	21	NA
Case 2	1	M	6	Fever	60	Normal;thorax CT, pulmonary nodule
Case 3	1	NA	5	Fever cough, rash	4	Infiltration, multiple pulmonary nodules
Kobayashi et al., 2006, [24]
Case 1	1	F	24	Fever, cracked lips, rash	5	Infiltration, pleural effusion
Case 2	1	F	24	Fever, cough, nasal discharge	4	Atelectasis
Vaidya et al., 2017, [8]	1	F	3	Fever, rash, dyspnea	32	Hydropneumothorax, consolidation, pneumatoceles
Akagi et al., 2017, [19]
Case 1	1	F	4	Fever, erythema of the lips, rash	NA	NA;thorax MRI, bilateral multiplepulmonary nodules
Case 2	1	F	5	Fever, erythema of the lips, rash	9	Infiltration;thorax CT, bilateral pulmonary nodules

^aNumber of the patients who had noted findings
^bValues are expressed as mean for 11 patients
CT computed tomography, F female, M male, MRI magnetic resonance imaging, NA not available

Table 3. Treatment, coronary artery involvement, follow-up, and outcomes of patients who had pulmonary involvement associated with Kawasaki disease
Authors, year, reference number	Infectious agent	Antibiotic treatment	CAI	Treatment	Follow-up and outcome
Singh et al., 2018, [6]	2 ^a	11 ^a	3 ^a	2 ^a, Second dose of IVIG	9 ^a Normal, 2 ^a NA
Alhammadi and Hendaus, 2013, [12]	No	Yes	Yes	IVIG	Normal
Lee et al., 2011, [11]	NA	NA	NA	NA	NA
Lee et al., 2010, [14]	No	No	Yes	Second dose of IVIG and corticosteroid	Normal
Falcini et al., 2009, [15]	No	Yes	Yes	Second dose of IVIG and corticosteroid	Normal
Elizabeth et al., 2007, [16]	No	Yes	Yes	Second dose of IVIG and corticosteroid	Normal
Yavuz et al., 2007, [17]	No	Yes	Yes	Second dose of IVIG	Normal
Sittiwangkul and Pongprot, 2004, [13]	No	Yes	Yes	Second dose of IVIG and corticosteroid	Aneurysm persisted in 2 years
de Magalhães et al., 2012, [21]	No	Yes	Yes	Second dose of IVIG, corticosteroid, MTX, and ETN	Aneurysm decrease but persisted
Hamada et al., 2005, [18]	No	Yes	No	Second dose of IVIG and corticosteroid	Normal
D'Souza et al., 2006, [20]	No	Yes	No	IVIG	Normal
de Maddi et al., 2009, [22]
Case 1	No	Yes	No	IVIG	Normal
Case 2	No	Yes	No	Not given IVIG	Normal
Case 3	No	Yes	No	IVIG	Normal
Freeman et al., 2003, [23]
Case 1	No	Yes	Yes	IVIG	Death
Case 2	NA	Yes	Yes	IVIG	Normal
Case 3	No	Yes	Yes	IVIG	Normal
Kobayashi et al., 2006, [24]
Case 1	Yes	Yes	NA	IVIG	Normal
Case 2	Yes	Yes	NA	IVIG	Normal
Vaidya et al., 2017, [8]	No	Yes	Yes	IVIG	NA
Akagi et al., 2017, [19]
Case 1	No	No	Yes	IVIG	Normal
Case 2	No	Yes	Yes	IVIG	Normal

^aNumber of the patients
CAI coronary artery involvement, ETN etanercept, IVIG intravenous immunoglobulin, MTX methotrexate, NA not available

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Pleural Effusion as an Atypical Presentation of Kawasaki Disease

A Case Report and Review of the Literature

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