Neuropediatrics 2018; 49(06): 392-396
DOI: 10.1055/s-0038-1660855
Short Communication
Georg Thieme Verlag KG Stuttgart · New York

A Pilot Study Evaluating Cerebral Vasculitis in Kawasaki's Disease

Jung Sook Yeom
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Young Hye Cho
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
,
Chung Mo Koo
3   Department of Pediatrics, Gyeongsang National University Hospital, Changwon, Republic of Korea
,
Jin Su Jun
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Ji Sook Park
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Eun Sil Park
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Ji-Hyun Seo
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Jae-Young Lim
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Hyang-Ok Woo
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
,
Hee-Shang Youn
1   Department of Pediatrics, Gyeongsang National University of Hospital, Jinju, Republic of Korea
2   Institute of Health Science, Gyeongsang National University, Jinju, Republic of Korea
› Author Affiliations
Funding No funding to declare.
Further Information

Publication History

28 November 2017

04 May 2018

Publication Date:
18 June 2018 (online)

Abstract

Cerebral vasculitis is thought to be a possible underlying mechanism of severe neurological complications of Kawasaki's disease (KD), such as cerebral infarct or aneurysm rupture. To evaluate the intracranial inflammatory response in patients with acute-stage KD, we measured the levels of cytokines (interleukin [IL]-6 and tumor necrosis factor [TNF]-α) and pentraxin-3 (PTX3) in the cerebrospinal fluid of patients with KD (n = 7) and compared the levels to those of the age- and sex-matched febrile control patients (bacterial meningitis [n = 5], enteroviral meningitis [n = 10], nonspecific viral illness without central nervous system involvement [n = 10]). PTX3 and TNF-α were rarely detected and only in trace concentration in KD, and the levels of IL-6 were not different from those of nonspecific viral illnesses. These mediators are not established biomarkers for cerebral vasculitis but might reflect vascular inflammation in various diseases including KD. Therefore, intracranial inflammation including vasculitis seems to be insignificant in our patients with KD. However, our results might be attributed to the fact that these patients lacked any clinical signs of cerebral or coronary vessel involvement. None of them underwent brain imaging. To clarify this issue, further studies involving patients with neurologic symptoms and proven involvement of cerebral vessels are needed.

 
  • References

  • 1 Landing BH, Larson EJ. Pathological features of Kawasaki disease (mucocutaneous lymph node syndrome). Am J Cardiovasc Pathol 1987; 1 (02) 218-229
  • 2 Dietz SM, Tacke CE, de Groot E, Kuipers IM, Hutten BA, Kuijpers TW. ; Dutch Kawasaki Study Group. Extracardial vasculopathy after Kawasaki disease: a long-term follow-up study. J Am Heart Assoc 2016; 5 (07) e003414
  • 3 Sabatier I, Chabrier S, Brun A. , et al. Stroke by carotid artery complete occlusion in Kawasaki disease: case report and review of literature. Pediatr Neurol 2013; 49 (06) 469-473
  • 4 Ichiyama T, Nishikawa M, Hayashi T, Koga M, Tashiro N, Furukawa S. Cerebral hypoperfusion during acute Kawasaki disease. Stroke 1998; 29 (07) 1320-1321
  • 5 Ahn JH, Phi JH, Kang HS. , et al. A ruptured middle cerebral artery aneurysm in a 13-month-old boy with Kawasaki disease. J Neurosurg Pediatr 2010; 6 (02) 150-153
  • 6 Takahashi K, Oharaseki T, Yokouchi Y, Hiruta N, Naoe S. Kawasaki disease as a systemic vasculitis in childhood. Ann Vasc Dis 2010; 3 (03) 173-181
  • 7 Hirohata S, Tanimoto K, Ito K. Elevation of cerebrospinal fluid interleukin-6 activity in patients with vasculitides and central nervous system involvement. Clin Immunol Immunopathol 1993; 66 (03) 225-229
  • 8 Salvarani C, Brown Jr RD, Calamia KT. , et al. Efficacy of tumor necrosis factor alpha blockade in primary central nervous system vasculitis resistant to immunosuppressive treatment. Arthritis Rheum 2008; 59 (02) 291-296
  • 9 Licata G, Pinto A, Tuttolomondo A. , et al. Anti-tumour necrosis factor alpha monoclonal antibody therapy for recalcitrant cerebral vasculitis in a patient with Behçet's syndrome. Ann Rheum Dis 2003; 62 (03) 280-281
  • 10 Fazzini F, Peri G, Doni A. , et al. PTX3 in small-vessel vasculitides: an independent indicator of disease activity produced at sites of inflammation. Arthritis Rheum 2001; 44 (12) 2841-2850
  • 11 Katsube Y, Akao M, Hashimoto Y. , et al. Pentraxin 3 can be a candidate for biomarker of Kawasaki disease. Circulation 2015; 131: A86
  • 12 Tombetti E, Di Chio MC, Sartorelli S. , et al. Pentraxin 3 can be a candidate for biomarker of Kawasaki disease. Paper presented at: 11th International Kawasaki Disease Symposium; February 4, 2015; Honolulu, Hawaii
  • 13 Carrizzo A, Lenzi P, Procaccini C. , et al. Pentraxin 3 induces vascular endothelial dysfunction through a P-selectin/matrix metalloproteinase-1 pathway. Circulation 2015; 131 (17) 1495-1505 , discussion 1505
  • 14 Sprague AH, Khalil RA. Inflammatory cytokines in vascular dysfunction and vascular disease. Biochem Pharmacol 2009; 78 (06) 539-552
  • 15 Witasp A, Rydén M, Carrero JJ. , et al. Elevated circulating levels and tissue expression of pentraxin 3 in uremia: a reflection of endothelial dysfunction. PLoS One 2013; 8 (05) e63493
  • 16 Rajkovic I, Denes A, Allan SM, Pinteaux E. Emerging roles of the acute phase protein pentraxin-3 during central nervous system disorders. J Neuroimmunol 2016; 292: 27-33
  • 17 Ummenthum K, Peferoen LA, Finardi A. , et al. Pentraxin-3 is upregulated in the central nervous system during MS and EAE, but does not modulate experimental neurological disease. Eur J Immunol 2016; 46 (03) 701-711
  • 18 Garlanda C, Bottazzi B, Moalli F. , et al. Pentraxins and atherosclerosis: the role of PTX3. Curr Pharm Des 2011; 17 (01) 38-46
  • 19 Ogihara Y, Ogata S, Nomoto K. , et al. Transcriptional regulation by infliximab therapy in Kawasaki disease patients with immunoglobulin resistance. Pediatr Res 2014; 76 (03) 287-293
  • 20 Takahashi K, Oharaseki T, Yokouchi Y. Update on etio and immunopathogenesis of Kawasaki disease. Curr Opin Rheumatol 2014; 26 (01) 31-36
  • 21 Snyder RD, Stovring J, Cushing AH, Davis LE, Hardy TL. Cerebral infarction in childhood bacterial meningitis. J Neurol Neurosurg Psychiatry 1981; 44 (07) 581-585
  • 22 Korematsu S, Uchiyama S, Miyahara H. , et al. The characterization of cerebrospinal fluid and serum cytokines in patients with Kawasaki disease. Pediatr Infect Dis J 2007; 26 (08) 750-753
  • 23 Terasawa K, Ichinose E, Matsuishi T, Kato H. Neurological complications in Kawasaki disease. Brain Dev 1983; 5 (04) 371-374