Analysis of the Clinical Characteristics and Follow-up Study of Children with Cutaneous Polyarteritis Nodosa

Author(s): Qiang He, Jing Shu*, Fang Chen, Xiao-Fang Zhen.

Journal Name: Current Neurovascular Research

Volume 16 , Issue 3 , 2019

Become EABM
Become Reviewer

Abstract:

Objective: This study aims to analyze the clinical characteristics, treatment and prognosis of children with cutaneous polyarteritis nodosa (CPAN), in order to improve the understanding of this disease.

Methods: Data of 14 children with CPAN, who were hospitalized in the Beijing Children's Hospital of Capital Medical University from January 2006 to December 2016, were collected. The clinical characteristics of all patients were summarized, the antistreptolysin-O (ASO)-positive and ASO-negative groups were compared, and the follow-up results were analyzed. X2-test, Fisher’s exact probability test, t-test and Mann-Whitney test were used for statistical analysis.

Results: Among these 14 CPAN patients, nodular rash was the most common manifestation (14/14). The ASO-positive group had more nodules in the lower limbs and the ASO-negative group appeared more in the upper limbs, which were statistically significant (p<0.05). ASOpositive children were more likely to have joint symptoms (P<0.05), and were more prone to elevated white blood cells (P<0.05). Follow-ups were performed on nine patients, and the prognoses were all good. The occurrence of systemic polyarteritis nodosa was not observed.

Conclusion: The main clinical manifestation of children with CPAN is skin nodules, which rarely affects the internal organs. Streptococcal infection is often the main cause. Anti-infection treatment should be simultaneously considered.

Keywords: Cutaneous polyarteritis nodosa (CPAN), children, group A β-hemolytic streptococcus, mutation, infection, aneurysm.

[1]
Balow JE. Renal vasculitis. Kidney Int 1985; 27(6): 954-64.
[2]
Jennette JC, Falk RJ, Bacon PA, et al. 2012 revised International Chapel Hill Consensus Conference Nomenclature of Vasculitides. Arthritis Rheum 2013; 65(1): 1-11.
[3]
Sato O, Cohn DL. Polyarteritis and microscopic polyangiitis. In: Rheumatology, Klippel JH, Dieppe PA (Eds.), Mosby, St Louis: Mosby 2003 pp. 1290-99.
[4]
Chen KR. Cutaneous polyarteritis nodosa: A clinical and histopathological study of 20 cases. J Dermatol 1989; 16(6): 429-42.
[5]
Ozen S, Ruperto N, Dillon MJ, et al. EULAR/PReS endorsed consensus criteria for the classification of childhood vasculitides. Ann Rheum Dis 2006; 65(7): 936-41.
[6]
Tania M, Santiago G, Zavialov A, et al. Dermatologic features of ADA2 deficiency in cutaneous polyarteritis nodosa. JAMA Dermatol 2015; 151(11): 1230-4.
[7]
Meyts I, Aksentijevich I. Deficiency of adenosine deaminase 2 (DADA2): Updates on the phenotype, genetics, pathogenesis, and treatment. J Clin Immunol 2018; 38: 569-78.
[8]
Hubert de Boysson. Loïc Guillevin. Polyarteritis Nodosa Neurologic Manifestations. Neurol Clin 2019; 37(2): 345-57.
[9]
Lindberg K. Ein beitrag zur kenntnis der periarteritis nodosa. Acta Med Scand 1931; 76: 183.
[10]
Merlina E, Mouy R, Pereira B, et al. Long-term outcome of children with pediatric-onset cutaneous and visceral polyarteritis nodosa. Joint Bone Spine 2015; 82(4): 251-7.
[11]
Bansal N-K, Houghton KM. Cutaneous polyarteritis nodosa in childhood: A case report and review of the literature. Arthritis (Egypt) 2010; 2010: 687547.
[12]
Daoud MS, Hutton KP, Gibson LE. Cutaneous periarteritis nodosa: A clinicopathological study of 79 cases. Br J Dermatol 1997; 136(5): 706-13.
[13]
Criado PR, Marques RF, Morita TCAB, Carvalho JF. Epidemiological, clinical and laboratory profiles of cutaneous polyarteritis nodosa patients: Report of 22 cases and literature review. Autoimmun Rev 2016; 15(6): 558-63.
[14]
Fathalla BM, Miller L, Brady S, Schaller JG. Cutaneous polyarteritis nodosa in children. J Am Acad Dermatol 2005; 53(4): 724-8.
[15]
Nakamura T, Kanazawa N, Ikeda T, et al. Cutaneous polyarteritis nodosa: Revisiting its definition and diagnostic criteria. Arch Dermatol Res 2009; 301(1): 117-21.
[16]
Odhav A, Odhav C, Dayal NA. Rare adverse effect of treatment with minocycline. Minocycline-induced cutaneous polyarteritis nodosa. JAMA Pediatr 2014; 168(3): 287-8.
[17]
Tschetter AJ, Liu V, Wanat KA. Cutaneous polyarteritis nodosa presenting as a solitary blue toe. J Am Acad Dermatol 2014; 71(3): e95-7.
[18]
Till SH, Amos RS. Long-term follow-up of juvenile-onset cutane- ous polyarteritis nodosa associated with streptococcal infection. Br J Rheumatol 1997; 36(8): 909-11.
[19]
Gibson KM, Morishita KA, Dancey P, et al. PedVas investigators network. Identification of novel Adenosine Deaminase 2 gene variants and varied clinical phenotype in pediatric vasculitis. Arthritis Rheumatol 2019. [Epub ahead of print].
[20]
Sriskandan S, Faulkner L, Hopkins P. Streptococcus pyogenes: Insight into the function of the streptococcal superantigens. Int J Biochem Cell Biol 2007; 39(1): 12-9.
[21]
Diaz-Perez JL, Schroeter AL, Winkelmann RK. Cutaneous periarteritis nodosa: Immunofluorescence studies. Arch Dermatol 1980; 116(1): 56-8.
[22]
Kawakami T, Yamazaki M, Mizoguchi M, Soma Y. High titer of antiphosphatidylserine-prothrombin complex antibodies in patients with cutaneous polyarteritis nodosa. Arthritis Rheum 2007; Dec 15 57(8): 1507-13.
[23]
Kawakami T, Soma Y. Correlation of livedo racemosa, cutaneous inflammatory plaques, and antiphospholipid antibodies in patients with cutaneous polyarteritis nodosa. Medicine (Baltimore) 2011; 90(2): 119-24.
[24]
Ozen S, Pistorio A, Iusan SM, et al. Paediatric Rheumatology International Trials Organisation (PRINTO). EULAR/PRINTO/PRES criteria for Henoch-Schonlein purpura, childhood plyarteritis nodosa, childhood Wegener granulomatosis and childhood Takyasu arteritis: Ankara 2008. Part II: Final classification criteria. Ann Rheum Dis 2010; 69(5): 798-806.
[25]
Kawakami T, Okano T, Takeuchi S, Kimura S, Soma Y. Complete resolution of refractory cutaneous arteritis by intravenous cyclophosphamide pulse therapy. Int J Dermatol 2015; 54(8): e323-5.


Rights & PermissionsPrintExport Cite as

Article Details

VOLUME: 16
ISSUE: 3
Year: 2019
Page: [208 - 214]
Pages: 7
DOI: 10.2174/1567202616666190618112705
Price: $58

Article Metrics

PDF: 33
HTML: 2