Vasculitis mimics caused by Pythium infection and literatures review (Eng)

Main Article Content

Angkana Norasetthada

Abstract

While early diagnosis of vasculitis is important to induce remission and prevent organ damage, an incorrect diagnosis can result in harmful consequences from missing the underlying condition and exposure to immunosuppressive therapy. Evaluation of vasculitis should include consideration of its mimics. Important mimic categories include infection, vasculopathy, non-inflammatory conditions like atherosclerosis, thrombotic states, calciphylaxis and rare neoplasms.Human pythiosis is a rare life-threatening disease caused by Pythium insidiosum, a fungus-like organism that belongs to the Kingdom Straminiphila.Infected patients were mostly reported from Thailand and usually had an agricultural background. Clinical presentations are documented, cutaneous/subcutaneous, ocular, vascular, and disseminated pythiosis. The majority of the reported patients with vascular pythiosis have lower limb involvement.Known risk factors for vascular pythiosis include thalassemia, hemoglobinopathy, paroxysmal nocturnal hemoglobinuria, aplastic anemia, and leukemia. Furthermore, the rarity of the disease has led to underrecognition, under-diagnosis, and delays in diagnosis, and this has contributed to the occurrence of advanced disease, which affects survival. I, herein, report a case of diabetes mellitus and alcoholism, 31 year-old male presented with ischemic leg ulcers, which skin biopsy revealed leukocytoclastic vasculitis and CT- angiogram suspected vasculitis, and then P. insidiosum has been isolated from vascular sites, and relevant literature reviews.

Article Details

How to Cite
1.
Norasetthada A. Vasculitis mimics caused by Pythium infection and literatures review (Eng). Thai J Rheu [internet]. 2025 Jun. 6 [cited 2025 Jun. 24];2(2):27-36. available from: https://he04.tci-thaijo.org/index.php/tjr/article/view/3237
Section
Case Report

References

Jennette JC, Falk RJ, Bacon PA et al. 2012 revised International Chapel Hill Consensus Conference Nomenclature of Vasculitides. Arthritis Rheum 2013;65:1–11.

Bateman H, Rehman A, Valeriano-Marcet J. Vasculitislike syndromes. Curr Rheumatol Rep 2009;11:422–9.

Keser G, Aksu K. Diagnosis and differential diagnosis oflarge-vessel vasculitides. Rheumatol Int 2019;39:169–85.

Miloslavsky EM, Stone JH, Unizony SH. Challengingmimickers of primary systemic vasculitis. Rheum Dis Clin North Am 2015;41:141–60.

Molloy ES, Langford CA. Vasculitis mimics. Curr Opin Rheumatol 2008;20:29–34.

Jason M. Springer, Alexandra Villa-Forte. Vasculitis mimics and Other

Related Conditions. Rheum Dis Clin N Am. 2023;49:617–631.

Maningding E, Kermani TA. Mimics of vasculitis. Rheumatology. 2021;60:34–47.

Zarka F , Veillette C , Makhzoum JP. A Review of Primary Vasculitis Mimickers Based on the Chapel Hill Consensus Classification.Int J Rheumatol. 2020 Feb 18;2020:8392542.

http://doi: 10.1155/2020/8392542

L. Kaufman. Penicilliosis marneffei and pythiosis: emerging tropical diseases. Mycopathologia,1998;143:3-8.

Gaastra W., Lipman L.J., De Cock A.W., Exel T.K., Pegge R.B., Scheurwater J. Pythium insidiosum: an overview. Veterinary Microbiology. 2010;146:1–16.

Sathapatayavongs B, Leelachaikul P, Prachaktam R, Atichartakarn V, Sriphojanart S, Trairatvorakul P, et al. Human pythiosis associated with thalassemia hemoglobinopathy syndrome. Infect Dis. 1989;159:274–280.

Chitasombat MN, Larbcharoensub N, Chindomporn A, Krajaejun T. Clinicopathological features and outcomes of pythiosis. International journal of infectious diseases. 2018;71:33-41.

Imwidthaya P. Human pythiosis in Thailand. Postgrad Med J. 1994;70:558–560.

Keoprasom N, Chularojanamontri L, Chayakulkeeree M, Chaiprasert A, Wanachiwanawin W, Ruangsetakit C. Vascular pythiosis in a thalassemic pateints. presenting as bilateral leg ulcers. Medical mycology. 2013;2:25-28.

Thianprasit M. Fungal infection in Thailand. Jpn J Dermatol. 1986;96:1343-45.

Sudjaritruk T, Sirisanthana V. Successful treatment of a child with vascular pythiosis. BMJ Infectious Diseases 2011;11:33

http://www.biomedcentral.com/1471-2334/11/33

Chitasombat MN, Jongkhajornpong P, Lekhanont K, Krajaejun T. Recent update in diagnosis and treatment of human pythiosis. Peer J. 2020;8:e8555. https://doi.org/10.7717/peerj.8555

Reanpang T, Orrapin Saritphat, Orrapin Saranat, Arworn S, Kattipatanapong T, Srisuwan T, et al. Vascular Pythiosis of the Lower Extremity in Northern Thailand: Ten Years’ Experience.The International Journal of Lower Extremity Wounds. 2015;14(3):245–250.

Krajaejun T, Sathapatayavongs B, Pracharktam R, Nitiyanant P, Leelachaikul P, Wanachiwanawin W,et al. Clinical and epidemiological analyses of human pythiosis in Thailand. Clinical Infectious Diseases. 2006b;43(5):569–576.

Pan JH, Kerkar SP, P,Siegenthaler MP, Hughes M,. Pandalaia PK. A complicated case of vascular Pythium insidiosum infection treated with limb-sparing surgery Int J Surg Case Rep. 2014; 5(10): 677–680.)

Ghorishi A, Alayon A, Ghaddar T, Kandah M, Amundson PK. MR and CT angiography in the diagnosis of vasculitides. BJR. 2023 Sep 25;5(1):20220020.

http://doi: 10.1259/bjro.20220020

Küker W, Gaertner S, Nägele T, Dopfer C,Schöning M, Fiehler J, et al. Vessel Wall Contrast Enhancement: A Diagnostic Sign of Cerebral Vasculitis. Cerebrovasc Dis. 2008;26(1):23–29.

Torvorapanit P, Chuleerarux N, Plongla R, Worasilchai N, Manothummetha K, Thongkam A, et al. Clinical Outcomes of Radical Surgery and Antimicrobial Agents in Vascular Pythiosis: A Multicenter Prospective Study. J Fungi (Basel). 2021:7(2):114.

http://doi: 10.3390/jof7020114

Permpalung N., Worasilchai N., Plongla R., Upala S., Sanguankeo A., Paitoonpong L,et al. Treatment outcomes of surgery, antifungal therapy and immunotherapy in ocular and vascular human pythiosis: A retrospective study of 18 patients. J Antimicrob Chemother. Antimicrob. Chemother. 2015;70

http://doi: 10.1093/jac/dkv008.

Worasilchai N., Chindamporn A., Plongla R., Torvorapanit P., Manothummetha K., Chuleerarux N.,et al. In Vitro Susceptibility of Thai Pythium insidiosum Isolates to Antibacterial Agents. Antimicrob Agents Chemother. 2020 Mar 24;64(4):e02099-19. http://doi: 10.1128/AAC.02099-19

Susaengrat N, Torvorapanit P, Plongla R, Chuleerarux N., Manothummetha K., Tuangsirisup J,et al. Adjunctive antibacterial agents as a salvage therapy in relapsed vascular pythiosis patients. Int. J. Infect. Dis. 2019;88:27–30.

Sermsathanasawadi N, Praditsuktavorn B, Hongku K, Wongwanit C, Chinsakchai K, Ruangsetakit C, et al. Outcomes and factors influencing prognosis in patients with vascular pythiosis J Vasc Surg. 2016;64(2):411-417.

Yolanda H, Krajaejun.T. History and Perspective of Immunotherapy for Pythiosis. Vaccines 2021, 9(10), 1080; https://doi.org/10.3390/vaccines9101080

Permpalung N, Worasilchai N, Plongla R, Upala S, Sanguankeo A, Paitoonpong L, et al Treatment outcomes of surgery, antifungal therapy and immunotherapy in ocular and vascular human pythiosis: a retrospective study of 18 patients. Journal of Antimicrobial Chemotherapy. 2015;70(6):1885–1892.

Wanachiwanawin W, Mendoza L, Visuthisakchai S, Mutsikapan P,

Sathapatayavongs B, Chaiprasert A, et al. Efficacy of immunotherapy using antigens of Pythium insidiosum in the treatment of vascular pythiosis in humans. Vaccine. 2004 Sep 9;22(27-28):3613-21 http://doi: 10.1016/j.vaccine.2004.03.031

Worasilchai N, Permpalung N, Chongsathidkiet P, Leelahavanichkul A,Mendoza AL, Palaga T, et al. Monitoring Anti-Pythium insidiosum IgG Antibodies and (1→3 )-β-d-Glucan in Vascular Pythiosis. J Clin Microbiol. 2018;56(8): e00610-18.

https://doi.org/10.1128/jcm.00610-18