Abstract
BackgroundHuman pythiosis is an emerging and life-threatening infectious disease caused by Pythium insidiosum. It occurs primarily in tropical, subtropical and temperate areas of the world, including Thailand. The aim of this report is to present the first pediatric case of typical vascular pythiosis.Case PresentationA 10-year-old boy with underlying β-thalassemia presented with gangrenous ulcers and claudication of the right leg which were unresponsive to antibiotic therapy for 6 weeks. Computerized tomography angiography indicated chronic arterial occlusion involving the right distal external iliac artery and its branches. High-above-knee amputation was urgently done to remove infected arteries and tissues, and to stop disease progression. Antibody to P. insidiosum was detected in a serum sample by the immunoblot and the immunochromatography tests. Fungal culture followed by nucleic sequence analysis was positive for P. insidiosum in the resected iliac arterial tissue. Immunotherapeutic vaccine and antifungal agents were administered. The patient remained well and was discharged after 2 months hospitalization without recurrence of the disease. At the time of this communication he has been symptom-free for 2 years.ConclusionsThe child presented with the classical manifestations of vascular pythiosis as seen in adult cases. However, because pediatricians were unfamiliar with the disease, diagnosis and surgical treatment were delayed. Both early diagnosis and appropriate surgical and medical treatments are crucial for good prognosis.
Highlights
Human pythiosis is an emerging and life-threatening infectious disease caused by Pythium insidiosum
The child presented with the classical manifestations of vascular pythiosis as seen in adult cases
We described the first case of typical vascular pythiosis in a child in whom therapy was successful
Summary
We report a case of thalassemic boy with vascular pythiosis who was successfully treated by surgery together with P. insidiosum immunotherapeutic vaccine and antifungal therapy. In cases of vascular pythiosis, the fungal culture is usually negative because viable fungus is only present at the advancing edge of the diseased artery which is difficult for the surgeon to obtain [1,2,4,10]. PCR amplification and identification of the 18s rRNA gene of P. insidiosum are useful for organism identification in both the clinical specimens and culture specimens [9,16] Imaging studies, such as angiography and CTA, usually demonstrate an occlusion or aneurysm in medium-to-large-sized arteries of the lower extremities and trunk. Fungal culture was positive for P. insidiosum in the resected iliac arterial tissue His CTA study showed typical findings of vascular pythiosis. It has high rates of morbidity and mortality. A copy of the written consent is available for review by the Editor-in-Chief of this journal
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