Case Report: Gastrointestinal Basidiobolomycosis with Multi-Organ Involvement Presented with Intussusception

Zahra Jafarpour Professor Alborzi Clinical Microbiology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran;

Search for other papers by Zahra Jafarpour in
Current site
Google Scholar
PubMed
Close
,
Gholamreza Pouladfar Professor Alborzi Clinical Microbiology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran;

Search for other papers by Gholamreza Pouladfar in
Current site
Google Scholar
PubMed
Close
,
Alireza Dehghan Radiology Department, Shiraz University of Medical Sciences, Shiraz, Iran;

Search for other papers by Alireza Dehghan in
Current site
Google Scholar
PubMed
Close
,
Mohammad Hossein Anbardar Pathology Department, Shiraz University of Medical Sciences, Shiraz, Iran;

Search for other papers by Mohammad Hossein Anbardar in
Current site
Google Scholar
PubMed
Close
, and
Hamid Reza Foroutan Department of Surgery, School of Medicine, Laparoscopy Research Center, Shiraz University of Medical Sciences, Shiraz, Iran

Search for other papers by Hamid Reza Foroutan in
Current site
Google Scholar
PubMed
Close
Restricted access

ABSTRACT.

Gastrointestinal basidiobolomycosis (GIB) is a rare, life-threatening fungal infection affecting immunocompetent individuals in tropical and subtropical regions. A diverse presentation of GIB has been reported, but no report has yet been published on intussusception. We describe a 23-month-old immunocompetent boy from a subtropical area in Iran who presented with intussusception. Prolonged fever, an abdominal mass, hepatomegaly, high erythrocyte sedimentation rate, and peripheral eosinophilia strongly suggested GIB. Accordingly, GIB was diagnosed based on the characteristic histopathology (the Splendore-Hoeppli phenomenon) detected in a liver sample taken via biopsy. Exploratory laparotomy showed several organs, including the colon, gall bladder, liver, and abdominal wall, were involved. Antifungal therapy with trimethoprim/sulfamethoxazole, liposomal amphotericin B, a saturated solution of potassium iodide, and surgical resection of involved tissues were used with improved outcome. The presence of non-septate fungal hyphal elements in the colonic mucosa led to the thickening of the bowel wall, leading to secondary intussusception.

Author Notes

Address correspondence to Gholamreza Pouladfar, Professor Alborzi Clinical Microbiology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran. E-mail: pouladfar_ghr@hotmail.com

Authors’ addresses: Zahra Jafarpour and Gholamreza Pouladfar, Professor Alborzi Clinical Microbiology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran, E-mails: zjafarpoor54@yahoo.com and pouladfar_ghr@hotmail.com. Alireza Dehghan, Radiology Department, Shiraz University of Medical Sciences, Shiraz, Iran, E-mail: alirezadehghan@sums.ac.ir. Mohammad Hossein Anbardar, Pathology Department, Shiraz University of Medical Sciences, Shiraz, Iran, E-mail: anbardarm@sums.ac.ir. Hamid Reza Foroutan, Department of Surgery, School of Medicine, Laparoscopy Research Center, Shiraz University of Medical Sciences, Shiraz, Iran, E-mail: forotanh@yahoo.com.

  • 1.

    Mendoza L, Vilela R, Voelz K, Ibrahim AS, Voigt K, Lee SC, 2015. Human fungal pathogens of Mucorales and Entomophthorales. Cold Spring Harb Perspect Med 5: a019562.

  • 2.

    Pezzani MD et al., 2019. Gastrointestinal basidiobolomycosis: an emerging mycosis difficult to diagnose but curable: case report and review of the literature. Travel Med Infect Dis 31: 101378.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3.

    Ghadimi Moghadam A, Alborzi A, Eilami O, Bediee P, Pouladfar GHR, 2015. Zygomycosis in immunocompetent children in Iran: case series and review. Scholars J Appl Med Sci 3: 450–454.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4.

    Mohammadi R, Ansari Chaharsoghi M, Khorvash F, Kaleidari B, Sanei MH, Ahangarkani F, Abtahian Z, Meis JF, Badali H, 2019. An unusual case of gastrointestinal basidiobolomycosis mimicking colon cancer: literature and review. J Mycol Med 29: 75–79.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5.

    Zekavat OR, Abdolkarimi B, Pouladfar G, Fathpour G, Mokhtari M, Shakibazad N, 2017. Colonic basidiobolomycosis with liver involvement masquerading as gastrointestinal lymphoma: a case report and literature review. Rev Soc Bras Med Trop 50: 712–714.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6.

    Abdollahi A, Sadeghpour A, 2018. Gastrointestinal basidiobolomycosis: an unusual fungal disease. Acad J Surg 5: 21–23.

  • 7.

    Mousavi MR, Pouladfar Gh R, Taherifard E, Badiee P, Anbardar MH, 2020. An infant with acute bloody diarrhea and gastrointestinal basidiobolomycosis: an unusual presentation of a rare disease. Trop Pediatr fmaa039. Available at: https://doi.org/10.1093/tropej/fmaa039.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8.

    Balkhair A, Al Wahaibi A, Al-Qadhi H, Al-Harthy A, Lakhtakia R, Rasool W, Ibrahim S, 2019. Gastrointestinal basidiobolomycosis: beware of the great masquerade: a case report. IDCases 18: e00614.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9.

    Al-Juaid A, Al-Rezqi A, Almansouri W, Maghrabi H, Satti M, 2017. Pediatric gastrointestinal basidiobolomycosis: case report and review of literature. Saudi J Med Med Sci 5: 167–171.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10.

    Sanaei Dashti A, Nasimfar A, Hosseini Khorami H, Pouladfar G, Kadivar MR, Geramizadeh B, Khalifeh M, 2018. Gastro-intestinal basidiobolomycosis in a 2-year-old boy: dramatic response to potassium iodide. Paediatr Int Child Health 38: 150–153.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11.

    Shaikh N, Hussain KA, Petraitiene R, Schuetz AN, Walsh TJ, 2016. Entomophthoramycosis: a neglected tropical mycosis. Clin Microbiol Infect 22: 688–694.

  • 12.

    Esmaeili-Dooki MR, Moslemi L, Hadipoor A, Osia S, Fatemi SA, 2016. Pediatric intussusception in northern Iran: comparison of recurrent with non-recurrent cases. Iran J Pediatr 26: e3898.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13.

    Navarro O, Daneman A, 2004. Intussusception: part 3: diagnosis and management of those with an identifiable or predisposing cause and those that reduce spontaneously. Pediatr Radiol 34: 305–312.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14.

    Geramizadeh B, Heidari M, Shekarkhar G, 2015. Gastrointestinal basidiobolomycosis, a rare and under-diagnosed fungal infection in immunocompetent hosts: a review article. Iran J Med Sci 40: 90–97.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15.

    Darre T, Saka B, Mouhari-Toure A, Djiwa T, Pitche P, Napo-Koura G, 2018. Basidiobolomycosis in Togo: clinicopathological study of a series of 12 presumed cases. BMC Res Notes 11: 667.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16.

    Rodig SJ, Dorfman DM, 2001. Splendore-Hoeppli phenomenon. Arch Pathol Lab Med 125: 1515–1516.

  • 17.

    Vilela R, Mendoza L, 2018. Human pathogenic Entomophthorales. Clin Microbiol Rev 31: e00014–18.

  • 18.

    Gaastra W, Lipman LJ, De Cock AW, Exel TK, Pegge RB, Scheurwater J, Vilela R, Mendoza L, 2010. Pythium insidiosum: an overview. Vet Microbiol 146: 1–16.

  • 19.

    Krajaejun T et al., 2006. Clinical and epidemiological analyses of human pythiosis in Thailand. Clin Infect Dis 43: 569–576.

Past two years Past Year Past 30 Days
Abstract Views 1606 523 93
Full Text Views 254 120 0
PDF Downloads 108 10 0
 
 
 
 
Affiliate Membership Banner
 
 
Research for Health Information Banner
 
 
CLOCKSS
 
 
 
Society Publishers Coalition Banner
Save