Mini-review
Diagnosis of gastrointestinal basidiobolomycosis: a mini-review
Mortada H. El-Shabrawi,
1
Naglaa Mohamed Kamal,
2
Kerstin Kaerger
3
and Kerstin Voigt
4,5
1
Faculty of Medicine, Cairo University, Cairo, Egypt,
2
Faculty of Medicine, Cairo University, Cairo, Egypt,
3
Faculty of Biology and Pharmacy, Institute of
Microbiology, University of Jena, Jena, Germany,
4
Leibniz Institute for Natural Product Research & Infection Biology, Jena, Germany and
5
Faculty of
Biology and Pharmacy, Institute of Microbiology, University of Jena, Jena, Germany
Summary Basidiobolus ranarum (Entomophthoromycotina) very rarely affects the gastrointesti-
nal (GI) tract. To date, reported paediatric GI basidiobolomycosis cases are 27 world-
wide; 19 from Saudi Arabia and 8 from other parts of the world. Often these cases
present a diagnostic dilemma, are prone to misdiagnosis and lack of disease confir-
mation by proper molecular methodologies. The fungal mass removed by surgery is
usually sent for conciliar histopathology, isolation by fungal cultures and final
molecular testing for basidiobolomycosis. The incidence of basidiobolomycoses, their
predisposing factors and the molecular diagnosis of the fungus causing the disease in
combination with a phylogenetic framework are reviewed.
Key words: 18S rRNA, gastrointestinal basidiobolomycosis, identification, molecular typing, Splendore–Hoeppli
phenomenon.
Basidiobolomycosis: main characteristics of a
rare chronic subcutaneous disease
Basidiobolomycosis is an unusual, rare fungal skin
infection causing chronic subcutaneous zygomyco-
sis.
1,2
It is caused by Basidiobolus ranarum (Entomopht-
horomycotina)
3,4
with human disease concentrated in
tropical and subtropical regions.
Extracutaneous involvement is extremely rare
5
with
gastrointestinal (GI) involvement being exceedingly
rare
6–10
; with only 66 adult and 27 paediatric cases
reported worldwide. Most adult cases, 19 patients, were
from the United States of whom 17 [89%] were from
Arizona
11
; whereas 14 patients were from Iran,
11
12
patients from Iraq,
12
11 from the Kingdom of Saudi
Arabia (KSA)
11
and 4 from Brazil.
11
The remaining six
patients were one from each of Nigeria, India,
Bangladesh, Italy, Netherlands and one with unreported
country of origin.
11
The 27 reported paediatric patients are summarised
in Table 1,
12–24
where 19 patients are from KSA, 3 from
Iran, 2 from Iraq, 2 from Brazil and 1 from Nigeria.
All reported paediatric gastrointestinal basidiobol-
omycosis (GIB) cases were males with no significant
medical history or apparent predisposing factor(s), age
ranged between 1.5–13 years, and presented with
fever and abdominal pain as their main symptoms.
Leucocytosis with marked eosinophilia, high erythro-
cyte sedimentation rate (ESR) and C-reactive protein
(CRP) were found in all cases.
10,25
Abdominal exami-
nation revealed intra-abdominal masses in all cases
and were confirmed by abdominal ultrasonography
and computed tomography.
Almost all cases were misdiagnosed as other chronic
granulomatous diseases or malignancies.
25
Some
examples are: (i) AlJarie series,
16
where two patients
were misdiagnosed as appendicitis with appendicular
mass, two as abdominal tuberculosis and two as lym-
phomas, (ii) Khan and his colleagues’ patient was also
misdiagnosed as intestinal tuberculosis,
9
(iii) Fahimzad
and his colleagues,
17
initially didn’t achieve diagnosis
and titled their patient as inflammatory granuloma
with undetermined aetiology, (iv) Nguyen’s patient
was misdiagnosed as Crohn’s disease,
2
etc.
Correspondence: Naglaa Mohamed Kamal, MD, Associate Professor of
Pediatrics and Pediatric Hepatology, Faculty of Medicine, Cairo University,
Cairo, Egypt.
Tel.: +00201141991114. Fax: +20237619012.
E-mail: nagla.kamal@kasralainy.edu.eg or nagla.kamal@medicine.cu.edu.eg
Submitted for publication 21 December 2013
Revised 18 June 2014
Accepted for publication 18 June 2014
© 2014 Blackwell Verlag GmbH
Mycoses, 2014, 57 (Suppl. 3), 138–143 doi:10.1111/myc.12231
mycoses
Diagnosis,Therapy and Prophylaxis of Fungal Diseases