Indian Journal of Medical Microbiology IAMM  | About us |  Subscription |  e-Alerts  | Feedback |  Login   
  Print this page Email this page   Small font sizeDefault font sizeIncrease font size
 Home | Ahead of Print | Current Issue | Archives | Search | Instructions  
Users Online: 1288 Official Publication of Indian Association of Medical Microbiologists 
  Search
 
  
 ~  Similar in PUBMED
 ~  Search Pubmed for
 ~  Search in Google Scholar for
 ~Related articles
 ~  Article in PDF (735 KB)
 ~  Citation Manager
 ~  Access Statistics
 ~  Reader Comments
 ~  Email Alert *
 ~  Add to My List *
* Registration required (free)  

 
 ~  Abstract
 ~  Introduction
 ~  Case Report
 ~  Discussion
 ~  References
 ~  Article Figures

 Article Access Statistics
    Viewed9828    
    Printed121    
    Emailed1    
    PDF Downloaded138    
    Comments [Add]    
    Cited by others 3    

Recommend this journal

 


 
  Table of Contents  
CASE REPORT
Year : 2011  |  Volume : 29  |  Issue : 2  |  Page : 186-188
 

Intestinal obstruction due to Basidiobolus ranarum: An unusual case


Perunthalaivar Kamaraj Medical College, Puducherry - 605 009, India

Date of Submission25-Mar-2010
Date of Acceptance23-Mar-2011
Date of Web Publication2-Jun-2011

Correspondence Address:
Angeline Neetha Radjou
Perunthalaivar Kamaraj Medical College, Puducherry - 605 009
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0255-0857.81790

Rights and Permissions

 ~ Abstract 

A case of intestinal obstruction caused by extensive soft tissue fungal infection of the perineum due to Basidiobolus ranarum is presented here. There was excellent response to antifungal treatment. A literature search revealed the case report of intestinal obstruction due to intrinsic mucosal involvement by the fungus, but extensive soft tissue involvement of the perineum resulting in extraneous obstruction to the rectum, has not been reported so far.


Keywords: Intestinal obstruction, itraconazole, soft tissue infection


How to cite this article:
Radjou AN, Rajesh N G. Intestinal obstruction due to Basidiobolus ranarum: An unusual case. Indian J Med Microbiol 2011;29:186-8

How to cite this URL:
Radjou AN, Rajesh N G. Intestinal obstruction due to Basidiobolus ranarum: An unusual case. Indian J Med Microbiol [serial online] 2011 [cited 2019 Oct 13];29:186-8. Available from: http://www.ijmm.org/text.asp?2011/29/2/186/81790



 ~ Introduction Top


Basidiobolus ranarum infection is prevalent in tropical and subtropical regions. A few cases have been reported from Asia. In India, it has been reported from Pondicherry. It usually affects children, and is more prevalent in male gender, usually affecting the soft tissues of the limbs. Here, we present an adult female patient with extensive soft tissue involvement of the perineum who had presented with features of acute large intestinal obstruction.


 ~ Case Report Top


A 24-year-old female patient had presented to our hospital with features of acute intestinal obstruction. She had normal vaginal delivery 2 months ago and presented with episode of intestinal obstruction at another hospital a week earlier, which subsided with conservative treatment. She had presented with the second attack of intestinal obstruction to our hospital. The abdomen was gaseously distended, with visible intestinal peristalsis and hyperactive bowel sounds. She did not have any symptoms referable to any other region. During the per rectal examination as part of the abdominal examination for intestinal obstruction, the perineum as such appeared woody. A detailed examination of the perineum revealed areas of thickening and nodularity extending up to the buttocks and part of the labia, including the episiotomy site. The lesions were non-tender, the edges were vaguely demarcated and there were no sinuses [Figure 1]. The rectal mucosa felt normal on palpation and on visualization. However, the pararectal tissues appeared indurated. A vaginal examination revealed normal vaginal mucosa, but the paravaginal tissues appeared indurated. The regional lymph nodes were not significant. There were no similar lesions elsewhere in the body. On clinical grounds, it was obvious that there was a soft tissue pathology surrounding the rectum and vagina with sparing of mucosal structures. An X-ray of the abdomen revealed features of intestinal obstruction [Figure 2] for which she was treated conservatively and she recovered in 48 h. Ultrasound of the pelvis revealed circumferential wall thickening of rectum of about 1.5 cm. A colonoscopy now revealed normal rectal mucosa. Regarding the primary problem, a differential diagnosis of desmoid/fungal infection was entertained. A deep biopsy was performed and sent for histopathology, fungal culture and tuberculosis culture. Serology for human immunodeficiency virus, hepatitis B virus and hepatitis C virus was negative. She was not a diabetic and X-ray chest was normal. Complete blood count was normal. The erythrocyte sedimentation rate was 60 mm.
Figure 1: Induration and multiple nodules in the perineum and labia (biopsy site is seen)

Click here to view
Figure 2: Dilated bowel loops of intestinal obstruction

Click here to view


Histopathology revealed normal epidermis. Dermis revealed a granuloma with eosinophilic infiltrates with thin-walled fungal hyphae and Splendore Hoeppli phenomenon [Figure 3]. Culture on Sabouraud dextrose agar with gentamicin and incubated at 37°C on the 5 th day of culture revealed heavy growth densely furrowed with a waxy appearance [Figure 4]. Lactophenol cotton blue staining revealed thick-walled zygospores with protruberance (copulatory beak), typical of Basidiobolus ranarum.
Figure 3: Four months after treatment with itraconazole shows complete healing of the lesions

Click here to view
Figure 4: Biopsy revealed broad non-septate hyphae within a foreign body giant cell in the dermis. Arrow shows Splendore Hoeppli phenomenon (H&E, ×400)

Click here to view


The tissue sent for TB culture was negative for growth.

The patient was started on itraconazole 100 mg bd for 4 months. There was complete resolution of the lesions [Figure 1] with treatment and she is on regular follow-up. Per rectal examination and per vaginal examination did not reveal any induration, and she has no further attacks of intestinal obstruction


 ~ Discussion Top


Basidiobolus ranarum is a filamentous fungi belonging to the class Zygomycetes, order Entomopthorales, family Basidiobolaceae and genus Basidiobolus. It is commonly found in subtropical and tropical regions. Zygomycetes are primitive fast growing, terrestrial, largely saprophytic fungi with a cosmopolitan distribution in Africa and South America. The causative organism is present in soil, decaying organic matter and in the gastrointestinal tract of toads, reptiles and fish. The most common presentation is a soft tissue-indolent infection of the limbs causing hard, non-ulcerating lesions in the subcutaneous tissues and, unlike other fungi, it affects immunocompetent hosts. The organism gains entry through a breach in the skin and usually affects children and males. [1] Our patient is an adult female. We postulate that the organism gained entry through an episiotomy site in this patient, as the symptomatology started after the delivery. Cases from south India have been reported, especially from Pondicherry. [2] A few cases of direct involvement of the intestine have also been reported. [3],[4] A direct involvement of the gastrointestinal tract was ruled out in this patient as the rectal mucosa was normal on colonoscopy. Extension into the retroperitoneum from the buttock but not causing intestinal obstruction had been reported. [5],[6] However, soft tissue involvement of the perineum resulting in extramural obstruction to the rectum had not been reported so far.

A meticulous examination of the perineum helps clinch the problem as the patient had no symptoms referable to the perineum, even on retrospective history.

Histopathology revealed a fungal granuloma. The Splendor Hoeppli phenomenon (radiating intensely eosinophilic granular material surrounding the fungal elements) is characteristic but may not always be present. Definitive diagnosis can be made only on culture, for which Sabouraud dextrose agar is usually adequate and shows characteristic waxy growth with furrows, and special staining with lactophenol cotton blue would show the characteristic protuberances (beaked appearance). Antifungal susceptibility can be carried out.

A serological study can be performed using the immune diffusion method. [7]

The treatment of choice is potassium iodide, but itraconazole is also being used. Our patient was breast feeding and hence we started her on itraconazole, due to which the lesions healed completely in 4 months. [8] Surgery for just soft tissue involvement is not an option. Surgery has been performed only when there had been mucosal involvement of the colon/rectum as treatment for obstructing lesion and for settling diagnostic confusion with inflammatory bowel disease. [9]

The disease is usually localized, but can be extensive; however, fatal disseminated disease in an immunocompetent host has been reported, where angioinvasion and necrosis is typically seen. [10]

 
 ~ References Top

1.Gugnani AC. A review of zygomycosis due to Basidiobolus ranarum. Eur J Epidemiol 1999;15:923-9.  Back to cited text no. 1
    
2.Sujatha S, Sheela Devi C, Khyreim AB, Parija SC, Thappa DM. Subcutaneous zygomycosis caused by Basidiobolus ranarum: A case report. Indian J Med Microbiol 2003;21:205-6.  Back to cited text no. 2
[PUBMED]  Medknow Journal  
3.Zavasky DM, Samcoitz W, Loftus T, Segal H, Carroll K. Gastrointestinal zygomycotic infection caused by Basidiobolus ranarum: Case report and review. Clin Infect Dis 1999;28:1244-8.  Back to cited text no. 3
    
4.Khan ZU, Khoursheed M, Makar R, Al-Waheeb S, Al-Bader I, Al-Muzaini A, et al. Basidiobolus ranarum as an etiologic agent of gastrointestinal zygomycosis. J Clin Microbiol 2001;39:2360-3.   Back to cited text no. 4
[PUBMED]  [FULLTEXT]  
5.Vianna LM, de Lacerda MV, de Moraes MA. Case report of subcutaneous entomophthoromycosis with retroperitoneal invasion. Rev Soc Bras Med Trop 2005;38:348-50.   Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.Van den Berk EL Guido, Noorduyn L Arnold, van Ketel J Ruud, van Leeuwen Jeannouel, Bemelman A Willem and Prins M Jan Case report. Available from: http://www.biomedcentral.com/content/download/yml/1471-2334-6-140.xml.   Back to cited text no. 6
    
7.KauffmanL, Mendoza L Standard PG. Immunodiffusion test for srodiagnosing subcutaneous zygomycosis. J Clin Microbiol 1990;28:1887-90.  Back to cited text no. 7
    
8.Mathew R, Kumaravel S, Kuruvilla S, Varghese RG, Shashikala, Srinivasan S, et al. Successful treatment of extensive basidiobolomycosis with oral itraconazole in a child. Int J Dermatol 2005;44:572-5.  Back to cited text no. 8
[PUBMED]  [FULLTEXT]  
9.Lyon GM, Smilack JD, Komatsu KK, Pasha TM, Leighton JA, Guarner J, et al. Gastrointestinal Basidiobolomycosis in Arizona: Clinical and epidemiological characteristics and review of literature. Clin Infect Dis 2001;32:1448-5.  Back to cited text no. 9
[PUBMED]  [FULLTEXT]  
10.Bigliazzi C, Poletti V, Dell′Amore D, Saragoni L, Colby TV. Disseminated basidiobolomycosis in an immunocompetent woman. Clin Microbiol 2004;42:1367-9.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

This article has been cited by
1 Gastrointestinal basidiobolomycosis treated with posaconazole
Stacey R. Rose,Mark D. Lindsley,Steven F. Hurst,Christopher D. Paddock,Thara Damodaran,John Bennett
Medical Mycology Case Reports. 2013; 2: 11
[Pubmed] | [DOI]
2 The emerging epidemiology of mould infections in developing countries
Chakrabarti, A., Singh, R.
Current Opinion in Infectious Diseases. 2011; 24(6): 521-526
[Pubmed]
3 The emerging epidemiology of mould infections in developing countries
Arunaloke Chakrabarti,Rachna Singh
Current Opinion in Infectious Diseases. 2011; 24(6): 521
[Pubmed] | [DOI]



 

Top
Print this article  Email this article
 

    

2004 - Indian Journal of Medical Microbiology
Published by Wolters Kluwer - Medknow

Online since April 2001, new site since 1st August '04