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 ~  Abstract
 ~ Introduction
 ~ Case Report
 ~ Discussion
 ~ Conclusion
 ~  References
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  Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 36  |  Issue : 3  |  Page : 447-449
 

Cutaneous botryomycosis of the foot: A case report and review of literature


1 Department of Medicine Unit V, Christian Medical College, Vellore, Tamil Nadu, India
2 Department of General Pathology, Christian Medical College, Vellore, Tamil Nadu, India

Date of Web Publication14-Nov-2018

Correspondence Address:
Dr. Karthik Gunasekaran
Department of Medicine Unit V, Christian Medical College, Vellore - 632 004, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmm.IJMM_18_347

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 ~ Abstract 

Botryomycosis is a chronic bacterial infection that manifests clinically as tumours or plaques that are often ulcerated and have discharging sinuses draining small white-coloured granules. Therefore, they closely mimic mycetoma or other fungal infections. It is most commonly caused by Staphylococcus aureus. It can present as cutaneous or visceral disease. The cutaneous form can invade deep tissue leading to extensive destruction and disfigurement. A 31-year-old female presented with progressive swelling of her right foot over a period of 8 years. She had a disfigured right foot with multiple sinuses discharging pus. X-ray and magnetic resonance imaging of the foot showed invasion and destruction of the deep layers of the foot including the bone. Deep biopsy from the foot showed an abscess cavity with Gram-positive cocci within and bacterial culture grew S. aureus establishing the diagnosis of botryomycosis. Botryomycosis is a rare presentation of a common pathogen and needs to be considered while evaluating a chronic invasive subcutaneous infection.


Keywords: Botyromycosis, cutaneous, Staphylococcus aureus


How to cite this article:
John K, Gunasekaran K, Kodiatte TA, Iyyadurai R. Cutaneous botryomycosis of the foot: A case report and review of literature. Indian J Med Microbiol 2018;36:447-9

How to cite this URL:
John K, Gunasekaran K, Kodiatte TA, Iyyadurai R. Cutaneous botryomycosis of the foot: A case report and review of literature. Indian J Med Microbiol [serial online] 2018 [cited 2019 Aug 18];36:447-9. Available from: http://www.ijmm.org/text.asp?2018/36/3/447/245396



 ~ Introduction Top


Botryomycosis is a chronic suppurative infection caused by bacterial pathogens, most commonly by Staphylococcus aureus.[1] Skin is the most common site of infection; however, other visceral organs may also be involved. The infection can infiltrate deep into the tissues causing widespread destruction and disfigurement. The diagnosis of botryomycosis is made by tissue culture, and the treatment consists of debridement with prolonged course of antibiotics. Here, we describe a patient who presented to us with cutaneous botryomycosis of the foot.


 ~ Case Report Top


A 31-year-old female, without any previous comorbidities, allergies or addictions, presented with a history of swelling of the right foot with multiple discharging sinuses for the past 8 years. She gave a history of crush injury to the foot, 8 years ago. The wound had healed with the application of topical antibiotics. However, over the course of the next few months, the foot became enlarged and disfigured without any associated pain, redness or warmth. She also developed multiple sinuses from the foot which drained purulent fluid. She had been evaluated at hometown for these complaints before presenting to us and was prescribed multiple courses of antibiotic without permanent resolution of symptoms.

On examination, she was afebrile, with a blood pressure of 100/60 mmHg in both upper limbs. Her heart rate was 76 beats per minute, respiratory rate was 18 breaths per minute and arterial oxygen saturation on room air was 98%.

Her right foot was swollen and scars of healed sinuses were seen [Figure 1]a. There was active purulent discharge from multiple sinuses. The foot was not tender or warm and all distal pulses were palpable. There was a restriction of mobility of the ankle joint, where only 20 degrees of plantar and dorsiflexion were possible. Systemic examination was unremarkable.
Figure 1: (a) Right foot swollen with active purulent discharge from multiple sinuses. (b) X-ray of ankle and foot showing destruction and remodelling of the bones of the foot. (c) Magnetic resonance imaging of the foot showing ill-defined short inversion time inversion-recovery hyperintense and T1 hypointense areas with multiple areas of cortical breech and sinuses draining into the skin

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The differential diagnoses considered were actinomycosis, nocardiosis, eumycetoma, mycobacterial tuberculosis, non-tubercular mycobacteria and botryomycosis. Laboratory investigations in the form of haemogram and comprehensive biochemical profile were normal [Table 1]. Blood cultures were sterile.
Table 1: Laboratory parameters of the patient with cutaneous botryomycosis

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X-Ray of the ankle and foot showed destruction and remodelling of the bones of the foot [Figure 1]b. Magnetic resonance imaging (MRI) of the foot showed ill-defined short inversion time inversion-recovery hyperintense and T1 hypointense areas with multiple areas of cortical breech and sinuses draining into the skin [Figure 1]c. A deep biopsy of the foot was performed. It showed an abscess cavity filled with purulent material showing neutrophils, lymphocytes, plasma cells and histiocytes [Figure 2]a and [Figure 2]b. It contained variably sized, irregularly shaped basophilic granules. The granules were composed of colonies of cocci which were positive for Gram-stain [Figure 2]c and negative for Grocott's methenamine silver, Periodic acid–Shiff's and Ziehl-Neelsen stains. Splendore-Hoeppli phenomenon was noted. The culture of the biopsied tissue grew S. aureus. Hence, she was diagnosed to have cutaneous botryomycosis of the foot. She was initiated on oral cloxacillin and was kept on follow-up.
Figure 2: (a and b) Abscess cavity filled with purulent material showing neutrophils, lymphocytes, plasma cells, histiocytes and variably sized, irregularly shaped basophilic granules. (c) The granules composed of colonies of cocci which were positive for Gram-stain

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 ~ Discussion Top


Botryomycosis is a misnomer. In Greek, 'botryose' means 'a bunch of grapes' and 'mycosis' refers to 'fungi'. This is because the etiological agent was wrongly thought to be a fungus. In reality, botryomycosis is caused by a chronic inflammatory reaction to bacterial pathogens.[1] As it is an exceedingly rare disease, data on the incidence and prevalence of this disease are lacking. The understanding of its pathogenesis is also poor. It is hypothesised that a combination of a hypovirulent pathogen and immunocompromised host is responsible for this unusual disease.[2]

Although the most common cause of botryomycosis is S. aureus, other Gram-positive cocci (streptococci and coagulase-negative staphylococci), Gram-negative bacteria (Pseudomonas aeruginosa, Escherichia coli and Proteus, Serratia) and anaerobic bacteria (Peptostreptococci and Actinobacillus) can also cause botryomycosis.[3] Cutaneous botryomycosis is more common form and can present with non-healing ulcers, sinuses and fistulae, as was seen in our patient. The infection can invade deep structures without respecting tissue planes and can cause extensive destruction of tendons, muscles and bones. Visceral botryomycosis can involve practically any organ and is often wrongly diagnosed as an abscess or a tumour at first. The correct diagnosis is established after histopathological examination of the removed specimen.[4] Botryomycosis has been observed in unusual locations like the heart on autopsy.[5] The differential diagnosis of botryomycosis includes nocardia, Actinomycosis, Eumycetoma, Mycobacterium tuberculosis and non-tubercular mycobacteria. Rarely, Kaposi sarcoma can also present in a similar fashion.

The gold standard of diagnosis of botryomycosis is histopathology and culture. On traditional eosin-haematoxylin preparation, botryomycosis is characterised by a central focus of necrosis surrounded by a chronic inflammatory reaction containing histiocytes, epithelioid cells, multinucleated giant cells and fibrosis. There may be granules called 'Bollinger granules' which consist of the bacteria surrounded by an eosinophilic matrix with club-like projections. This appearance is called the Splendore-Hoeppli phenomenon. In addition to eosin-haematoxylin stain, Gram-stain can be used.[6] The growth of the bacterial pathogen on routine culture media will establish the diagnosis. Granules may be present within the pus draining from sinuses and can be examined under a microscope. The granules are usually white, although rarely, other colours have also been described.[7] In addition, radiographic studies such as X-ray, computed tomography scan or MRI can be used to assess the depth of invasion of the disease.

Treatment of botryomycosis depends on the clinical presentation and pathogen. Debridement is done for cutaneous disease when feasible; however, if this is not possible prolonged antibiotic therapy is used. Visceral disease or drug-resistant pathogens may need parenteral therapy. Usually, therapy is continued till complete clinical resolution.[8]


 ~ Conclusion Top


Botryomycosis is an uncommon disease characterised by chronic suppurative inflammatory response to bacterial pathogens and is most commonly caused by S. aureus. It can present as cutaneous or visceral disease. The cutaneous form can invade deep tissue leading to extensive destruction and disfigurement.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initial will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 ~ References Top

1.
Winslow DJ. Botryomycosis. Am J Pathol 1959;35:153-67.  Back to cited text no. 1
    
2.
Brunken RC, Lichon-Chao N, van der Broek H. Immunologic abnormalities in botryomycosis. A case report with review of the literature. J Am Acad Dermatol 1983;9:428-34.  Back to cited text no. 2
    
3.
Chintaginjala A, Harshavardhan K, Kumar AS. Cutaneous botryomycosis: A Rare case report. Indian J Dermatol 2016;61:126.  Back to cited text no. 3
    
4.
Vasishta RK, Gupta N, Kakkar N. Botryomycosis-a series of six integumentary or visceral cases from India. Ann Trop Med Parasitol 2004;98:623-9.  Back to cited text no. 4
    
5.
Gupta K, Das A, Radotra BD, Bhalla A. Cardiac botryomycosis: An autopsy report. J Clin Pathol 2008;61:972-4.  Back to cited text no. 5
    
6.
Neafie RC, Marty AM. Unusual infections in humans. Clin Microbiol Rev 1993;6:34-56.  Back to cited text no. 6
    
7.
Katkar V, Mohammad F, Raut S, Amir R. Red grain botryomycosis due to staphylococcus aureus-a novel case report. Indian J Med Microbiol 2009;27:370-2.  Back to cited text no. 7
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8.
Kadyan RS. Botryomycosis. Indian J Dermatol Venereol Leprol 1996;62:157-8.  Back to cited text no. 8
[PUBMED]  [Full text]  


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]



 

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