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  Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 10  |  Issue : 3  |  Page : 311-315  

Cutaneous botryomycosis in immunocompetent patients: A case series


1 Department of Dermatology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
2 Department of Pathology, Self Employed, Bhubaneswar, Odisha, India

Date of Web Publication17-May-2019

Correspondence Address:
Swetalina Pradhan
Department of Dermatology, All India Institute of Medical Sciences, Bhubaneswar - 751 019, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_370_18

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   Abstract 


Botryomycosis is a rare chronic suppurative bacterial infection of skin and viscera mostly reported in immunocompromised adults. Most of published literature on botryomycosis are case reports. Though morphological presentation of cutaneous botryomycosis has been described as nodules, sinus, abscesses, and ulcers discharging seropurulent exudates, sequential evolution of lesions is not clear. We report a series of three cases of cutaneous botryomycosis in immunocompetent patients (one child and two adults). Two cases had localized lesion, while adult male had lesions in a sporotrichoid distribution. In all cases the lesions evolved in the form of appearance of subcutaneous swelling which later on developed multiple nodules and papules on surface which either developed erosion, ulceration or sinus on surface associated with seropurulent discharge. The organisms isolated from discharge and tissue culture were coagulase negative staphylococcus and methicillin sensitive staphylococcus aureus. All cases were treated with monotherapy of sensitive systemic antibiotic. Two patients fully recovered and one lost to follow-up in the middle of therapy.

Keywords: Cutaneous botryomycosis, evolution, lobulated swelling, morphology, sporotrichoid pattern, ulcerated papule and nodule


How to cite this article:
Sirka CS, Dash G, Pradhan S, Naik S, Rout AN, Sahu K. Cutaneous botryomycosis in immunocompetent patients: A case series. Indian Dermatol Online J 2019;10:311-5

How to cite this URL:
Sirka CS, Dash G, Pradhan S, Naik S, Rout AN, Sahu K. Cutaneous botryomycosis in immunocompetent patients: A case series. Indian Dermatol Online J [serial online] 2019 [cited 2019 Sep 18];10:311-5. Available from: http://www.idoj.in/text.asp?2019/10/3/311/258608




   Introduction Top


Cutaneous botryomycosis is a rare and chronic suppurative bacterial infection with commonest causative organism being S. aureus.[1],[2] Most of published literature on botryomycosis are case reports. It has been commonly reported in immunocompromised adults and usually occurs over trauma prone areas following local injury or at post-operative sites.[3] Skin lesions usually present as papules, nodules, fistulas, abscesses, and ulcers with seropurulent discharge.[3] However, characteristic morphological features giving clue to the diagnosis of cutaneous botryomycosis are unknown. It is usually treated with specific antibiotic based on culture sensitivity either single or combination of multiple drugs for prolonged duration.

We present three immunocompetent patients of cutaneous botryomycosis with lesions present on various parts of body with different distribution patterns and evolution of lesions; and were successfully treated with sensitive antibiotic monotherapy.


   Case Report Top


Three immunocompetent patients (one child and two adults) presented with subcutaneous swellings with seropurulent discharge at various sites of body. Case 1 and 2 had localized lesions whereas case 3 had lesions in a sporotrichoid distribution. Case 1, 15-year old female child came with multiple erythematous mildly painful, subcutaneous swellings over left side of forehead and upper eyelid with ulceration and seropurulent discharge since 4 years [Figure 1]a. There was preceding history of trauma over forehead 6 years back. The lesions started as subcutaneous swellings at one end of the scar and underwent ulceration on surface of swelling with few lesions developing sinuses giving seropurulent discharge after around 4 to 6 weeks. Surgical drainage was done on two occasions within one year without any significant improvement. Based on the history and clinical examination, differential diagnoses of botryomycosis, actinomycetoma or eumycetoma, scrofuloderma were considered. Routine haematological parameters were within normal limits. HIV 1and 2 were negative. X-ray of skull didn't reveal any bony abnormalities. Mantoux test was negative. Culture of discharge and tissue showed growth of coagulase negative staphylococcus sensitive to doxycycline. Histopathology from the nodule showed bacterial colonies in dermis with an eosinophilic rim at the periphery suggestive of Splendore–Hoeppli phenomenon [Figure 1]b and [Figure 1]c. A diagnosis of botryomycosis was made and oral doxycycline 100 mg twice daily was started. The lesions resolved completely after 4 weeks [Figure 1]d. We continued the drug for 6 more weeks after the clinical cure. The patient didn't develop any side effects to doxycycline. Currently the patient is under follow up without any recurrence in last 5 months. Demographic details, and morphology and evolution of lesions in all cases have been described in [Table 1].
Figure 1: a) Nodules with ulceration and crusting on surface on left side of forehead and left upper eyelid. b) H and E, 10×: bacterial colonies in dermis with an eosinophilic rim at the periphery. c) H and E, 40×: bacterial colonies in dermis with an eosinophilic rim at the periphery. d) Complete healing of lesions after treatment

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Figure 2: a) Diffuse swelling on upper back and nape of the neck studded with skin coloured to erythematous papules, nodules, ulcers and scars. b) Plate like mass on nape of neck and nodules with granulation tissue giving pyogenic granuloma like morphology. c) Clustered basophilic granules surrounded by prominent eosinophilic club of Splendore-Hoeppli material deep in dermis. d) Complete healing of lesions following treatment

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Figure 3: a) Subcutaneous lobulated swellings over left knee studded with multiple papules, nodules and ulcers. b) Nodules with granulation tissue giving pyogenic granuloma like morphology. c) Plate like mass in proximal thigh studded with papules and nodules. d) Sporotrichoid pattern with normal skin in between the lesions on proximal thigh and knee

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Figure 4: Bacterial granules in dermis with eosinophilic rim. H and E, 10×

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Figure 5: Healing of lesions following treatment

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Table 1: Demographic details, history, evolution, morphology, treatment and outcome of patients

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   Discussion Top


Botryomycosis can involve skin and viscera. Around 200 cases have been reported from all over the world.[3] The present report is series of three cases of cutaneous botryomycosis.

Majority of reported cases of cutaneous botryomycosis are immunocompromised adults and has been reported rarely in children.[4] All our cases were immunocompetent with one child and two adults. Common organisms causing botryomycosis include Staphylococcus aureus, Pseudomonas aeruginosa, coagulase-negative staphylococci, Streptococcus spp, Escherichia coli and Proteus spp.[3] In the present series, the organism isolated were coagulase negative staphylococcus in child and adult female and MSSA (methicillin sensitive staphylococcus aureus) in adult male. Various predisposing factors and co-morbid conditions associated with botryomycosis include altered immune function, diabetes mellitus, liver disease, alcoholism, lupus, cystic fibrosis, asthma, malnutrition, immunoglobulin deficiency, hyperglobulinemia E (Job syndrome), glomerulonephritis, long term treatment with corticosteroids and human immunodeficiency virus/acquired immunodeficiency syndrome.[5],[6] Factors like accident and post-operative skin trauma help in entry of the organism and thereby leading to localized infections.[7] Except prior history of local trauma in case 1 and 3, there was no other associated co-morbid condition in our cases. Described morphological presentation of cutaneous botryomycosis include nodules, sinus, abscesses, and ulcers giving a sero-purulent exudate with occasional granules of bacteria.[4],[8] In the present series, besides the appearance of nodule, subcutaneous swelling, sinus and ulcer, we observed few nodules with ulceration and granulation tissue giving morphology of pyogenic granuloma in case 2 and 3. Secondly, the third case was interesting since the lesions were in a linear sporotrichoid pattern which has not been described previously in literature.

In botryomycosis, there occurs formation of a rim of eosinophilic matrix composed of antigen-antibody complex, tissue debris and fibrin around the bacterial granules and colonies called Splendore–Hoeppli phenomenon (SHP), which prevents phagocytosis and intracellular destruction of the bacteria leading to chronic infection.[7],[9],[10]

The grains in botryomycosis are large and lobulated which are cluster of bacteria that does not have a filamentous structure (like grains of mycetoma and actinomycosis). All our cases had bacterial granules which demonstrated SHP in histopathology.

Botryomycosis should be clinically differentiated from other conditions like mycetoma, actinomycosis, actinomycetoma, nocardiosis and tuberculosis that presents as swellings with sinus and discharge. However, there has been no clear cut morphological differentiation of above entities. In the present case series, we observed evolution of lesions of botryomycosis which could help in reaching at clinical diagnosis of botryomycosis. All cases developed subcutaneous swelling, followed by papule and nodule over the swelling which either developed, erosion, ulceration or sinus giving seropurulent discharge. In case 2 and 3 few sinuses and nodules developed pedunculated granulation tissue giving an appearance of pyogenic granuloma. Such an evolution noted in our cases could be possibly due to localized inflammation occurring around the organisms giving rise to subcutaneous swelling, which later on extruded out by formation of nodules, papule, and sinus on the surface of subcutaneous swelling. These lesions underwent secondary changes like erosion, ulceration and sinus on the papules and nodules. The sporotrichoid distribution in case 3 could be due the lymphatic spread.

The diagnosis of botryomycosis is usually confirmed by demonstration of specific bacteria in discharge, tissue culture and characteristic histopathology findings. The causative organisms detected in tissue and pus culture were coagulase negative staphylococci in case 1 and 2 and MSSA in case 3 which ruled out other differentials like mycetoma, nocardiosis and tuberculosis.

The treatment with prolonged course of antibiotics depending upon pus culture and sensitivity is recommended for botryomycosis. However, appropriate antibiotics in combination with surgical excision is the most effective therapy for botryomycosis.[3] All our cases were treated with prolonged treatment with single sensitive antibiotic. Two cases were treated with doxycycline monotherapy whereas the adult female was treated with ciprofloxacin similar to the report by Pradhan et al.[4] None of the cases needed any surgical intervention.


   Conclusion Top


From the present series of three cases, we suggest the evolution of lesions along with demonstration of bacteria in the discharge and tissue culture and characteristic histopathology findings can help to diagnose botryomycosis. Secondly, we also highlight new morphological findings like pyogenic granuloma like lesions and sporotrichoid distribution that can occur in the botryomycosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Bonifaz A, Carrasco E. Botryomycosis. Int J Dermatol 1996;35:381-8.  Back to cited text no. 1
    
2.
Machado CR, Schubach AO, Conceição-Silva F, Quintella LP, Lourenço MC, Carregal E, et al. Botryomycosis. Dermatology 2005;211:303-4.  Back to cited text no. 2
    
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Padilla-Desgarennes C, Vázquez-González D, Bonifaz A. Botryomycosis. Clin Dermatol 2012;30:397-402.  Back to cited text no. 3
    
4.
Pradhan S, Sirka CS, Panda M, Baisakh M. Cutaneous botryomycosis treated successfully with injectable ceftriaxone sodium in an immunocompetent child. Indian J Dermatol Venereol Leprol 2018;84:485-7.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
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. 5
    
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Saadat P, Ram R, Sohrabian S, Vadmal MS. Botryomycosis caused by Staphylococcus aureus and Pneumocystis carinii in a patient with acquired immunodeficiency disease. Clin Exp Dermatol 2008;33:266-9.  Back to cited text no. 6
    
7.
Follows GA, Mathew J, Lucas S, Black MJ, Goodship TH. Cutaneous botryomycosis in a patient with lupus nephritis. Nephrol Dial Transplant 1998;13:3200-1.  Back to cited text no. 7
    
8.
Chintaginjala A, Harshavardhan K, Senthil Kumar AL. Cutaneous botryomycosis: A rare case report. Indian J Dermatol 2016;61:126.  Back to cited text no. 8
    
9.
Bersoff-Matcha SJ, Roper CC, Liapis H, Little JR. Primary pulmonary botryomycosis: Case report and review. Clin Infect Dis 1998;26:620-4.  Back to cited text no. 9
    
10.
Akiyama H, Kanzaki H, Tada J, Arata J. Staphylococcus aureus infection on cut wounds in the mouse skin: Experimental staphylococcal botryomycosis. J Dermatol Sci 1996;11:234-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
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