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CASE REPORT Table of Contents   
Year : 2007  |  Volume : 18  |  Issue : 3  |  Page : 131-134
Micrococcal botryomycosis of the left temporal region


Dept. of Oral and Maxillofacial Surgery, Saveetha Dental College and Hospitals, No 162, P. H. Road, Chennai - 600 077, India

Click here for correspondence address and email

Date of Submission13-Jul-2006
Date of Decision23-Nov-2006
Date of Acceptance19-Jan-2007
 

   Abstract 

Botryomycosis arises from chronic infections produced by low-virulence organisms in an altered host environment. Staphylococci have been the most common organisms implicated, but various other bacteria have also been identified in human botryomycosis lesions. The relative balance between the host's resistance and the microorganism's virulence may be altered in some way that perpetuates the growth of the lesions in a symbiotic fashion. The diagnosis of botryomycosis is one that is often easily overlooked because it can be confused with other mycetomas such as actinomycosis and nocardosis. We report here a case of micrococcal botryomycosis that occurred in the left temporal region in a 70 year-old male, which was diagnosed by the help of a histopathological examination and microbial cultures.

Keywords: Botryomycosis, micrococci, sulfur-like granules

How to cite this article:
Sivaraj S, Muthu Sekhar M R, Baig M F. Micrococcal botryomycosis of the left temporal region. Indian J Dent Res 2007;18:131-4

How to cite this URL:
Sivaraj S, Muthu Sekhar M R, Baig M F. Micrococcal botryomycosis of the left temporal region. Indian J Dent Res [serial online] 2007 [cited 2020 Oct 29];18:131-4. Available from: https://www.ijdr.in/text.asp?2007/18/3/131/33790

   Introduction Top


The term "botryomycosis" was previously synonomous with granular bacteriosis, staphylococcal actinophytosis and bacterial pseudomycosis. The nomenclature has been further confused by some dermatologic literature that describes the pyogenic granuloma as "pseudobotryomycosis" or "botryomycoma". Several of the earlier cases of botryomycosis were not well-documented and some have appeared in obscure journals. Due to these difficulties, it is not possible to determine the actual number of human cases with certainty.

In 1983, there were an estimated 77 cases reported in literature worldwide. Since then, another five have been reported bringing the total to 82. Winslow summarized the literature in 1959 and subcategorized botryomycosis into integumental and visceral forms. [1] The integumental form is more common and is characterized by localized granulomatous skin infections often associated with trauma, a foreign body or chronic wound contamination. It usually involves exposed body surfaces such as the hands, feet or the head. In rare cases, it can cause osteomyelitis by contiguous spread.

Visceral botryomycosis has been known to affect the liver, [2],[3],[4] lung [1],[4],[5] kidney [1],[4],[6] prostate, [6] spleen, [4] bowel, [7] omentum, [7] brain [8] and lymphatic tissues (submandibular lymph node [1] and tonsil). [9] Disseminated visceral botryomyosis is associated with chronic disease or systemic immunocompromise such as cystic fibrosis [10] or diabetes mellitus [4],[5],[11] and can be fatal. [4],[6],[10] The diagnosis of botryomycosis is one that is often easily overlooked because it can be confused with other mycetomas such as actinomycosis and nocardosis.


   Case Report Top


A 70 year-old man presented with a soft fibrotic mass in the left temporal region that had been slowly growing for the past three months. The patient's left temporal region had been injured in a road traffic accident five months back. The patient was suffering from chronic diabetes being controlled by oral hypoglycemic drugs.

On evaluation, the patient was noted to have marked oedema and continuous pain with induration of adjacent soft tissues of the left temporal region. There were multiple sinuses with chronic pus discharge surrounding the fibrotic mass [Figure - 1]. Clear saliva could be expressed from the left parotid papilla. There was no cervical lymphadenopathy. Random blood sugar was 120 mg/dl.

Under local anaesthesia, the soft fibrotic mass was excised and the wound was irrigated with hydrogen peroxide in normal saline. This was followed by the placement of a corrugated drain for subsequent drainage of pus [Figure - 2]. The excised mass and the pus were sent for microscopic examination. Microscopic examination showed necrotizing granulomatous inflammation and fibrosis with small sulfur-like granules containing micrococci [Figure - 3]. Silver and acid-fast stains were negative.

Routine culture demonstrated rare growth of micrococci that were sensitive to ofloxacin, cephalothin and vancomycin. No organisms were recovered from anaerobic, fungal or mycobacterial cultures. A diagnosis of botryomycosis was finally made after sharing these findings with several pathologists and infectious disease specialists. The patient was placed on a course of oral ofloxacin, which he took for three weeks. At the end of the first week, the condition showed gradual improvement [Figure - 4].


   Discussion Top


The disease, later referred to as botryomycosis, was first described involving the lung of a horse by Bollinger in 1870. [12] He reported the presence of multiple fibrous nodules resembling those of actinomycosis. Microscopically, there was no evidence of the branching filaments characteristic of actinomycosis; rather, there were coccus-like bodies surrounded by a homogeneous capsule. Rivolta [13] found the same process in a tumor from the cut spermatic cord of a horse in 1884 and believing that the globular granules were related to Actinomyces, named it Discomyces equi. After further study, he renamed the disease botryomycosis in 1887 ("botryos" from Greek for bunch of grapes). Scattered accounts of the disease involving superficial wounds of horses, cattle, sheep and swine were found in veterinary medicine literature at the turn of the century. These were usually a complication of castration or were located in areas of irritation or friction such as under a harness.

The first human cases of botryomycosis were reported in 1897 by Poncet and Dor. [14] These involved three small, pedunculated growths, two found on the hand and one on the stump of an arm amputated at the shoulder. Later, pathologists [15] disputed the interpretation of these lesions and believed them to actually represent more common entities such as pyogenic grauloma. In 1913, Opie published a case report of an 11 year-old girl who died with a large mass in the liver. [3] The mass contained coccus-like bodies with a homogeneous eosinophilic capsule forming lobulated colonies. There was an abundant background of macrophages, polymorphonuclear leukocytes, plasma cells, purulent exudates and fibrous connective tissue. This was the first human case reported in the United States and the first account of visceral involvement.

Botryomycosis is best considered a reactive process initiated by low-grade bacterial infections. The lesions are generally indurated fibrotic masses that may form draining sinuses and fistulae. Microscopic examination of haematoxylin-eosin-stained specimens shows chronic suppurative and granulomatous inflammation with giant cells, epithelioid macrophages and scattered microabscesses. There is marked fibrosis and desmoplasia of the connective tissue. [16] Within the areas of purulent inflammation are characteristic grains, the size of fine sand particles, which have been referred to as Bollinger's granules. [17] On gross examination, Bollinger's granules are indistinguishable from the sulfur granules of actinomycosis and nocardiosis. Gram and Giemsa stains can be used to identify the causative bacterial organisms within the granules.

The granules are surrounded by an amorphous, eosinophilic, refringent matrix called the Splendore-Hoeppli phenomenon [18],[19] [Figure - 5]. These deposits may form radial, club-like projections around the granules, possibly in response to cellular debris. The Splendore-Hoeppli phenomenon is not unique to botryomycosis and has been observed around colonies of certain bacteria, fungi, helminthes, actinomyces, mycetoma, nasofacial and subcutaneous phycomycosis and around silk sutures. [18] It has been suggested that this eosinophilic matrix might represent antigen-antibody complexes. [1],[18],[20]

The limited numbers of studies using direct immunofluorescence have not clarified this issue with some reporting no identifiable complexes, [21],[22] and another finding deposits of gamma G immunoglobulin (IgG) [23],[24] or C 3 . [24] Botryomycosis arises from chronic infections produced by low-virulence organisms in an altered host environment. Staphylococci have been the most common organisms implicated, but various other bacteria have also been identified in human botryomycosis lesions. The relative balance between the host's resistance and the microorganism's virulence may be altered in some way that perpetuates the growth of the lesion in a symbiotic fashion. Local host defences may be compromised by postoperative wound infections, [22] tetanus, [24] osteomyelitis, [1],[15],[24] follicular mucinosis [17] or foreign bodies. [12],[25]

Botryomycosis has been associated with immune-compromised individuals with diabetes mellitus, [4],[5],[11] chronic corticosteroid therapy, [22] chronic granulomatous disease, [2] recurrent mucocutaneous candidiasis, [26] acquired immune deficiency syndrome (AIDS) [23] and severe malnutrition. [27] Most individuals with botryomycosis have normal humoral [17],[21],[22],[26] and cellular [17],[22] immunity. There has been one reported serum IgA deficiency, [24] one quantitative B cell deficiency, [17] one salivary IgA deficiency [26] and one combined T and B cell deficiency. [21] Few cases of oral and facial botryomycosis have been reported. Kansky [28] in 1964 described a case of staphylococcal botryomycosis emanating from a scar across the cheek.

Botryomycosis of the tongue was reported by Small and Kobernick. [29] Alavandar [30] found staphylococcal botryomycosis of the mandible in a 7 year-old anemic Indian boy who presented with the clinical picture of actinomycotic "lumpy jaw". He had bilateral mandibular involvement and multiple draining sinuses. Bunken et al.[21] described botryomycosis of the submandibular skin. In 1989, Maves et al.[31] reported a 54 year-old man with a painless submucosal mass between the parotid duct and the retromolar trigone. Treatment in all these cases was combined surgical and antibiotic therapy. Our case illustrates many of the characteristic features of botryomycosis and had severe predisposing factors for the development of this condition. As typically seen in other reports, recognition of the correct diagnosis was delayed.

 
   References Top

1.Winslow DJ. Botryomycosis. Am J Pathol 1959;35:153-67.  Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Washburn RG, Bryan CS, DiSalvo AF, Macher AM, Gallin JI. Visceral botryomycosis caused by Neisseria mucosa in a patient with chronic granulomatous disease. J Infect Dis 1985;151:563-4.  Back to cited text no. 2  [PUBMED]  
3.Opie EL. Human botryomycosis of the liver. Arch Intern Med 1913;11:425.  Back to cited text no. 3    
4.Beaver DC, Thompson L. Actinobacillosis in man: Report of a fatal case. Am J Pathol 1933;9:603.  Back to cited text no. 4    
5.Spier WA Jr, Mitchener JW, Galloway RF. Primary pulmonary botryomycosis. Chest 1971;60:92-3.  Back to cited text no. 5    
6.Winslow DJ, Chamblin SA. Disseminated visceral botryomyosis. Report of a fatal case probably caused by Pseudomonas aeruginosa. Am J Clin Pathol 1960;33:43-7.  Back to cited text no. 6    
7.Rouschkolb EW. Staphylococci actinophytosis (botryomy cosis). Arch Dermatol 1961;83:161.  Back to cited text no. 7    
8.Wu WQ, Cattaneo EA, Lapi A, Halde C. Botryomycosis: First report of human brain involvement. South Med J 1978;71:1530-3.  Back to cited text no. 8  [PUBMED]  
9.Martins RH, Heshiki Z, Luchesi NR, Marques ME. Actinomycosis and botryomycosis of the tonsil. Auris Nasus Larynx 1991;18:377-81.  Back to cited text no. 9  [PUBMED]  
10.Katznelsen D, Vawter GF, Foley GE, Shwachman H. Botryomycosis A complication in cystic fibrosis. Report of 7 cases. J Pediatr 1964;65:525-39.  Back to cited text no. 10    
11.Kimmelsteil P, Oden PW. Botryomycosis: Report of two cases of Intra abdominal granuloma. Arch Pathol 1939;27:313.  Back to cited text no. 11    
12.Bollinger O. Mycosis der lunge beim pferde. Virchows arch (PA) 1870;49:586.  Back to cited text no. 12    
13.Rivolta S. Del micelio e delle varieta e specie di discomiceti pagogeni. Giorn Anat Fisiol Patol Animali 1884;16:181, (cited by Opie, p. 426).  Back to cited text no. 13    
14.Poncet, Dor: XI Congr. Francais de chirurgie, paris, 1897; Lyon Med 43:213, 1897; 5: 145, 1898: Arch Gen Med iii: 129, 274, 1900. Quoted by Galli-Vallerio: Centralbl Bakteril xxxi: 508, 1902 Cited by Opie. [10] P 427.  Back to cited text no. 14    
15.Berger L, Vallee A, Vezina C. Genital staphylococci actinophytosis (botryomycosis) in human beings. Arch Pathol 1936;21:273.  Back to cited text no. 15    
16.Binford CH, Dooley JR. Diseases caused by fungi and actinomycetes. In : Binford CH, Connor DH, editors. Pathology of tropical and extraordinary disease. vol. 2. Armed Forces Institute of Pathology: Washington, DC; 1976. p. 561.  Back to cited text no. 16    
17.Harman RR, English MP, Halford M, Saihan EM, Greenham LW. Botryomycosis: A complication of extensive follicular mucinosis. Br J Dermatol 1980;102:215-22.  Back to cited text no. 17  [PUBMED]  
18.Johnson FB. Splendore-Hoeppli phenomenon. In : Binford CR, Connor DH, editors. Pathology of tropical and Extraordinary Disease Vol 2. Armed Forces Institute of Pathology: Washington, DC; 1976. p. 681.  Back to cited text no. 18    
19.Swartz MN, Weinberg AN. Infections due to Gram positive bacteria. In : Fitzpatrick TB, Eisen AZ, Wolff K, et al, editors. Dermatology in General Medicine, Vol. 2, 4 th ed. McGraw Hill: New York; 1993. p. 2330.  Back to cited text no. 19    
20.Winslow DJ, Steen FG. Considerations in the histologic diagnosis of mycetoma. Am J Clin Pathol 1964;42:164-9.  Back to cited text no. 20  [PUBMED]  
21.Brunken RC, Lichon-Chao N, van der Broek H. Immunologic abnormalities in botryomycosis. J Am Acad Dermatol 1983;9:428-34.  Back to cited text no. 21  [PUBMED]  
22.Bishop GF, Greer KE, Horwitz DA. Pseudomonas botryomycosis. Arch Dermatol 1976;112:1568-70.  Back to cited text no. 22  [PUBMED]  
23.Patterson JW, Kitces EN, Neafie RC. Cutaneous botryomycosis in a patient with acquired immunodeficiency syndrome. J Am Acad Dermatol 1987;16:238-42.  Back to cited text no. 23  [PUBMED]  
24.Martin-Pascual A, Perez AG. Botryomycosis. Dermatologica 1975;151:302-8.  Back to cited text no. 24    
25.Kimmelsteil P, Easley CA. Experimental botryomycosis. Am J Pathol 1940;16:95.  Back to cited text no. 25    
26.Buckley RH, Lucas ZJ, Hattler BG Jr, Zmijewski CM, Amos DB. Defective cellular immunity associated with chronic mucocutaneous moniliasis and recurrent. Staphylococcal botryomycosis: Immunological reconstruction by allogenic bone marrow. Clin Exp Immunol 1968;3:153-69.  Back to cited text no. 26    
27.Picou K, Batres E, Jarratt M. Botryomycosis: A bacterial cause of mycetoma. Arch Dermatol 1979;115:609-10.  Back to cited text no. 27  [PUBMED]  
28.Kansky A. Botryomycosis. Acta Derm Venereol (Stockh) 1964;44:369.  Back to cited text no. 28    
29.Small IA, Kobernick S. Botryomycosis of the tongue. Report of a case. Oral Surg Oral Med Oral Pathol 1967;24:503-9.  Back to cited text no. 29    
30.Alavandar G. Botryomycosis of the mandible. J Indian Dent Assoc 1979;51:181-4.  Back to cited text no. 30  [PUBMED]  
31.Maves MD, Aguiar SW, Massanari RM, Robinson RA. Botryomyosis of the oral cavity. Head Neck 1989;11:274-8.  Back to cited text no. 31  [PUBMED]  

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Correspondence Address:
M R Muthu Sekhar
Dept. of Oral and Maxillofacial Surgery, Saveetha Dental College and Hospitals, No 162, P. H. Road, Chennai - 600 077
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.33790

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