Chhonkar A*, Nayak CS**, Tambe S***
(*Third Year Resident, **Professor and Head of Department, *** Assistant Professor, Department of Skin and V.D., T.N.M.C. & B.Y.L. Ch. Nair hospital, Dr. A.L.Nair Road, Mumbai central, Mumbai, Maharashtra-400008)
Botryomycosis is a chronic granulomatous inflammatory reaction to bacterial antigens. It may present with cutaneous or, less commonly, visceral involvement. It is a relatively rare infection that is more prevalent among immunocompromised patients. In cutaneous botryomycosis, the lesions may be pleomorphic including cysts, abscesses, fistulas, nodules, plaques or ulcers. Here we report an immunocompetent patient who developed botryomycosis after lower segment cesarean section. High frequency ultrasound imaging is very helpful in evaluating completion of healing.
Botryomycosis is a chronic granulomatous inflammatory reaction to bacterial antigens. It was initially mistaken for a fungal infection, thus the term botryomycosis (from Greek word botrys which means a bunch of grapes and mycosis which means of fungal origin) was used. Other less commonly used terms are bacterial pseudomycosis, staphylococcal actinophytosis and granular bacteriosis.[2,3] In cutaneous botryomycosis, the lesions may be pleomorphic including cysts, abscesses, fistulas, nodules, plaques or ulcers.The general swollen and suppurative aspect of the lesion suggests local inoculation of a foreign body as the initial cause of infection and perpetuation of the disease. It is more prevalent among immunocompromised patients.[5-7] However, it has also been reported in immunocompetent individuals as seen in our patient.
A 24 year old housewife presented with painful reddish swelling in groin of seven months duration with pus discharge following a lower segment cesarean section nine months earlier. A week after the surgery, she developed wound site infection which spread despite taking oral antibiotics and the wound site was excised and re-sutured. One week later, a tender boggy swelling developed over the excision site with multiple discharging sinuses. On examination, two oval, tender sinuses of 0.5x1cm with hyperpigmentation of the surrounding skin in the right inguinal region and a solitary, oval, tender, boggy swelling, with few pustules over it in the left inguinal region were seen and felt. (Figure 1). Routine hemogram, liver and renal function tests and other biochemical tests were normal. A differential diagnosis of actinomycosis, deep mycotic infection, botryomycosis and lupus vulgaris were considered. On investigation erythrocyte sedimentation rate (ESR) was 80mm/ hour while chest x-ray and mantoux test were negative. Cultures for AFB, fungal and bacterial infections were negative. Smear from discharge showed gram positive cocci. Histopathology revealed chronic granulomatous inflammation. She had received oral cefixime and amoxicillin-clavulinic acid combination in the past which led to partial resolution of the lesions but the lesions recurred on stopping the treatment. Ultrasonography (USG) of the local part revealed hypoechoic mass lesions involving the skin and subcutaneous tissue in both the inguinal regions suggestive of abscesses with bilateral lymphadenopathy. (Figure-2,3). She was given tablet cotrimoxazole (trimethoprin 160 mg + sulphamethoxazole 800 mg) twice a day for four months with complete resolution of the lesions (Figure-4) which was confirmed on USG before discontinuing the treatment.(Figure-5).
Figure 1. Two oval, tender sinuses with hyperpigmentation of surrounding skin in the right inguinal region and a solitary, oval, tender, boggy swelling, with few pustules over it in the left inguinal region.
Figure 2. Hypoechoic masses in skin and subcutaneous tissue in left inguinal region suggestive of abscesses with lymphadenopathy.
Figure 3. Hypoechoic masses in skin and subcutaneous tissue in right inguinal region suggestive of abscesses with lymphadenopathy.
Figure 4. Post treatment photograph showing resolution of lesion with scarring.
Figure 5. Post treatment USG showing decrease in size of left inguinal lymph node and resolution of abscess. Left image is pre treatment and right image is post treatment.
Botryomycosis was first described in 1870 by Bollinger, by observing granulomatous lesions as a complication after horse castration. In 1884, Rivolta coined the name Botryomycosis. The pathogenesis of the disease is not clear but it may be related to low virulence of infectious agents, large bacterial inoculum or change in specific cellular immunity or in humoral immune response.
Associated risk factors include alcoholism, diabetes mellitus, trauma and surgery, HIV infection, cystic fibrosis, chronic granulomatous disease.[3-5] A history of injury is common in cutaneous form of botryomycosis, which shows the role of foreign body as well as infection in the causation of the disease.
Cellular response in botryomycosis is similar to that seen in actinomycetoma and eumycetoma. The grain consisting of bacterial colonies is surrounded by acute suppurative response. The inflammatory infiltrate consists of numerous neutrophils surrounding the grain, lymphocytes, histiocytes, eosinophils, plasma cells, few foreign body giant cells and fibroblasts surrounding the central suppuration. Surrounding this infiltrate is the fibrosis and granulation response, with new vessel formation.
The most common causative agent is Staphylococcus aureus (40%), followed by Pseudomonas species (20%). Other microorganisms reported are Escherichia coli, Proteus vulgaris, Klebsiella, Neisseria, Streptococcus, Staphylococcus epidermidis, N Bacillus species and Actinobacillus lignieresii.[1,2]
There are two forms of botryomycosis- cutaneous and visceral. In cutaneous form, hands, pinna, feet and head are commonly affected. Skin folds in overweight individuals are also vulnerable areas. The lesions may be pleomorphic including cysts, abscesses, fistulas, nodules, plaques or ulcers. In visceral involvement, pulmonary botryomycosis is the most common entity.[7,8,9] There have also been reports of intraoral granulomatous pyogenic botryomycosis.
Diagnosis is confirmed on histopathological examination which reveals ‘Splendore Hoeppli’ phenomenon; grains of bacteria surrounded by eosinophilic material and inflammatory cells consisting of epitheloid cells, histiocytes, eosinophils and giant cells. Since multiple agents may be responsible in the formation of granules, cultures are essential to pin-point the causative organism. Ultrasonography with frequency higher than 7 MHz allows the visualization of superficial as well as deeper structures. Frequencies higher than 15 MHz are increasingly used in dermatology as they allow differentiation of skin layers. High frequency ultrasound along with color Doppler helps in the measurement of skin thickness as well as in investigation of tumors and inflammatory diseases and also helps in the evaluation of effectiveness of proposed treatments. 
Treatment requires antibiotic therapy and, in most cases surgical debridement. The selection of antibiotics should be in keeping with the results of bacterial cultures.
This case highlights the importance of prolonged anti-bacterial therapy, preferably based on antibiotic sensitivity reports, till complete clinical cure. This case also highlights the efficacy of age old but less used anti-bacterials like co-trimoxazole in some cases. Ultrasonography of skin helped us to locate the pus pockets and the repeat scan helped us confirm that all pus collections had cleared before we decided to stop the treatment. Therefore, USG of skin is useful in confirming complete resolution of inflammatory lesions.
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Chhonkar A, Nayak CS, Tambe S. Botryomycosis of Anterior Abdominal Wall following Cesarean Section. JPGO 2017. Volume 4 No.2. Available from: http://www.jpgo.org/2017/02/botryomycosis-of-anterior-abdominal.html