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CASE REPORT
Year : 2006  |  Volume : 52  |  Issue : 2  |  Page : 134-135

Isolated breast mucormycosis


1 Departments of Surgery, Seth G.S. Medical College & K.E.M. Hospital, Parel, Mumbai - 400012, India
2 Departments of Pathology, Seth G.S. Medical College & K.E.M. Hospital, Parel, Mumbai - 400012, India

Correspondence Address:
Vinay Kumar Thapar
Departments of Surgery, Seth G.S. Medical College & K.E.M. Hospital, Parel, Mumbai - 400012
India
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Source of Support: None, Conflict of Interest: None


PMID: 16679679

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  Abstract 

Mucormycosis is unusual in surgical practice. Awareness of the classical findings leads to early detection. Excisional therapy whenever possible along with systemic antifungal treatment is the key to successful outcome. A 70 year old female, a known case of diabetes mellitus and chronic obstructive pulmonary disease, on inhalational steroids and oral hypoglycemic agents, presented to us with complaints of sudden onset pain, redness and swelling of left breast. A diagnosis of severe mastitis was made and a release incision was taken. The entire breast became gangrenous in next 24 hours and simple mastectomy had to be performed as a life saving measure. Histopathology revealed mucormycosis of breast. To the best of our knowledge this is the first reported case of mucormycosis of breast in the English literature.


Keywords: Mucormycosis, breast gangrene, mastectomy


How to cite this article:
Thapar VK, Deshpande A, Jain V K, Bhowate P, Madiwale C. Isolated breast mucormycosis. J Postgrad Med 2006;52:134-5

How to cite this URL:
Thapar VK, Deshpande A, Jain V K, Bhowate P, Madiwale C. Isolated breast mucormycosis. J Postgrad Med [serial online] 2006 [cited 2019 Nov 12];52:134-5. Available from: http://www.jpgmonline.com/text.asp?2006/52/2/134/25160


Mucormycosis is the common name given to several different diseases caused by fungi of the order Mucorales.[1] Rhizopus species are the most commonly isolated agents of mucormycosis, followed by Rhizomucor .[1] These fungi, which are widely distributed in nature usually, affect patients who are severely immunocompromised or those with diabetes mellitus or trauma.[1] Solid organ transplant recipients represent a growing population at risk.[2] More than half of patients have rhinocerebral disease; approximately 10% have pulmonary, cutaneous, or disseminated disease; and 2% have kidney or gastrointestinal involvement.[1] Breast involvement has not been described to date in the English literature reviewed by the authors.


  Case History Top


A 70 - year - old female presented with sudden onset of pain, redness and swelling of the left breast of 2 days duration. There was no history of trauma. She was a known case of diabetes mellitus on irregular treatment with oral hypoglycemic agents. She was also receiving inhaled corticosteroids for chronic obstructive pulmonary disease. On examination, patient was toxic, febrile and had pulse rate of 110 beats/minute. Her left breast was swollen, red, tender and warm. Investigations revealed a hemoglobin concentration of 10 gm% with a white cell count of 15,500/cu.mm. Renal and liver function tests were within normal limits as was the chest radiograph. A working diagnosis of severe mastitis with probable abscess was made. A release incision was taken over the breast to reduce the tissue tension and drain underlying pus collections if any. The entire breast tissue was edematous and there were no localized pus pockets. However, within 24 hours the underlying breast tissue showed necrosis with black eschar formation and gangrenous changes. A simple mastectomy was required to control the spread of necrosis. Histopathological examination revealed extensive necrosis of the breast parenchyma with polymorphonuclear infiltration, hemorrhage and thrombosed blood vessels. Seen amidst this inflammation were many broad aseptate hyaline fungal hyphae branching at 90o suggestive of mucormycosis. [Figure - 1] Patient was started on intravenous Amphotericin-B (dose of 1 mg/kg/day in the form of infusion in 5% dextrose) for a total of seven days and had an uneventful recovery. The open wound was grafted with split thickness skin graft after 4 weeks. She is asymptomatic at 2 month follow up.


  Discussion Top


Mucormycosis (phycomycosis, zygomycosis) as an infection is uncommon and is largely confined to severely immunocompromised patients such as diabetics, solid organ transplant recipients and trauma.[1],[2] The Mucoraceae are ubiquitous fungi that are common inhabitants of decaying matter. In contrast to the widespread distribution of these fungi, the infrequency of disease caused by these organisms attests to their low virulence potential in the human host.[1] The exact mechanisms responsible for the increased susceptibility to mucormycosis in various patient groups are not clear. Undefined defects of macrophages and neutrophils, present in diabetics and steroid-treated patients, may be important in allowing the replication of the Mucorales as was seen in this patient.[1],[3] Once the fungus begins to grow, the hyphae invade tissue and have a special affinity for blood vessels. Direct penetration and growth through the blood vessel wall explain the propensity for thrombosis and tissue necrosis, two major hallmarks of the histopathology of mucormycosis.[1]

There are six different manifestations of mucormycosis based on clinical presentation and involvement of a particular body site: (1) rhinocerebral, (2) pulmonary, (3) cutaneous, (4) gastrointestinal, (5) central nervous system, and (6) miscellaneous.[4] In general, the predilection for type of presentation varies with the underlying or predisposing condition. For example, patients with diabetes most often develop rhinocerebral mucormycosis, leukemic patients who have neutropenia or patients who become neutropenic during bone marrow transplantation for other diseases develop rhinocerebral or pulmonary mucormycosis, and those with protein-calorie malnutrition most often present with gastrointestinal disease. Disseminated disease results from progression from any of the primary anatomic location and is particularly troublesome in patients with severe immunologic deficits, such as those with bone marrow transplants or acute leukemia.[4],[5]

The hallmark of disease caused by the Mucorales are vascular invasion and tissue necrosis which results in black eschar and discharges. Diagnosis depends on demonstration of the organism in the tissue of a biopsy specimen. Grocott-Gomori methenamine-silver (GMS) stain is the best stain to use, but hematoxylin and eosin and periodic acid-Schiff are also useful.[1] The classical finding is that of necrotic and edematous tissue with neutrophilic infiltrate and presence of broad, nonseptate hyphae with branching at 90°.[1] Growth of this fungus occurs best in brain-heart infusion agar, potato dextrose agar, or Sabouraud dextrose agar.[1]

Systemic Amphotericin-B is the mainstay of treatment.[1] Liposomal Amphotericin-B may be more efficacious; it is less toxic, allowing higher dose of medications to be given.[1] Complete treatment of underlying medical disease is required. Any steroid medication, antimetabolites or immunosuppressants that the patient is on should be addressed and discontinued if necessary.[1] Aggressive surgical debridement of all necrotic tissue is necessary, sometimes requiring multiple debridements. Frozen section-guided debridement has been advocated as a method for operative intervention and as an alternative to the extensive debridement that has traditionally been performed in patients with invasive mucormycosis.[6] Adjunctive hyperbaric oxygen therapy has been considered beneficial in a small number of patients.[7] Overall, the earlier the diagnosis of mucormycosis is made, the better is the outcome. Survival of patients with pulmonary or disseminated mucormycosis is rare.[1]

In our patient, spores are likely to have gained entry to breast through unknown trivial trauma and multiplied and invaded the breast tissue with uncontrolled diabetes, old age and inhalational steroid acting as predisposing factors. Cases of cutaneous mucormycosis have been described after minor trauma; some of these patients had diabetes.[8],[9] Prompt excisional therapy complemented with systemic Amphotericin B led to a favorable response.

 
  References Top

1.Sugar AM. Agents of Mucormycosis and Related Species. In : Mandell GL, Bennett JE, Dolin R, editors. Principles and Practice of Infectious Diseases, 5th edn. New York: Churchill Livingstone; 2000. pp. 2685-91.  Back to cited text no. 1    
2.Singh N, Gayowski T, Singh J, Yu VL. Invasive gastrointestinal zygomycosis in a liver transplant recipient:Case report and review of zygomycosis in solid-organ transplant recipients. Clin Infect Dis 1995;20:617-20.   Back to cited text no. 2  [PUBMED]  
3.Chinn RY, Diamond RD. Generation of chemotactic factors by Rhizopus oryzae in the presence and absence of serum:Relationship to hyphal damage mediated by human neutrophils and effects of hyperglycemia and ketoacidosis. Infect Immun 1982;38:1123-9.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Parfrey NA. Improved diagnosis and prognosis of mucormycosis:A clinicopathologic study of 33 cases. Medicine (Baltimore) 1986;65:113-23.   Back to cited text no. 4  [PUBMED]  
5.St-Germain G, Robert A, Ishak M, Tremblay C, Claveau S . Infection due to Rhizomucor pusillus: Report of four cases in patients with leukemia and review. Clin Infect Dis. 1993;16:640-5.  Back to cited text no. 5    
6.Langford JD, McCartney DL, Wang RC. Frozen section-guided surgical debridement for management of rhino-orbital mucormycosis. Am J Ophthalmol 1997;124:265-7.  Back to cited text no. 6    
7.De La Paz MA, Patrinely JR, Marines HM, Appling WD. Adjunctive hyperbaric oxygen in the treatment of bilateral cerebro-rhino-orbital mucormycosis. Am J Ophthalmol 1992;114:208-11.  Back to cited text no. 7    
8.Rothburn MM, Chambers DK, Roberts C, Downie RJ. Cutaneous mucormycosis:A rare cause of leg ulceration. J Infect 1986;13:175-8.   Back to cited text no. 8    
9.Hicks WL Jr, Nowels K, Troxel J. Primary cutaneous mucormycosis. Am J Otolaryngol 1995;16:265-8.  Back to cited text no. 9    


    Figures

[Figure - 1]

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