Fulminant necrotizing fasciitis caused by zygomycetes

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J Cutan Pathol 2009: 36: 815–816 John Wiley & Sons. Printed in Singapore Copyright # 2009 John Wiley & Sons A/S Journal of Cutaneous Pathology Letter to the Editor Fulminant necrotizing fasciitis caused by zygomycetes To the Editor, Necrotizing fasciitis (NF) is a progressive, rapidly spreading, inflammatory infection located in the deep fascia, with secondary necrosis of the subcutaneous tissues. Most cases are bacterial in origin, caused by mixed aerobic and anaerobic organisms (70%), anaerobes (20%) and aerobes (10%). 1 It is commonly associated with severe systemic toxicity and high mortality in the range of 25–70%. 2 Opportunistic fungal infections are an important cause of cutaneous necrotizing infections in immunocompromised pa- tients, and zygomycosis in the debilitated patient is the most acute and fulminant fungal infection known. 3 Percutaneous risks for developing infection with these fungi are well described, 4 and here in, we present a case of fulminant NF caused by zygomycetes following an intramuscular injection. A 23-year-old female presented in casualty with high-grade fever and a deep ulcer involving the right gluteal region, extending to her thigh and lower back. She gave a history of an unknown intramus- cular injection 1 month back. Two weeks later, she had developed a painful ulcer at the site of the injection. She had received some local treatment (details unknown) from a private medical practi- tioner, but as the lesion started extending, she was referred to our hospital. On examination, patient was febrile, toxic and had a large soft tissue infection spreading along the fascial planes producing necrosis of overlying skin. The area involved included the right gluteal region, thigh and lower back. (Fig. 1A) A clinical diagnosis of NF was established, and an emergency surgical debridement was carried out, and broad-spectrum antibiotics active against both aerobic and anaerobic bacteria ware started. Laboratory studies revealed hemoglobin of 3 gm/ dl, total white cell count of 15,000/mm 3 with raised neutrophils. Serum biochemistry showed deranged renal function test with urea and creatinine 80 and 3 mg/dl, respectively, urine analysis showed pro- teinuria. Bacterial culture of excised tissue showed growth of Proteus mirabilis, susceptible to third generation cephalosporins and aminoglycosides. In spite of aggressive management, the patient contin- ued to deteriorate. The diagnosis was reviewed, and considering a possibility of cutaneous mycosis, surgi- cally excised tissues were sent for mycological evalu- ation. KOH wet mount showed broad, nonseptate hyphae (Fig. 1B), and in sections of resected tissue stained by hematoxylin and eosin (Fig. 1C), periodic acid-Schiff (Fig. 1D) and Grocott methenamine silver stains broad hyphae of varying diameters with minimal septation and irregular branching were seen. Fungal culture on Sabouraud’s dextrose agar showed no growth. Amphotericin-B could not be started because patient was in renal failure, and the clinical condition of patient kept on deteriorating despite surgical de- bridement and intensive medical management. The patient and her family denied further treatment and took leave against medical advice. Zygomycetes class of fungi includes three orders that are Mucorales, Mortierellales and Entomo- phthorales. The majority of human illness is caused by the Mucorales. 4 Zygomycosis is an emerging cause of NF, and in a recent study, zygomycosis was responsible for 31.03% cases of NF. 5 Early diagnosis is the corner stone of successful treatment of zygomycosis. Treatment of zygomycosis requires several simultaneous approaches: surgical interven- tion, antifungal therapy and correction of the under- lying predisposing condition. Surgical debridement of grossly necrotic tissue is always required; in addition, antifungal agents such as Amphotericin B/posacona- zole should be coadministered. Hyperbaric oxygen, Granulocyle colony stimulating factor and interferon- g might give some benefit, as adjunctive treatment and their role require further evaluation. 4,6 Continued expansion of the wound despite broad- spectrum antibiotic therapy, failure to isolate bacterial organisms and demonstration of ribbon like, aseptate hyphae in tissue section are some of features that are helpful in the early diagnosis of NF of fungal etiology. Rapid progression and unacceptably high mortality 815

Transcript of Fulminant necrotizing fasciitis caused by zygomycetes

J Cutan Pathol 2009: 36: 815–816John Wiley & Sons. Printed in Singapore

Copyright # 2009 John Wiley & Sons A/S

Journal of

Cutaneous Pathology

Letter to the Editor

Fulminant necrotizing fasciitis causedby zygomycetes

To the Editor,Necrotizing fasciitis (NF) is a progressive, rapidly

spreading, inflammatory infection located in the deepfascia, with secondary necrosis of the subcutaneoustissues. Most cases are bacterial in origin, caused bymixed aerobic and anaerobic organisms (70%),anaerobes (20%) and aerobes (10%).1 It is commonlyassociated with severe systemic toxicity and highmortality in the range of 25–70%.2 Opportunisticfungal infections are an important cause of cutaneousnecrotizing infections in immunocompromised pa-tients, and zygomycosis in the debilitated patient is themost acute and fulminant fungal infection known.3

Percutaneous risks for developing infection with thesefungi are well described,4 and here in, we presenta case of fulminant NF caused by zygomycetesfollowing an intramuscular injection.A 23-year-old female presented in casualty with

high-grade fever and a deep ulcer involving the rightgluteal region, extending to her thigh and lowerback. She gave a history of an unknown intramus-cular injection 1 month back. Two weeks later, shehad developed a painful ulcer at the site of theinjection. She had received some local treatment(details unknown) from a private medical practi-tioner, but as the lesion started extending, she wasreferred to our hospital. On examination, patientwas febrile, toxic and had a large soft tissue infectionspreading along the fascial planes producing necrosisof overlying skin. The area involved included theright gluteal region, thigh and lower back. (Fig. 1A)A clinical diagnosis of NF was established, and anemergency surgical debridement was carried out,and broad-spectrum antibiotics active against bothaerobic and anaerobic bacteria ware started.Laboratory studies revealed hemoglobin of 3 gm/dl, total white cell count of 15,000/mm3 with raisedneutrophils. Serum biochemistry showed derangedrenal function test with urea and creatinine 80 and3 mg/dl, respectively, urine analysis showed pro-teinuria. Bacterial culture of excised tissue showedgrowth of Proteus mirabilis, susceptible to third

generation cephalosporins and aminoglycosides. Inspite of aggressive management, the patient contin-ued to deteriorate. The diagnosis was reviewed, andconsidering a possibility of cutaneous mycosis, surgi-cally excised tissues were sent for mycological evalu-ation. KOH wet mount showed broad, nonseptatehyphae (Fig. 1B), and in sections of resected tissuestained by hematoxylin and eosin (Fig. 1C), periodicacid-Schiff (Fig. 1D) and Grocott methenaminesilver stains broad hyphae of varying diameters withminimal septation and irregular branching wereseen. Fungal culture on Sabouraud’s dextrose agarshowed no growth.

Amphotericin-B could not be started becausepatient was in renal failure, and the clinical conditionof patient kept on deteriorating despite surgical de-bridement and intensive medical management. Thepatient and her family denied further treatment andtook leave against medical advice.

Zygomycetes class of fungi includes three ordersthat are Mucorales, Mortierellales and Entomo-phthorales. The majority of human illness is causedby the Mucorales.4 Zygomycosis is an emergingcause of NF, and in a recent study, zygomycosiswas responsible for 31.03% cases of NF.5 Earlydiagnosis is the corner stone of successful treatmentof zygomycosis. Treatment of zygomycosis requiresseveral simultaneous approaches: surgical interven-tion, antifungal therapy and correction of the under-lying predisposing condition. Surgical debridement ofgrossly necrotic tissue is always required; in addition,antifungal agents such as Amphotericin B/posacona-zole should be coadministered. Hyperbaric oxygen,Granulocyle colony stimulating factor and interferon-g might give some benefit, as adjunctive treatmentand their role require further evaluation.4,6

Continued expansion of the wound despite broad-spectrum antibiotic therapy, failure to isolate bacterialorganisms and demonstration of ribbon like, aseptatehyphae in tissue section are some of features that arehelpful in the early diagnosis of NF of fungal etiology.Rapid progression and unacceptably high mortality

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rate has caused many to rethink the importance ofzygomycosis, and clinicians should be cautious intreating patients with NF because all the time, thesecases may not be caused by bacterial infection.

Atul Garg1, MD, DNBSistla Sujatha1, MD

Jaya Garg1, MD, DNBSistla Sarath Chandra2, MS

Debdatta Basu3, MD;Subhash Chandra Parija1, MD, PhD, FRCPath

1Department of Microbiology, 2Department ofSurgery, 3Department of Pathology Jawaharlal

Institute of Postgraduate Medical Education andResearch (JIPMER), Pondicherry, India

E-mail: [email protected]

References

1. Stone DR, Gorbach SL. Necrotizing fasciitis. The changing

spectrum. Dermatol Clin 1997; 15: 213.

2. Schwartz RA, Kapila R. Necrotizing fasciitis. from e-medicine

avilable at www.emedicine.com/DERM/topic743.htm accessed

on 15/5/2008.

3. Warnock DW, Richardson MD. Fungal infection in the

compromised patient, 2nd ed. John Wiley & Sons, New york

1991.

4. Ellis DH. Systemic zygomycosis. In Merz WG, Hay RJ, eds.

Topley and Wilson’s microbiology and microbial infections.

Medical mycology, 10th ed. London: Arnold, 2005; 659.

5. Jain D, Kumar Y, Vasishta RK, Logasundaram R, Pattari SK,

Chakrabarti A. Zygomycotic necrotizing fasciitis in immuno-

competent patients: a series of 18 cases. Mod Pathol 2006;

19: 1221.

6. Petrikkos G, Skiada A. Recent advances in antifungal chemo-

therapy. Int J Antimicrob Agents 2007; 18: 108.

Fig. 1. A) Necrotizing fasciitis involving the

right gluteal region, upper part of right thigh

and lower part of right back. B) KOH wet

mount showed broad, nonseptate hyphae with

right-angle branching, characteristic of zygo-

mycetes (3400). C) Hematoxylin and eosin

stained section showing irregular hyphae of

varying width surrounded by eosinophilic

sheath, showing splendore-hoeppli phenome-

non (3400). D) Periodic acid-Schiff stain

section showing irregular hyphae of varying

width (3400).

Letter to the Editor

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