Indian Journal of Medical Microbiology IAMM  | About us |  Subscription |  e-Alerts  | Feedback |  Login   
  Print this page Email this page   Small font sizeDefault font sizeIncrease font size
 Home | Ahead of Print | Current Issue | Archives | Search | Instructions  
Users Online: 1678 Official Publication of Indian Association of Medical Microbiologists 
  Search
 
  
 ~  Similar in PUBMED
 ~  Search Pubmed for
 ~  Search in Google Scholar for
 ~Related articles
 ~  Article in PDF (1,163 KB)
 ~  Citation Manager
 ~  Access Statistics
 ~  Reader Comments
 ~  Email Alert *
 ~  Add to My List *
* Registration required (free)  

 
 ~  Abstract
 ~ Introduction
 ~ Case Report
 ~ Discussion
 ~ Conclusion
 ~  References
 ~  Article Figures
 ~  Article Tables

 Article Access Statistics
    Viewed2490    
    Printed71    
    Emailed6    
    PDF Downloaded120    
    Comments [Add]    
    Cited by others 1    

Recommend this journal

 


 
  Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 32  |  Issue : 4  |  Page : 448-451
 

A rare case of subcutaneous mucormycosis due to Syncephalastrum racemosum: Case report and review of literature


1 Department of Internal Medicine, Maharaja Krishna Chandra Gajapati Medical College, Berhampur, Odisha, India
2 Department of Pathology, Maharaja Krishna Chandra Gajapati Medical College, Berhampur, Odisha, India
3 Department of Microbiology, Maharaja Krishna Chandra Gajapati Medical College, Berhampur, Odisha, India

Date of Submission03-Sep-2013
Date of Acceptance06-Feb-2014
Date of Web Publication4-Oct-2014

Correspondence Address:
S Mangaraj
Department of Internal Medicine, Maharaja Krishna Chandra Gajapati Medical College, Berhampur, Odisha
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0255-0857.142252

Rights and Permissions

 ~ Abstract 

Zygomycosis represent a group of uncommon but potentially fatal fungal infections. The incidence of zygomycosis has increased manifold in recent years. Despite aggressive treatment, it can lead to a highly invasive disease state with fatal outcomes, especially among immuno-compromised. Syncephalastrum racemosum is a fungus belonging to Zygomycetes. Very few cases of human disease caused by this particular fungus have been documented. However, it has been clearly implicated in causing highly invasive disease in recent reported cases. Knowledge about the pathogenicity and clinical presentation of this rare fungal infection will alert the clinicians for instituting an early appropriate therapy leading to better outcomes.


Keywords: Mucormycosis, Subcutaneous mucormycosis,Syncephalastrum racemosum


How to cite this article:
Mangaraj S, Sethy G, Patro M K, Padhi S. A rare case of subcutaneous mucormycosis due to Syncephalastrum racemosum: Case report and review of literature . Indian J Med Microbiol 2014;32:448-51

How to cite this URL:
Mangaraj S, Sethy G, Patro M K, Padhi S. A rare case of subcutaneous mucormycosis due to Syncephalastrum racemosum: Case report and review of literature . Indian J Med Microbiol [serial online] 2014 [cited 2019 Nov 14];32:448-51. Available from: http://www.ijmm.org/text.asp?2014/32/4/448/142252



 ~ Introduction Top


Mucormycosis (Zygomycosis) represent a spectrum of emerging infections caused by ubiquitous fungal pathogens. S. racemosum is a fungus belonging to order Mucorales, which has very rarely been implicated in causing human disease. Less than 10 cases have been reported in medical literature. We report a case of an adult diabetic male who developed locally invasive cutaneous disease due to S. racemosum.


 ~ Case Report Top


A 45-year-old male, a farmer, presented to us with development of a swelling in anterior chest wall in the left side during a routine follow up. There was no history of fever, cough, haemoptysis, weight loss, headache, altered sensorium, nasal discharge, visual problem or vomiting. There was no history of any trauma to the affected site. His significant past history included an episode of diabetic ketoacidosis 2 months back, which was successfully managed at our unit. The patient was a diagnosed case of type 2 diabetes mellitus for past 1 year and was on oral hypoglycaemic drugs. Despite repeated advice, his adherence to medication was poor. The patient was normotensive. There was no history of tuberculosis in the past.

On examination, the patient was conscious and afebrile. He had a blood pressure of 130/80 mmHg and a respiratory rate of 16/min. There was no evidence of pallor, icterus, clubbing or lymphadenopathy. Systemic examination was unremarkable. There was a solitary swelling of about 3 × 2 cm in the left side of anterior chest wall. The lesion was firm, non-fluctuant, adherent to underlying fascia and was slightly tender. There were no local signs of inflammation or any evidence of necrotic tissue or discharging sinus. The swelling had developed gradually over a period of 1 month.

His routine blood investigation including complete blood count, renal function test, liver function test, electrolyte panel all were within normal limit. Fasting and post-prandial blood glucose levels were 160 and 240 mg/dl, respectively. He had a glycosylated haemoglobin level of 9% (normal <7%). Urine examination was normal. There was no evidence of blood ketonemia or ketonuria. Serologic tests for human immunodeficiency virus (HIV), Hepatitis B and C were negative.

Fine needle aspiration (FNA) from the swelling was done and purulent material was obtained. One part of the aspirate was used for cytological evaluation and the second part was kept for culture study. Cytosmear revealed presence of branching septate fungal hyphae with plenty of macrophages, polymorphs and oeosinophils over a dirty necrotic background, suggesting tissue invasion [Figure 1]. Culture growth on Sabouraud's dextrose agar without cycloheximide revealed abundant, erected mycelium (around 0.5 cm tall) within 3 days. The surface colour of the colony was at first white to yellow; after a few days the centre turned black [Figure 2]. There was no growth on the Sabouraud's agar supplemented with cycloheximide. Lactophenol Cotton Blue (LCB) stain showed wide hyphae with sparse septae and sporangiophores terminated in swollen vesicles with radial merosporangiae filled with spores [Figure 3]. No growth of any other organism was seen in the culture. A repeat aspirate was sent for culture and cytological study (from a close but different site of the swelling), which also confirmed presence of above-said fungus. Blood and urine cultures for bacteria and fungi were sterile. A diagnosis of subcutaneous zygomycosis due to S. racemosum was made. A computed tomography (CT) scan of chest was under taken to look for invasion of the infection and pulmonary involvement. It showed a discrete subcutaneous mass of size 33.6 × 17.7 mm in the chest wall [Figure 4]. However, it had not invaded the underlying ribs. There was no evidence of pulmonary disease. Realising the risk of invasive mucormycosis especially rhino cerebral form in diabetics, a CT scan of paranasal sinuses was done to rule out occult infection. However, it did not show any evidence of growth. Also, nasal swab cultures did not grow any fungi.
Figure 1: Cytosmear showing branching fungal hyphae in a dirty necrotic background. [Diff Quik, ×400]

Click here to view
Figure 2: White colour colony with a necrotic black centre (wooly to cotton-candy like texture) on SDA culture medium

Click here to view
Figure 3: Microscopic examination showing wide hyphae with sparse septae and sporangiophores terminated in swollen vesicles with radial merosporangiae filled with spores.[LPCB stain, ×400]

Click here to view
Figure 4: CT scan showing discrete subcutaneous growth in anterior chest wall

Click here to view


He was put on intensive short acting insulin regimen to achieve optimum glycaemic control. As there was high risk of invasive mucormycosis and dissemination from primary site, a surgical debridement was done and infected tissue was removed. He was put on intravenous Liposomal Amphotericin B (5 mg/kg) and broad spectrum antibiotics to prevent superadded bacterial infection. He was treated conservatively for a period of 10 days with the above agents. He made uneventful recovery with healthy wound healing.


 ~ Discussion Top


Zygomycosis can be divided into primarily six types - rhino cerebral, pulmonary, cutaneous, gastrointestinal, disseminated and miscellaneous. [1] According to a review by Roden et al., cutaneous zygomycosis represented the third most common (19%) form of zygomycosis after rhino-orbito-cerebral (39%) and pulmonary forms (24%). [1] Depending on the extent of the infection, cutaneous zygomycosis is classified as localised when it affects only the skin or subcutaneous tissue; deep extension when it invades muscle, tendons or bone; and disseminated when it involves other non-contiguous organs. [2] The cutaneous zygomycosis may be primary by direct inoculation in skin or secondary to dissemination from a distant focus seeding the bloodstream. The risk factors for zygomycosis include diabetes mellitus, neutropaenia, sustained immunosuppressive therapy, chronic prednisone use, iron chelation therapy, broad-spectrum antibiotic use, severe malnutrition and primary breakdown in the integrity of the cutaneous barrier such as trauma, surgical wounds, needle sticks or burns. [3]

Cutaneous zygomycosis may be gradual and slowly progressive or may be aggressive and fulminant leading to necrotizing lesions and haematogenous dissemination. [2],[4] It usually manifests as a necrotic eschar with surrounding oedema and induration, but can have myriad of presentations. Clinical presentations of cutaneous zygomycosis described by authors include dark yellow, nodular lesions; black discoloration with surrounding oedema; superficial lesions having only slightly elevated circinate and squamous borders resembling tinea corporis; targetoid plaques with outer erythematous rim. [2],[4] Our case presented with a slowly growing and slightly tender subcutaneous swelling.

S. racemosum is a ubiquitous saprophytic fungi belonging to order Mucorales with very low pathogenicity. It was until recently debated whether this fungus can cause disease in humans, but later it was considered an opportunistic pathogen. [3] Cultures are hyaline, with surface coloration varying from nearly white to various shades of green, olive and grey to almost black. [3] The vegetative mycelium is aseptate. Sporulation occurs readily on routine medium at room temperature and at temperatures above 37°C. There have been only less than 10 documented cases of Syncephalastrum infection in humans till date. We have provided a list of all published cases till now [5],[6],[7],[8],[9],[10],[11] [Table 1] (search word - S. racemosum and zygomycosis).
Table 1: Cases reports of Syncephalastrum racemosum infection in humans

Click here to view


The most critical part of diagnosing a case of zygomycosis is the suspicion of the entity by the clinician. The increase in detection rates of zygomycosis in recent decades is due to better awareness among clinicians coupled with improved techniques of fungal detection. Early diagnosis and prompt management holds the key for successful outcome. All efforts should be made to correct the underlying and predisposing factor. Surgical debridement should be done at the earliest to remove maximum amount of devitalised tissue that is possible. Intravenous amphotericin B (liposomal) is the main stay of therapy. It is given at a dose of 5-10 mg/kg/day and the duration depends on the clinical response. A dose of at least 2 g is necessary in most cases. [6] Posconazole [12] has emerged as an alternative salvage therapy with good clinical response. Diagnosis at the early stage of the disease is pivotal as mortality rate of 94% is seen among patients who develop disseminated disease as a consequence to primary cutaneous disease. [1]


 ~ Conclusion Top


Our case further strengthens and establishes Syncephalastrum as an aetiologic agent for zygomycosis. Mucuromycosis represent an uncommon but life-threatening infection in immuno-compromised, especially diabetics. Prompt treatment that includes correction of underlying condition, surgical debridement and intensive antifungal therapy form the cornerstone of successful management. Untreated, these agents can cause highly invasive and fatal disease. Patients having diabetes, organ transplants, haematopoetic stem cell transplant and AIDS are especially prone for contracting these diseases. The possibility of these atypical infections should always be borne in mind while dealing with the above-said patient groups.

 
 ~ References Top

1.Roden MM, Zaoutis TE, Buchanan WL, Knudsen TA, Sarkisova TA, Schaufele RL, et al. Epidemiology and outcome of zygomycosis: A report of 929 reported cases. Clin Infect Dis 2005;41:634-53.  Back to cited text no. 1
    
2.Petrikkos G, Skiada A, Lortholary O, Roilides E, Walsh TJ, Kontoyiannis DP. Epidemiology and clinical manifestations of mucormycosis. Clin Infect Dis 2012;54:S23-34.  Back to cited text no. 2
    
3.Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human disease. Clin Microbiol Rev 2000;13:236-301.  Back to cited text no. 3
    
4.Skiada A, Rigopoulos D, Larios G, Petrikkos G, Katsambas A. Global epidemiology of cutaneous zygomycosis. Clin Dermatol 2012;30:628-32.  Back to cited text no. 4
    
5.Kamalam A, Thambiah AS. Cutaneous infection by Syncephalastrum. Sabouraudia 1980;18:19-20.  Back to cited text no. 5
[PUBMED]    
6.Schlebusch S, Looke DF. Intraabdominal zygomycosis caused by Syncephalastrum racemosum infection successfully treated with partial surgical debridement and high-dose amphotericin B lipid complex. J Clin Microbiol 2005;43:5825-7.  Back to cited text no. 6
    
7.Pavloviæ MD, Bulajiæ N. Great toenail onychomycosis caused by Syncephalastrum racemosum . Dermatol Online J 2006;12:7.  Back to cited text no. 7
    
8.Baradkar VP, Mathur M, Panda M, Kumar S. Sino-orbital infection by Syncephalastrum racemosum in chronic hepatorenal disease. J Oral Maxillofac Pathol 2008;12:45-7.  Back to cited text no. 8
  Medknow Journal  
9.Amatya R, Khanal B, Rijal A. Syncephalastrum species producing mycetoma-like lesions. Indian J Dermatol Venereol Leprol 2010;76:284-6.  Back to cited text no. 9
[PUBMED]  Medknow Journal  
10.Ramesh V, Ramam M, Capoor MR, Sugandhan S, Dhawan J, Khanna G. Subcutaneous zygomycosis: Report of 10 cases from two institutions in North India. J Eur Acad Dermatol Venereol 2010;24:1220-5.  Back to cited text no. 10
    
11.Mathuram AJ, Mohanraj P, Mathews MS. Rhino-orbital-cerebral infection by Syncephalastrum racemosusm. J Assoc Physicians India 2013;61:339-40.  Back to cited text no. 11
    
12.Rogers TR. Treatment of zygomycosis: Current and new options. J Antimicrob Chemother 2008;61:i35-40.  Back to cited text no. 12
[PUBMED]    


    Figures

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

  [Table 1]

This article has been cited by
1 Mucormycosis in a Non-Hodgkin Lymphoma Patient Caused by Syncephalastrum racemosum: Case Report and Review of Literature
Georgina Rodríguez-Gutiérrez,Erika M. Carrillo-Casas,Roberto Arenas,Jorge O. García-Méndez,Sonia Toussaint,Mónica E. Moreno-Morales,Adrián A. Schcolnik-Cabrera,Juan Xicohtencatl-Cortes,Rigoberto Hernández-Castro
Mycopathologia. 2015;
[Pubmed] | [DOI]



 

Top
Print this article  Email this article
 

    

© 2004 - Indian Journal of Medical Microbiology
Published by Wolters Kluwer - Medknow

Online since April 2001, new site since 1st August '04