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

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.

Full text

www.ijmm.org
448 vol. 32, No. 4Indian Journal of Medical Microbiology
How to cite this article: Tankhiwale SS, Katkar VJ. Subdural empyma
due to Mycobacterium fortuitum in a non-HIV patient. Indian J Med
Microbiol 2014;32:446-8.
Source of Support: Nil, Conict of Interest: None declared.
5. Devi dRG, idumati VA, Indra S, BabuPRS,
Sridharan D, Belwadi MRS. Injection site abscess due
to Mycobacterium fortuitum: Indian. J Med Microbiol
2003;21(2):133-4
6. Sethi S, Sharma M, Ray P, Singh M, GuptaA.Microbacterial
wound infection following leproscopy. Indian J Med Res
2001;113:83-4
7. O` Brien RJ, Geiter L J, snider DE. Epidemiology of
nontubercuious mycobacterial diseases in the United States.
Am review of Respir Dis. 1987;135:1007-14
8. DalovisioJR Panky GA, Wallace RJ, Jones DB. Clinical
usefulness of Amikacin and Doxycycline in the treatment of
infection due to M fortuitum and M. chelonae, Review of Inf
Disease. 1981;3:1068-74
*Corresponding author (email: <drsmangaraj@gmail.com>
Department of Internal Medicine (SM, GS), Department of
Pathology (MKP), Department of Microbiology (SP),
Maharaja Krishna Chandra Gajapati Medical College, Berhampur,
Odisha, India.
Received: 03-09-2013
Accepted: 06-02-2014
A rare case of subcutaneous mucormycosis due to Syncephalastrum racemosum:
Case report and review of literature
*S Mangaraj, G Sethy, MK Patro, S Padhi
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.
Key words: Mucormycosis, Subcutaneous mucormycosis,Syncephalastrum racemosum
Introduction
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
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signicant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
Access this article online
Quick Response Code: Website:
www.ijmm.org
PMID:
***
DOI:
10.4103/0255-0857.142251
[Downloaded free from http://www.ijmm.org on Friday, April 29, 2016, IP: 14.139.214.233]

www.ijmm.org
449October - December 2014 Case Reports
lesion was rm, non‑uctuant, adherent to underlying
fascia and was slightly tender. There were no local signs
of inammation 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immunodeciency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 rst 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 lled 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 conrmed 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.
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
Figure 1: Cytosmear showing branching fungal hyphae in a dirty
necrotic background. [Diff Quik, ×400]
Figure 2: White colour colony with a necrotic black centre (wooly to
cotton-candy like texture) on SDA culture medium
Figure 3: Microscopic examination showing wide hyphae with sparse
septae and sporangiophores terminated in swollen vesicles with radial
merosporangiaelledwithspores.[LPCBstain,×400]
[Downloaded free from http://www.ijmm.org on Friday, April 29, 2016, IP: 14.139.214.233]

www.ijmm.org
450 vol. 32, No. 4Indian Journal of Medical Microbiology
Figure 4: CT scan showing discrete subcutaneous growth in anterior
chest wall
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
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classied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; supercial 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[5-11] [Table 1]
(search word - S. racemosum and zygomycosis).
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.
Table 1: Cases reports of Syncephalastrum racemosum infection in humans
Author Disease site Co-morbidity Treatment
Kamalam and Thambiah[5] Cutaneous
Osteomyelitis
Diabetes mellitus Local debridement
Sclebusch et al.[6] Intra-abdominal Immunocompetent Surgical debridement
amphotericin B
PavlovićandBulajić[7] Onychomycosis Immunocompetent Nail plate avulsion
topical nystatin
Baradkar et al.[8] Rhino-orbito-cerbral Cirrhosis of liver
Chronic hepato-renal disease
Surgical debridement
amphotericin B
Amatya et al.[9] Cutaneous Immunocompetent Not available
Ramesh et al.[10] Subcutaneous Immunocompetent Potassium iodide
itraconazole
Mathuram et al.[11] Rhino-orbito cerebral Diabetes mellitus Amphotericin B
[Downloaded free from http://www.ijmm.org on Friday, April 29, 2016, IP: 14.139.214.233]

www.ijmm.org
451October - December 2014 Case Reports
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
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
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.
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.
3. Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in
human disease. Clin Microbiol Rev 2000;13:236-301.
4. Skiada A, Rigopoulos D, Larios G, Petrikkos G,
Katsambas A. Global epidemiology of cutaneous
zygomycosis. Clin Dermatol 2012;30:628-32.
5. Kamalam A, Thambiah AS. Cutaneous infection by
Syncephalastrum. Sabouraudia 1980;18:19-20.
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.
7. PavlovićMD, BulajićN.Greattoenailonychomycosis caused
by Syncephalastrum racemosum. Dermatol Online J 2006;12:7.
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.
9. Amatya R, Khanal B, Rijal A. Syncephalastrum species
producing mycetoma-like lesions. Indian J Dermatol Venereol
Leprol 2010;76:284-6.
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.
11. Mathuram AJ, Mohanraj P, Mathews MS.
Rhino-orbital-cerebral infection by Syncephalastrum
racemosusm. J Assoc Physicians India 2013;61:339-40.
12. Rogers TR. Treatment of zygomycosis: Current and new
options. J Antimicrob Chemother 2008;61:i35-40.
How to cite this article: Mangaraj S, Sethy G, Patro MK, 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.
Source of Support: Nil, Conict of Interest: None declared.
Access this article online
Quick Response Code: Website:
www.ijmm.org
PMID:
***
DOI:
10.4103/0255-0857.142252
Sphingomonas paucimobilis bacteraemia and shock in a patient with rheumatic
carditis
*Y Yozgat, A Kilic, C Karadeniz, R Ozdemir, O Doksoz, G Guldan, T Mese
Abstract
Acute rheumatic fever (ARF) carditis is treated with steroids, which can cause changes in the cellular immune
response, especially decreased CD3 (+) T cells. Nosocomial infections due to steroid use for treatment of ARF carditis
or secondary to the changes in the cellular immune response have not been reported in the literature. Sphingomonas
paucimobilis is a Gram-negative bacillus causing community- and hospital-acquired infections. It has been reported as
causingbacteraemia/sepsis,pneumoniaorperitonitisinpatientswithmalignancies,immunosuppressionordiabetes.We
[Downloaded free from http://www.ijmm.org on Friday, April 29, 2016, IP: 14.139.214.233]