|Year : 2016 | Volume
| Issue : 3 | Page : 377-381
An unusual case of chronic nonhealing periorbital ulceration due to a new species of Corynebacterium sp. strain UCL557
Bipasa Chakraborty1, Kalidas Rit1, Rupali Dey1, Satadal Das2, Prasanta Kumar Maiti1
1 Department of Microbiology, Institute of Post-Graduate Medical Education and Research, Kolkata, West Bengal, India
2 Peerless Hospital and B K Roy Research Centre, Kolkata, West Bengal, India
|Date of Web Publication||17-May-2016|
70B T.C. Mukherjee Street, Rishra, Hooghly - 712 248, West Bengal
Source of Support: None, Conflict of Interest: None
Nondiphtherial Corynebacterium (diphtheroids) has been related to blood and wound infections but are an uncommon cause for soft tissue infection. We report a case of periorbital soft tissue infection with ulceration caused by multidrug-resistant Corynebacterium spp. in a 9-year-old girl who is apparently immunocompetant. Computed tomography scan showed soft tissue involvement of right periorbital region with no bony destructions or focal calcifications. Vision remained unaffected. Patient was treated by debridement and skin grafting, but condition did not improve. Pus collected from the periorbital ulcerated area was cultured in blood agar and Corynebacterium spp. was isolated from the pure culture, which was identified as a new species Corynebacterium sp. strain UCL557 using 16S rDNA- based molecular technique based on nucleotide homology and phylogenetic analysis. Antibiogram showed multiresistance pattern with sensitivity to ceftriaxone-sulbactum vancomycin and linezolid. After initiation of treatment with vancomycin infusion and oral linezolid, the patient responded well and lesion started to heal. To the best of our knowledge, this is the first ever case report of periorbital ulceration by new species of Corynebacterium sp. strain UCL557.
Keywords: Corynebacterium UCL557 , diphtheroids, multidrug-resistant, periorbital ulceration
|How to cite this article:|
Chakraborty B, Rit K, Dey R, Das S, Maiti PK. An unusual case of chronic nonhealing periorbital ulceration due to a new species of Corynebacterium sp. strain UCL557. Med J DY Patil Univ 2016;9:377-81
| Introduction|| |
The most prominent member of genus Corynebacterium is Corynebacterium diphtheriae. Recently, interest in diphtheroids have increased due to increasing frequency in isolation as nosocomial pathogen, , and emergence of multidrug-resistance. ,, Diphtheroids are often present as skin commensals. Hence establishing it as the etiologic agent, ruling out skin contamination is difficult.
Pediatric preseptal (periorbital) infections may develop from either exogenous sources (breaches in skin barrier) or endogenous sources (paranasal sinusitis).  Most common pathogens of these infections are Staphylococcus aureus, followed by Streptococcus species, and Haemophilus influenzae.  However, none of the current literature from India revealed periorbital infection due to any diphtheroid. We report a case of chronic, nonhealing periorbital ulceration caused by multidrug-resistant new species of Corynebacterium UCL557 in an apparently immunocompetent girl, with grade-I malnutrition (ICMR grading).
| Case Report|| |
A 9-year-old girl, from a rural background, presented with chronic periorbital ulceration of right side with profuse purulent discharge for 6 months. The patient initially had a small pustule on inner canthus of right eye 6 months ago. After 3 days, purulent discharge started and the lesion increased. The whole periorbital region was infected with profuse purulent discharge within 1 month, despite oral antibiotics. The patient was referred to tertiary care hospital. On admission, she had pain but no loss of vision, with low-grade fever, body temperature 100°F, pulse rate 92 beats/min, and respiratory rate 22/min. She is from low socioeconomic background, unimmunized, having grade-I malnutrition, but no history of weight loss during this period. She had no history of putting kajal or significant eye injury. Examination revealed, grossly edematous periorbital area, erythematous, thickened lateral canthus, with ulceration and purulent discharge from both canthi, eyelids and upper cheek of the right side with scarring of eyelids and loss of eyelashes due to blepharitis [Figure 1]. No regional lymphadenopathy detected. Tests for visual acuity were normal. Laboratory examinations revealed total count 10.95 × 10 9 /L, 66% neutrophils, hemoglobin 10.5 g/dl, and erythrocyte sedimentation rate 55 mm in 1 st h. Sugar, urea, and creatinine were normal. She had no history of recurrent infections and was nonreactive for HIV and HBsAg. Mantoux test was negative with normal chest roentgenogram and electrocardiography. The patient was treated with debridement and skin grafting. Piperacillin-tazobactum and erythromycin were administered, but infection increased and spread to the opposite side. Contrast-enhanced computed topography of orbit showed only heterogeneously enhancing soft tissue mass density at the root of the nose, extending from lateral canthus of the right eye up to preseptal portion of left eye inner canthus, and suggesting soft tissue infection. There was no extraocular muscles or bony involvement or focal calcifications [Figure 2].
|Figure 2: Contrast-enhanced computed tomography orbit shows periorbital soft tissue infection of right side (arrow)|
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Pus was again collected from deeper parts of ulcer and cultured in 5% sheep's blood agar (HiMedia), Sabouraud dextrose agar (HiMedia), Lowenstein-Jensen medium, and anaerobic Gaspak. A direct smear of pus on Gram's staining showed Gram-positive bacilli in "V" pattern with plenty of pus cells [Figure 3]. Alberts stain showed green bacilli with violet metachromatic granules, in Chinese letter and palisade arrangements. On Ziehl-Neelsen staining, no acid-fast bacilli detected. Cultures in blood agar showed pure growth of small, dry, whitish, and flat colonies. Gram-staining demonstrated similar Gram-positive Corynebacterium with typical V forms, or Chinese letters. To exclude skin contamination, again pus was collected from the ulceration and again isolated Corynebacterium, which was phenotypically found to be similar to Corynebacterium amycolatum. It was nonmotile and important biochemical tests results were catalase positive, glucose and sucrose fermenting, mannitol nonfermenting, no growth in citrate, nitrate reduction positive, PYR test positive, oxidase, esculin, urease test negative, alkaline phosphatase, and pyrazinamidase positive.  For further confirmation, sample was sent for genetic analysis by molecular methods using 16S rDNA-based molecular technique done by Xcelris Labs Ltd.The result was the isolation of a completely new species of Corynebacterium UCL557 (GenBank Accession Number: AJ012838.1) by 16S r DNA amplification technique based on nucleotide homology and phylogenetic analysis [Figure 4]a and 4b. No growth of organisms detected in anaerobic culture, SDA or LJ medium.
|Figure 3: Gram's stain of pus shows Gram-positive bacilli in "V" pattern or Chinese letter (arrow) along with pus cells (×1000)|
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The antimicrobial susceptibility test was done using Kirby-Bauer disc diffusion method on MuellerHinton agar (HiMedia) with 5% sheep blood.  The following antibiotic discs were used for testing: Ampicillin (10 μg), amoxicillin-clavulanic acid (20/10 μg), cefoxitin (30 μg), ceftriaxone (30 μg), ceftriaxone-sulbactum (30/10 μg), vancomycin (30 μg), chloramphenicol (30 μg), clindamycin (2 μg), erythromycin (15 μg), azithromycin (15 μg), ciprofloxacin (5 μg), levofloxacin (5 μg), gentamicin (10 μg), tetracycline (30 μg), doxycycline (30 μg), and linezolid (30 μg). The culture was sensitive to ceftriaxone-sulbactum vancomycin and linezolid.
She was treated with vancomycin infusion, 250 mg intravenous 12 hourly for 7 days and linezolid 300 mg orally 12 hourly. The condition of the patient improved. She became afebrile, pain reduced, and lesion started to heal. Follow-up after 2 weeks revealed the reduced inflammation with no purulent discharge from the lesion [Figure 5].
| Discussion|| |
The pathogenic potential of Corynebacterium has been underestimated and reported as skin contaminants. This is likely to be true when there is single, isolated positive culture, but repeated pure cultures from samples taken aseptically indicate true infection. In this case, heavy pure growth was obtained on culture on two different occasions from the ulcer, indicating the pathogenic role of Corynebacterium UCL557. Swabs were also taken from skin and fingertips of the patient as well as her mother, father, and grandfather along with their conjunctival swabs but did not grow any Corynebacterium. Furthermore no other parts of the body was affected, by Corynebacterium infection. Reports of pathogenic diphtheroids are infrequent and reported in immunocompromised person,  but in immunocompetent person reports are very rare even worldwide.  Diphtheroids species were often misidentified between C. amycolatum, Corynebacterium striatum, Corynebacterium xerosis and Corynebacterium minutissimum, for many years,  until recently due to better speciation methods. However, to our knowledge diphtheroids causing periorbital soft tissue ulceration has not been reported until date. To investigate the taxonomic affinities of the unidentified strain in more detail, a large fragment (1127 bases) of its 16S rDNA gene sequence was determined. Sequence database searches revealed that the strain was most closely related to the Actinobacteria, with species of the genus Corynebacterium exhibiting the highest sequence similarities. Phylogenetic tree confirmed the placement of the pathogenic bacterium within the genus Corynebacterium, with the unknown organism forming a distinct subline with Corynebacterium AJ012838.1 at its nearest position [Figure 4]a depicts the position of the unknown bacterium within a subset of Corynebacterium species. Both phenotypic and molecular phylogenetic data show unequivocally that the unidentified bacterium is a member of the genus Corynebacterium, hence a new species Corynebacterium UCL557. ,,,
Moreover, there are no proper Clinical and Laboratory Standards Institute guidelines for antibiogram of diphtheroids. Antibiogram using disc diffusion method showed multi-resistance pattern. Like reports of many others , it showed sensitivity to vancomycin and linezolid.
This also conveys a message that diphtheroids in clinical isolates should not be neglected as nonpathogenic skin contaminants without confirmation. They cause opportunistic infection, also infection in an apparently immunocompetant individual at any uncommon site, are multidrug-resistant with 100% vancomycin sensitivity. , Proper identification and appropriate treatment at the earliest is necessary to prevent complications by diphtheroid infections.
We express our sincere thanks to Prof Bijay Kumar Majumdar, Professor and HOD, Department of Plastic and Reconstructive surgery for his valuable help in giving us patient's clinical data.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]