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  Table of Contents    
CASE REPORT  
Year : 2019  |  Volume : 62  |  Issue : 4  |  Page : 621-623
A case report of Nocardia cyriacigeorgica presenting as cellulitis with multiple discharging sinuses


1 Department of Microbiology, Dr. S. N. Medical College, Jodhpur, Rajasthan, India
2 Department of Dentistry, Dr. S. N. Medical College, Jodhpur, Rajasthan, India

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Date of Web Publication14-Oct-2019
 

   Abstract 


Nocardia spp. are filamentous Gram positive bacteria that are ubiquitous soil saprophytes. The majority of nocardial infections occur in severely immunocompromised patients who are particularly susceptible to pulmonary disease and dissemination. Extrapulmonary nocardiosis is relatively common and can occur through hematogenous dissemination or a contiguous spread of necrotizing pneumonitis. Primary cutaneous and soft tissue nocardiosis can result from traumatic injury to the skin that involves contamination with soil. After skin inoculation, a superficial abscess or localized cellulitis can develop. Co-trimoxazole is the drug of choice for all types of nocardiosis. We are reporting a case of Nocardia cyriacigeorgica presenting as cellulitis followed tooth extraction.

Keywords: MALDI-TOF MS, Nocardia cyriacigeorgica, nocardiosis, 16S rRNA sequencing

How to cite this article:
Bora A, Rathore L, Khatri PK, Chattopadhyay C S. A case report of Nocardia cyriacigeorgica presenting as cellulitis with multiple discharging sinuses. Indian J Pathol Microbiol 2019;62:621-3

How to cite this URL:
Bora A, Rathore L, Khatri PK, Chattopadhyay C S. A case report of Nocardia cyriacigeorgica presenting as cellulitis with multiple discharging sinuses. Indian J Pathol Microbiol [serial online] 2019 [cited 2019 Nov 18];62:621-3. Available from: http://www.ijpmonline.org/text.asp?2019/62/4/621/269083





   Introduction Top


Nocardiosis occurs worldwide. The disease is more common among adults as compared to children. Nocardia species are most commonly isolated aerobic actinomycetes causing human diseases. Nocardia spp. are finely thin, beaded, filamentous Gram positive and weak acid fast bacteria.[1],[2] Nocardiosis may range from primary skin lesions resulting from trauma-related inoculation in immunocompetent to pulmonary infection through inhalation or CNS infection in immunocompromised individuals. Extrapulmonary infection may occur from lungs through hematogenous route, most common site being the brain. Occurrence of nocardiosis is increasing among immunocompromised and immunocompetent individuals as well.[3] Nocardia species that have been reported in extrapulmonary cases are Nocardia cyriacigeorgica, Nocardia nova, Nocardia farcinica and Nocardia asteroids.[1] Here, we report a case of extrapulmonary nocardial infection caused by Nocardia cyriacigeorgica presenting as cellulitis with multiple purulent discharging sinuses over submandibular region following tooth extraction.


   Case History Top


A 45-years old male who is a resident of Rajasthan, India, presented to dental OPD of our tertiary care hospital with chief complaints of pain, intraoral ulceration, left-sided swelling from molar to canine tooth. The patient had multiple purulent discharging sinuses over left chin and was unable to fully open his mouth for last 7–10 days. Patient's son gave history of left molar tooth extraction four months back which was done by an unauthorised medical practitioner. After tooth extraction, patient developed severe pain and intraoral ulceration with intraoral pustular swelling within 1–2 weeks. There was complaint of delayed healing along with intermittent pain, intraoral swelling, and abscess formation. Since then, the patient made multiple visits to several dental practitioners and was prescribed nonsteroidal anti-inflammatory drugs and antibiotics without any obvious clinical diagnosis, but the patient's symptoms did not improve. Before presenting to our OPD, the patient gave history of extraction of right lower canine tooth about 15 days back done by the same unauthorised medical practitioner. The patient denied any respiratory complaints including cough, shortness of breath, or pleuritic chest pain. The patient was farmer by profession. There was no history of diabetes mellitus, hypertension, or any chronic illness. The patient gave history of tobacco abuse for last 20 years. There was no family history of similar disease.

On examination, there were multiple purulent discharging sinuses over submandibular region. Intra-oral examination showed multiple erythematous ulcerative lesions with pustular swelling in lower jaw. Patient was nonreactive for HIV, HBV, and HCV viral markers. Based on the clinical features, diagnosis of facial actinomycosis was made by the clinician.

Orthopantomogram (OPG) was advised which showed bony erosion in left mandible near the 3rd molar and multiple teeth missing in both the jaws. Chest radiographs at presentation showed no evidence of pulmonary involvement. Direct smear in Gram's stain showed Gram-positive beaded thin filaments that were weak acid fast on modified Z-N staining with 1% sulfuric acid decolorization. Culture was carried out using standard microbiological techniques. After 72 hours, the culture showed growth of pearly white dry colonies on sheep blood agar. On staining, thin, beaded Gram positive, weak acid fast filamentous bacteria were seen [Figure 1]. The isolate was identified as Nocardia cyriacigeorgica by MALDI-TOF MS. For definite species level, the isolate was sent to the National Culture Collection for Pathogenic Fungi (NCCPF), PGI, Chandigarh. The isolate was identified as Nocardia cyriacigeorgica by 16S rRNA PCR followed by sequencing [Supplementary File]. The GenBank accession numbers for the 16S rRNA gene of Nocardia cyriacigeorgica determined in this study is MK642278-MK642279.
Figure 1: (a) Patient presents with multiple discharging sinuses. (b) Orthopantomogram shows multiple missing teeth in both the jaws without any obvious bony involvement. (c) Direct smear in Gram stain showing Gram-positive beaded thin filaments. (d) Modified Z-N staining with 1% sulfuric acid decolorization shows weak acid fast filamentous bacteria. (e) Pearly white dry colonies on sheep blood agar

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The patient was prescribed oral co-trimoxazole for four months duration and was called for weekly follow up in the first month and then once monthly. After completion of full course for four months, the patient was free of signs and symptoms of nocardiosis.


   Discussion Top


Nocardia spp. are filamentous Gram positive bacteria causing majority of infections in severely immunocompromised patients who are particularly susceptible to pulmonary disease and dissemination. Most systemic diseases involves Nocardia cyriacigeorgica, N. farcinica, N. pseudobrasiliensis and species in N. transvalensis and N. nova complexes.[2] Extrapulmonary nocardiosis is relatively common and can be contracted through hematogenous dissemination or by a contiguous spread of necrotizing pneumonitis. Primary cutaneous and soft tissue nocardiosis can result from traumatic inoculation that involves contamination with soil. Infection caused by Nocardia brasiliensis is usually limited to the skin.[2],[4] Nocardiosis after tooth extraction has previously been reported from India.[5] There are several case reports of Nocardia spp. causing extrapulmonary nocardiosis, involving skin, oral cavity, CNS, tibia, femur, sacrum and disseminated infection have also been reported.[5],[6],[7],[8],[9],[10] Our case is an example of trauma-associated inoculation of Nocardia, leading to cellulitis presenting as discharging sinuses.

Since the development of genotypic methods like 16S rRNA sequencing, most clinical isolates of nocardia are being identified to be Nocardia cyriacigeorgica.[11] To our knowledge, this is the first case report of Nocardia cyriacigeorgica from Rajasthan, India. Nocardiosis involving skin and soft tissue is not common, therefore this infection can be easily overlooked and misdiagnosed as other common bacterial infections.[12] Differential diagnosis may include actinomycetoma, eumycetoma, and botryomycosis. Gram staining is the most useful microbiological technique for visualizing and recognizing nocardia species and ruling out other etiologies. Diagnosis of nocardia to species level can only be confirmed by microbiological techniques such as MALDI-TOF MS and 16S rRNA sequencing.[2],[13]

In general, the antibacterial agents that are most effective against Nocardia spp. are the sulfonamides, amikacin, minocycline, imipenem, clarithromycin, ampicillin, amoxicillin-clavulanic acid, ceftriaxone and linezolid. Co-trimoxazole is effective against all species but the effectiveness of other antibiotics may vary among different Nocardia species. Therefore, it is important to distinguish between nocardia species because of the intrinsic resistance to certain antimicrobial agents in some species.[3] Methods for detection of antimicrobial susceptibility are available in Clinical Laboratory Standard Institute (CLSI) guidelines and should be performed in geographical areas showing higher rates of treatment failures. There are studies showing development of antimicrobial resistance against sulfonamides, imipenem, and tedizolid in nocardia species. Therefore, susceptibility testing should be performed when required.[14],[15],[16]

Initial treatment for presumed nocardiosis is generally empiric due to the fact that many laboratories may not be performing susceptibility testing. In general, the antibacterial agent most active against Nocardia spp. is sulphonamides. Co-trimoxazole is equally or more efficacious than other antibacterials and is the treatment of choice for nocardiosis with the best alternative being minocycline. In patients with sulfa drug allergy, desensitization allows continuation of therapy. Duration of treatment depends upon clinical presentation. In pulmonary nocardiosis, duration of treatment is 6–12 months. In cellulitis, the duration of treatment is 2 months. In AIDS patients with CD4+ T-lymphocyte count <200/μl or with chronic granulomatous disease, therapy must be continued indefinitely.[3]

In conclusion, intraoral cellulitis after tooth extraction may be caused by Nocardia cyriacigeorgica. This case is also an example of how a medical intervention by unauthorized persons can expose patients to severe infections including nocardiosis even in immunocompetent individuals.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Osoagbaka OU, Njoku-Obi AN. Presumptive diagnosis of pulmonary nocardiosis: Value of sputum microscopy. J Appl Bacteriol 1987;63:27-38.  Back to cited text no. 1
    
2.
Filice GA. Nocardiosis in persons with human immunodeficiency virus infection, transplant recipients, and large, geographically defined populations. J Lab Clin Med 2005;145:156-62.  Back to cited text no. 2
    
3.
Brown-Elliott BA, Brown JM, Conville PS, Wallace RJ Jr. Clinical and laboratory features of the Nocardia spp. based on current molecular taxonomy. Clin Microbiol Rev 2006;19:259-82.  Back to cited text no. 3
    
4.
Beaman BL, Beaman L. Nocardia species: Host-parasite relationships. Clin Microbiol Rev 1994;7:213-64.  Back to cited text no. 4
    
5.
Beena, Raja R, Sandeep, Karur K, Prakash PY, Indumathi VA. Nocardia in buccal space abscess - An oral manifestation: A case report. Int J Health Sci Res 2014;4:238-41.  Back to cited text no. 5
    
6.
Raszka D, Popelka S, Heřt J, Jahoda D, Landor I, Vavřík P. Rare case of osteomyelitis of tibial shaft caused by Nocardia cyriacigeorgica. Folia Microbiol (Praha) 2018;63:525-32.  Back to cited text no. 6
    
7.
Tucer B, Yildiz O, Akdemir H, Menk A, Kurtsoy A. Nocardia brain abscess mimicking brain tumour in immunocompetent patients: Report of two cases and review of the literature. Acta Neurochir (Wien) 2004;146:411-4.  Back to cited text no. 7
    
8.
George SJ, Rivera AM, Hsu S. Disseminated cutaneous nocardiosis mimicking cellulitis and erythema nodosum. Dermatol Online J 2006;12:13.  Back to cited text no. 8
    
9.
De Nardo P, Giancola ML, Noto S, Gentilotti E, Ghirga P, Tommasi C, et al. Left thigh phlegmon caused by Nocardia farcinica identified by 16S rRNA sequencing in a patient with Leprosy: A case report. BMC Infect Dis 2013;13:162.  Back to cited text no. 9
    
10.
Lee TG, Jin WJ, Jeong WS, Moon SH, Kwon TG, Lee SK, et al. Primary cutaneous nocardiosis caused by Nocardia takedensis. Ann Dermatol 2017;29:471-5.  Back to cited text no. 10
    
11.
Schlaberg R, Huard RC, Della-Latta P. Nocardia cyriacigeorgica, an emerging pathogen in the United States. J Clin Microbiol 2008;46:265-73.  Back to cited text no. 11
    
12.
Camozzota C, Goldman A, Tchernev G, Lotti T, Wollina U. A primary cutaneous nocardiosis of the hand. Open Access Maced J Med Sci 2017;5:470-2.  Back to cited text no. 12
    
13.
Verroken A, Janssens M, Berhin C, Bogaerts P, Huang TD, Wauters G, et al. Evaluation of matrix-assisted laser desorption ionization-time of flight mass spectrometry for identification of nocardia species. J Clin Microbiol 2010;48:4015-21.  Back to cited text no. 13
    
14.
Brown-Elliott BA, Biehle J, Conville PS, Cohen S, Saubolle M, Sussland D, et al. Sulfonamide resistance in isolates of Nocardia spp. from a US multicenter survey. J Clin Microbiol 2012;50:670-2.  Back to cited text no. 14
    
15.
Brown-Elliott BA, Wallace RJ. In vitro susceptibility testing of tedizolid against isolates of Nocardia. Antimicrob Agents Chemother 2017;61:1-5.  Back to cited text no. 15
    
16.
Hagerman A, Rodríguez-Nava V, Boiron P, Crisinel PA, Posfay-Barbe KM. Imipenem-resistant Nocardia cyriacigeorgica infection in a child with chronic granulomatous disease. J Clin Microbiol 2011;49:1185-7.  Back to cited text no. 16
    

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Correspondence Address:
Laxmi Rathore
Department of Microbiology, Dr. S. N. Medical College, Jodhpur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_714_18

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