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Year : 2016  |  Volume : 5  |  Issue : 3  |  Page : 273-275

Chronic tenosynovitis of the hand caused by Mycobacterium heraklionense

1 Department of Infectious Diseases, University of Rochester Medical Center, Rochester, NY, USA
2 Department of Orthopedic Surgery, University of Rochester Medical Center, Rochester, NY, USA
3 Department of Pathology, University of Rochester Medical Center, Rochester, NY, USA

Date of Web Publication13-Feb-2017

Correspondence Address:
Sonal S Munsiff
University of Rochester Medical Center, School of Medicine and Dentistry, 601 Elmwood Avenue, Box 689, Rochester, NY 14642
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Source of Support: None, Conflict of Interest: None

DOI: 10.1016/j.ijmyco.2016.05.005

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Non-tuberculous mycobacteria are increasingly recognized as a cause of infection in both immunocompromised and immunocompetent hosts. Mycobacterium heraklionense is a recently described member of the Mycobacterium terrae complex. Herein we report a case of M. heraklionense chronic flexor tenosynovitis in the hand, managed with surgery and antibiotics.

Keywords: Mycobacterium heraklionense, Tenosynovitis, Non tuberculous mycobacteria

How to cite this article:
Aburjania N, Hammert WC, Bansal M, Boyce BF, Munsiff SS. Chronic tenosynovitis of the hand caused by Mycobacterium heraklionense. Int J Mycobacteriol 2016;5:273-5

How to cite this URL:
Aburjania N, Hammert WC, Bansal M, Boyce BF, Munsiff SS. Chronic tenosynovitis of the hand caused by Mycobacterium heraklionense. Int J Mycobacteriol [serial online] 2016 [cited 2022 Dec 2];5:273-5. Available from: https://www.ijmyco.org/text.asp?2016/5/3/273/200064

  Case report Top

A 72-year-old man, with no notable past medical history was referred to our institution because for 9 months he had gradually worsening of right middle finger pain, swelling, and stiffness. He did not recall any trauma to his hand; however, he did report gardening and picking up golf balls as hobbies. Initial workup at another institution with magnetic resonance imaging done 7 months after the onset of symptoms showed notable inflammatory changes in the flexor tendon sheath. Rheumatoid factor and antinuclear antibody tests were negative. Cortisone injection was given to the affected finger, but produced only temporary symptomatic improvement. Three months prior to his presentation to us he underwent surgical exploration of the inflamed tissue, and histologic analysis of the specimen showed tenosynovitis with granulation tissue formation. Bacterial, fungal, and acid-fast bacilli (AFB) stains were negative. All cultures, including bacterial, fungal, and AFB cultures did not grow any organisms.

Swelling and pain of the hand persisted. Two months later, the patient was seen by one of the authors who noted substantial swelling in the right middle finger extending from the proximal palm to the proximal interphalangeal joint flexion crease. There was a central wound over the proximal phalanx with granulation tissue visible. The patient underwent radical tenosynovectomy of the right middle finger [Figure 1]A,[Figure 1]B,[Figure 1]C due to persistent symptoms. No bacteria were identified on gram-stained smears. Aerobic culture grew 1+methicillin-sensitive Staphylococcus aureus . Occasional AFB were identified after extensive examination of Ziehl–Neelsen-stained smears done at the microbiology laboratory, but AFB did not grow (the sample was incubated at 31 °C and 37 °C using our standard protocol for plating tissue samples from extremities).
Figure 1: Gross images of right middle finger taken at surgery. (A) Preoperative appearance of the right middle finger; (B) exposure of flexor sheath of the right middle finger with thickened tenosynovium; (C) specimen from the right middle finger flexor tendons for submission to surgical pathology following tenosynovectomy. Representative histologic images of inflamed tenosynovium. (D) Numerous necrotizing (black arrow) and nonnecrotizing granulomas with associated chronic inflammation (hematoxylin-eosin, original magnification × 20); (E) lymphocyte-rich epithelioid cell granulomas with Langhans-type giant cells and lymphocyte cuffing (hematoxylin-eosin, original magnification × 200); (F) necrotizing (black arrow) and nonnecrotizing, epithelioid cell granulomas with chronic inflammation (hematoxylin-eosin, original magnification × 100).

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Histologic examination of the tissue showed intense chronic inflammation with numerous necrotizing and nonnecrotizing granulomas with associated Langhans- and Touton-type giant cells and chronic inflammation [Figure 1]D,[Figure 1]E,[Figure 1]F). No fungal organisms were identified in a Gomori methenamine silver-stained tissue section. A fresh, frozen sample was sent to a reference laboratory at the University of Washington Medical Center, Seattle, WA, USA where DNA was extracted from the tissue. The sample tested negative for the Mycobacteria tuberculosis complex using primers for hsp65. A Mycobacterium species most likely related to Mycobacterium heraklionense DNA was detected using an rpoB primer set.

While awaiting the results of AFB culture and polymerase chain reaction, and considering that bacterial culture grew methicillin-sensitive S. aureus , the patient was started empirically on clarithromycin 500 mg twice a day and levofloxacin 750 mg daily. After a month of treatment, when polymerase chain reaction results became available, this regimen was changed to clarithromycin 500 mg twice a day, rifampin 600 mg daily, and ethambutol 1200 mg daily. Clarithromycin was subsequently changed to azithromycin 500 mg daily because of gastrointestinal side effects. The patient improved gradually. On follow-up in our clinic 2 months later, the inflammatory changes appeared diminished. Three months after his initial surgery, the incision was well healed and the swelling had substantially improved. However, he still had diminished motion of his middle finger.

  Discussion Top

Nontuberculous mycobacteria are increasingly recognized as a cause of infection in both immuno-compromised and- competent hosts. M. heraklionense is a member of the Mycobacterium terrae complex that was described recently [1]. M. heraklionense can be associated with substantial morbidity and mortality [2]. However, until recently, there have been very few cases of infection caused by M. heraklionense reported in the literature. In most of the cases in a series by Neonakis et al. [2] the bacteria were isolated from patients' sputum or broncho-alveolar lavage specimens. The majority of these cases, however, were patients with serious underlying diseases, potentially obscuring or confounding a causal link with M. heraklionense .

There have been only a few recently reported cases of a M. heraklionense -associated hand tenosynovitis [3],[4]. M. heraklionense has been one of the major causes of tenosynovitis/osteomyelitis in the US with little change over the past 20 years. However, it was only recently acknowledged as a substantial pathogen [4]. M. heraklionense is very difficult to grow using standard methods for culturing mycobacteria. Using a molecular detection method is likely essential for making a correct diagnosis. M. heraklionense is susceptible to clarithromycin and ethambutol, and resistant to quinolones, rifampicin, sulfamethoxazole, and doxycycline [1]. Treatment guidelines, specifically the duration of treatment, however, have not been established. Once nontuberculous mycobacterial infection of skin and soft tissues is suspected, treatment should be started immediately after a tissue sample is obtained, because the results might not be available until days or weeks later. The most effective empirical regimen is a combination of rifampin, ethambutol, and macrolide (such as clarithromycin) [5]. Because of the diversity of mycobacterial species, accurate identification of the causal organism is very important to guide appropriate chemotherapy.

Abedalthagafi et al. [3] proposed a combination of azithromycin with rifampin and ethambutol for the treatment of their M. heraklionense tenosynovitis case. Surgical debridement of the infected area is widely utilized as well to successfully control infection. However, it is not clear if medical therapy alone could be enough to control the infection. More data are needed to clearly establish a role for surgery in the treatment of this infection.

  Conflicts of interest Top

The authors declare no conflict of interest.

  References Top

E. Tortoli, Z. Gitti, H.P. Klenk, et al, Survey of 150 strains belonging to the Mycobacterium terrae complex and description of Mycobacterium engbaekii sp. nov., Mycobacterium heraklionensesp. nov. and Mycobacterium longobardum sp. nov, Int. J. Syst. Evol. Microbiol. 63 (2013) 401–411.  Back to cited text no. 1
I.K. Neonakis, D.A. Spandidos, Z. Gitti, Mycobacterium heraklionense sp. nov.: a case series, Exp. Ther. Med. 10 (2015) 1401–1403.  Back to cited text no. 2
M. Abedalthagafi, O. Rosenberg, S. Miller, First report oftenosynovitis in an immunocompetent person caused by Mycobacterium heraklionense, JMM Case Rep. 1 (2014). e002071–e002071.  Back to cited text no. 3
R. Vasireddy, S. Vasireddy, B.A. Brown-Elliott, et al, Mycobacterium arupense, Mycobacterium heraklionense, and anewly proposed species, “Mycobacterium virginiense” sp. nov,but not Mycobacterium nonchromogenicum, as species of the Mycobacterium terrae complex causing tenosynovitis/osteomyelitis, J. Clin. Microbiol. 54 (2016) 1340–1351.  Back to cited text no. 4
N. Balagué, I. Uçkay, P. Vostrel, et al, Nontuberculousmycobacterial infections of the hand, Chir. Main 34 (2015) 18–23.  Back to cited text no. 5


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