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Mycobacterium heckeshornense-induced deep abscessin the gluteus maximus muscle:a case report and review of the literature
Hirotoshi Kikuchi1,*, Kurumi Asako1, Hajime Kono 1,
Miwa Asahara2, Takashi Tanaka2, Go Kamosida3,
Takane Ueda3, Shigeru Nagakawa3, Tsuneyuki Ubagai3,
Yuko Kazumi4 and Yasuo Ono1, 3
1

Department of Internal Medicine, Teikyo University School of Medicine,Tokyo, Japan2
Department of Central Clinical Laboratory, Teikyo University Hospital,Tokyo, Japan3
Department of Microbiology and Immunology,
Teikyo University School of Medicine, Tokyo, Japan
4
The Research Institute of Tuberculosis, Japan Anti-Tuberculosis Association,Tokyo, Japan(Received for publication October 29, 2018)

Mycobacterium heckeshornense is rarely isolated from a clinical sample. We
report a case of a patient with polymyositis in whom a deep abscess formed in thegluteus maximus muscle and in which M. heckeshornense was identiﬁed by DNA
sequencing. Combination therapy of levoﬂoxacin and clarithromycin for 2 years
improved inﬂammatory ﬁndings and 5 years have passed without recurrence of thegluteal abscess. Identification of the bacterial strain in nontuberculous
mycobacterium (NTM) infection is important to determine the treatment plan andaccumulation of data on drug sensitivity is required to establish a therapeuticstrategy for rare pathogen such as M. heckeshornense.

Introduction
The incidence of nontuberculous mycobacterium (NTM) infection has shown a tendency toincrease in Japan. The rates for individual bacterial strains are 60% for Mycobacterium avium,
25% for M. intracellulare, and 8% for M. kansasii, while other strains including M. abscessus are*Corresponding auther: Tel: ＋81–3–3964–1211, Fax: ＋81–3–3964–4707, E-mail: hiro-k@med.teikyo-u.ac.jp90（ 90 ）THE JAPANESE JOURNAL OF ANTIBIOTICS

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rare1). In contrast, the incidence of NTM infection in Europe is highest for M. avium complex(MAC), followed by M. xenopi 2,3).
Herein, we report a case of a patient with polymyositis in whom a deep abscess formed in
the gluteus maximus muscle and in which M. heckeshornense is isolated.

Case Report
A 62-year-old man developed myalgia at the proximal muscle 10 years ago. An elevatedblood creatine kinase (CK) level and positive anti-Jo-1 antibody were found and he was diagnosed with polymyositis based on the results of muscle biopsy. Since the disease redeveloped
when the dose of adrenal cortical steroids was decreased, maintenance treatment was provided
with prednisolone (12 mg/day), cyclosporine (150 mg/day) and methotrexate (10 mg/week). The
patient became aware of an uncomfortable feeling in his left buttock 4 months ago and swellingin the same region 2 months ago.
At his ﬁrst visit to our hospital, he had a temperature of 36.0°C with no myalgia, arthralgia,
or obvious trauma. Swelling was conﬁrmed from the left buttock to the external side of the left
femoral region. A blood test showed a white blood cell count of 15,800/μL (stab cells 2.0%, segment cells 94.0%, and lymphocytes 4.0%), a C-reactive protein level of 1.90 mg/dL, and an erythrocyte sedimentation rate of 40 mm/h. The levels of CK (69 IU/L), IgG (1,270 mg/dL), IgA(163 mg/dL), IgM (142 mg/dL), antinuclear antibody (titer ＜40), and anti-Jo-1 antibody (142.5
IU/L, positive) were similar to those in a previous examination, suggesting stable polymyositis.
Magnetic resonance imaging of the area from the buttock to the femoral region suggested
ﬂuid accumulation in a deep area to the side of the left femoral muscle (Fig. 1). Puncture was performed under ultrasound guidance. A standard plate count of bacteria was negative in purulent aspirate however the concentrating method for tubercle bacillus was 1＋. Chest X-ray and chest
computed tomography performed in the same period gave no clear ﬁndings other than pulmonaryﬁbrosis associated with polymyositis, and acid fast bacterium was negative in gastric ﬂuid andblood culture. Puncture was performed twice for the abscess and growth of NTM was found inboth aspirates on the 15th day. In a DNA-DNA hybridization (DDH) test (DDH Mycobacteria,
Kyokuto Pharmaceutical Industrial Co., Ltd, Japan), the bacterium was identiﬁed as M. xenopi.
M. xenopi is rare in Japan and the number of reports on infection of tissues other than the lungs
is small 4). In addition, a drug susceptibility test for the detected bacterium showed sensitivity toethambutol (Table 1), which is inconsistent with M. xenopi. Furthermore, DNA sequencing of 16SrRNA and rpoB gene of the bacterium suggested concordance rates with M. heckeshornense of
100% and 99%, respectively, while those with M. xenopi were 95%. Thus, the NTM was identiﬁed
as M. heckeshornense. After drainage of the abscess, the patient was treated with concomitant
500 mg/day levoﬂoxacin and 400 mg/day clarithromycin for 2 years, and there has been no redevel-Mar. 2019

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72―191（ 91 ）Figure 1.

A. Magnetic resonance imaging (sagittal view) of the area from the buttock to the femoral region. Arrow: deep abscess in thegluteus maximus muscle. B. MRI ﬁndings after 10 months from treatment are shown. Abscess has disappeared.

Table 1. Results drug susceptibility testing in our case

Drug susceptibility tests were determined using a Vite Spectrum-SR
kit. (Kyokuto Pharmaceutical Industrial Co., Ltd, Japan). There are nocriteria of CLSI (Clinical and Laboratory Standards Institute) in M.
heckeshornense.

opment of the condition for 5 years without administration of these antibacterial agents.

Discussion
There are several methods for identiﬁcation of NTM bacterial strains. In 2011, Morimoto etal. suggested that M. heckeshornense, which cannot be identiﬁed using the DDH kit, may be incorrectly identiﬁed as M. xenopi5). In addition, two case reports showed that a NTM identiﬁed as
M. xenopi by the DDH kit was found to be M. heckeshornense, a slow acid fast bacterium 6).

Table 2. Summary of documented Mycobacterium heckeshornense case reports
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72―193（ 93 ）These reports indicate a problem with the method used to identify bacterial strains.
At ﬁrst, we used a DDH kit, with which 18 bacterial strains can be identiﬁed at one time byhybridization using DNA of a type strain and DNA of a suspected strain. However, the NTM wassensitive to ethambutol (Table 1) and the infection site was not in the lungs, both of which differ
from the typical characteristics of M. xenopi. Therefore, we performed DNA sequencing for 16S
rRNA and rpoB gene of the bacterium and identiﬁed the strain as M. heckeshornense.
M. heckeshornense was ﬁrst reported by Roth et al.7) and has been conﬁrmed in only 17cases (Table 2)5〜7, 9〜19), including our case, because DNA sequencing is required for identiﬁcation. In recent years, Yokoyama et al.19) diagnosed M. heckeshornense using matrix-assisted laser
desorption ionization-time of ﬂight mass spectrometry (MALDI-TOF MS). They had also conﬁrmed in the DNA sequence and had reported that MALDI-TOF MS was useful in the diagnosisof M. heckeshornense.
A half of these cases (47.1%) had infection at sites other than the lungs. The outcomes werefavorable in most cases (improvement: 81.2%, redevelopment: 18.8%) and improvement occurred without administration of antibacterial agents in some cases. Pulmonary infection with M.
xenopi is often unresponsive to drug treatment alone, and thus surgery is required in manycases8), however the pathogenicity of M. heckeshornense is unclear. van Hest et al. suggested thatM. heckeshornense detected from lung tissues with normal immunity was pathogenic9), but atherapeutic strategy has not been established because of the small number of reported cases.
In our case, multiple immunosuppressants were used to control the disease activity of polymyositis. Initially we were considering treatment with three drugs such as rifampicin, levoﬂoxacin and clarithromycin, but because the patient refused to increase the amount of steroids associated with rifampicin administration, we treated with two agents with reference to the case reportof M. xenopi. Since the possibility that the combination of levoﬂoxacin and clarithromycin induces drug resistance of M. heckeshornense cannot be denied either, studies on the resistancemechanism of M. heckeshornense by clarithromycin are desired.
We conclude that identiﬁcation of the bacterial strain in NTM infection is important to determine the treatment plan. Accumulation of data on drug sensitivity is required to establish a therapeutic strategy for rare pathogen such as M. heckeshornense.
Conﬂict of interests
We declare that we have no conﬂict of interest.
Acknowledgments
We thank the microbiology technologist (The Research Institute of Tuberculosis, Japan AntiTuberculosis Association) for performing 16S rRNA, rpoB, and hsp65 gene sequence analysis forthe identiﬁcation of M. heckeshornense. This study was supported by the Private University94（ 94 ）THE JAPANESE JOURNAL OF ANTIBIOTICS

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Research Branding Project.

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