J Infect Chemother 21 (2015) 691e694

Contents lists available at ScienceDirect

Journal of Infection and Chemotherapy journal homepage: http://www.elsevier.com/locate/jic

Case report

Rhinosinusitis and disseminated cutaneous infection caused by Mycobacterium chelonae in an immunocompromised patient Yasunori Enomoto a, e, *, Misao Oba b, Norihisa Ishii c, Kazue Nakanaga c, Yuki Yagi d, Hirotsugu Hasegawa a, Yuichi Ozawa a, Takashi Matsui a, Koshi Yokomura a, Takafumi Suda e a

Department of Respiratory Medicine, Respiratory Disease Center, Seirei Mikatahara General Hospital, Japan Department of Dermatology, Seirei Mikatahara General Hospital, Japan Leprosy Research Center, National Institute of Infectious Diseases, Japan d Department of Otorhinolaryngology, Seirei Mikatahara General Hospital, Japan e Second Department of Internal Medicine, Hamamatsu University School of Medicine, Japan b c

a r t i c l e i n f o

a b s t r a c t

Article history: Received 13 February 2015 Received in revised form 20 March 2015 Accepted 24 April 2015 Available online 12 May 2015

Mycobacterium chelonae frequently involves the skin, and the disseminated form can be observed in immunocompromised patients. In contrast, rhinosinusitis caused by the bacterium is a rare manifestation, which occurs independently of immune status. We report here a rare case of M. chelonae infection presenting as both disseminated cutaneous infection and rhinosinusitis in an immunocompromised patient. He had received systemic corticosteroids for 11 months due to cryptogenic organizing pneumonia. Before admission, he sustained injuries to his left arm and hand; those injuries succumbed to an infection that would subsequently spread to his other limbs, face, and even nasal cavities. This valuable case suggests that disseminated cutaneous infection by M. chelonae could spread to other organs. © 2015, Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases. Published by Elsevier Ltd. All rights reserved.

Keywords: Mycobacterium chelonae Disseminated cutaneous infection Rhinosinusitis

1. Introduction Cutaneous infections by nontuberculous mycobacteria (NTM) have been increasingly reported, particularly by Mycobacterium marinum and rapidly growing mycobacteria; e.g., Mycobacterium chelonae, Mycobacterium fortuitum, and Mycobacterium abscessus [1]. Infections caused by M. chelonae commonly involve the skin, eye, bone, and soft tissue. With skin involvement, a disseminated form is frequently observed in immunocompromised patients [2,3]. Several authors have reported other atypical presentations of M. chelonae infection, including lung infection [4], thyroid abscess [5], meningitis [6], and endocarditis [7]. Rhinosinusitis caused by M. chelonae is also uncommon, but has been observed in both

* Corresponding author. Department of Respiratory Medicine, Respiratory Disease Center, Seirei Mikatahara General Hospital, 3453 Mikatahara-cho, Kita-ku, Hamamatsu City 433-8558, Japan. Tel.: þ81 53 436 1251; fax: þ81 53 438 2971., E-mail addresses: [email protected], [email protected] (Y. Enomoto).

immunocompetent and immunocompromised patients [8e12]. We report here a rare case of M. chelonae infection presenting as both disseminated cutaneous infection and rhinosinusitis in an immunocompromised patient. 2. Case report An 86-year-old Japanese male was admitted to our hospital with complaints of general fatigue and appetite loss persisting for 2 months. He had been diagnosed with cryptogenic organizing pneumonia 11 months ago, and had received long-term treatment with systemic corticosteroids. The lung disease was controlled by oral predonisolone (PSL) at adjusted doses of 5e30 mg/day. He had also suffered from chronic heart failure with mitral and tricuspid moderate regurgitation, chronic atrial fibrillation, and chronic kidney disease. Warfarin, furosemide, and spironolactone had been administered for the diseases. There were no other past medical histories including cutaneous and nasal/paranasal diseases. He had some difficulties in walking and eating by himself and lived in a nursing care home. One month ago, he fell over and sustained injuries to his left arm and hand. These injuries had been treated with

http://dx.doi.org/10.1016/j.jiac.2015.04.014 1341-321X/© 2015, Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases. Published by Elsevier Ltd. All rights reserved.

692

Y. Enomoto et al. / J Infect Chemother 21 (2015) 691e694

white petroleum jelly and had partially crusted, but not fully healed; they had become itchy and painful, and had gradually spread to his other limbs and face. Those abnormalities had not been found by the nurses in the nursing care home before the episode of injury. His general condition as well as the skin disease gradually deteriorated, and consequently, he became bedridden. On admission, he was afebrile; blood pressure was 104/60 mm Hg, pulse was 90/min, and peripheral artery oxygen saturation was 98% on room air. His height and body weight were 157 cm and 45 kg respectively. Although his vital signs were stable, he appeared to be in poor health generally. The dosage of PSL was at 20 mg/day. Physical examination showed multiple ulcerated nodules with crusts on his face, subcutaneous nodules on his left elbow, and skin ulceration on his right leg and bilateral hands. Skin ulceration was severe, particularly on the dorsal parts of his fingers bilaterally (Fig. 1). Both nasal cavities were partially covered by crusts. After removing these, endoscopic intranasal evaluation revealed ulcerative mucosa, particularly in the nasal septum and middle nasal concha. White blood cell count was 6290/mL, hemoglobin was 13.9 g/dL, and platelet count was 41  104/mL. Although liver function tests were within normal ranges, serum creatinine and urea nitrogen were elevated (2.5 mg/dL and 77 mg/dL, respectively). C-reactive protein was also elevated (11.4 mg/dL). Anti-HIV antibody was negative. For infectious disease workup, blood, a respiratory tract specimen (collected by nasal suction), and pus from the skin eruptions on the limbs and face were extracted for cultivation. The smears of all samples except blood showed positive acid-fast bacillus on Ziehl-Neelsen staining. The DNA of Mycobacterium tuberculosis and Mycobacterium avium complex was not detected by PCR analysis in any of the samples. Chest CT revealed

no abnormal findings, including consolidation and micronodules, suggesting the relapse of cryptogenic organizing pneumonia or pulmonary nontuberculous mycobacterial infection. Instead, sinus CT suggested mild sinusitis (Fig. 2). Bony involvement was also observed. We performed multiple biopsies of the skin eruptions on the limbs and face, middle nasal meatus, and middle nasal concha. All of the biopsy slides typically showed moderate-to-severe inflammatory cell infiltration without evidence of granuloma formation, presenting with positive acid-fast bacillus (Fig. 3). We diagnosed a combined case of disseminated cutaneous infection and rhinosinusitis caused by NTM, and subsequently initiated empiric chemotherapy with a combination of clarithromycin (CAM) 400 mg/day, levofloxacin (LVFX) 250 mg/every other day, and rifampicin (RFP) 450 mg/day. The dosage of LVFX was adjusted on the basis of the patient's renal function. Although the skin eruptions showed some improvement after a week, the patient's general condition gradually worsened. He died suddenly, 12 days after initiating anti-mycobacterial treatment. We could not obtain consent to perform an autopsy. DNAeDNA hybridization identified the cultured mycobacteria as M. chelonae, which was consistent with the results of sequence analysis concerning 16S rRNA and rpoB [13]. The minimum inhibitory concentrations for CAM, LVFX, and RFP were 1.0 mg/mL, 16.0 mg/mL, and 32.0 mg/mL, respectively. 3. Discussion M. chelonae infection frequently involves the skin, eye, bone, and soft tissue, deriving from inoculation with the bacterium. Cutaneous infection by M. chelonae has been widely reported in the

Fig. 1. Disseminated skin eruptions. Ulcerated nodules with crusts on the face (a). Skin ulceration on the right leg (arrows) (b), fingers of the left hand (c), and fingers of the right hand (d). Subcutaneous nodules on the left elbow (arrows) (e). These lesions underwent skin biopsies and culture examination.

Y. Enomoto et al. / J Infect Chemother 21 (2015) 691e694

693

Fig. 2. Sinus CT. Medial wall thinning and bone destruction on right maxillary sinus at axial view (a) and coronal view (b). Mild retention of pus is also observed.

literature, both in immunocompromised and immunocompetent patients [1,3,14]. Wallace et al. identified three types of cutaneous infection in their large cohort: disseminated form, localized form, and catheter-related form [3]. The most common type was the disseminated form, in which as many as 92% of the patients had received corticosteroids. Second most common type was the localized form, with most patients having experienced previous traumatic injuries, surgeries, or other cutaneous interventions. Our patient had a significant history of injuries to his left arm and hand prior to admission. Although M. chelonae would initially colonize only on the localized lesions, it would disseminate to the other limbs and face on the background of immunosuppression due to long-term corticosteroid use. In contrast, rhinosinusitis is a rare manifestation of M. chelonae infection. Similarly to cutaneous infection, it can occur regardless of immune status [8e12]. However, to the best of our knowledge, a comorbid case of cutaneous infection has not been reported. In a

previous study of 8 patients with mycobacterium sinusitis, 4 cases were identified as M. chelonae infection [8]. All the patients had undergone prior endoscopic sinus surgeries. In addition, an outbreak of post-surgical nasal cellulitis caused by M. chelonae was reported in a previous case-control study [12]. M. chelonae rhinosinusitis as a primary lesion seems to be strongly associated with prior sinus surgeries. However, our patient had not undergone any such surgery. CT indicated that his sinusitis was mild, although the nasal findings were relatively severe. Thus, the bony destructive change was considered to originate from rhinitis, not sinusitis. His facial skin eruptions, including those on the nose, were itchy; they would have been touched and/or scratched frequently. Therefore, we speculate that his nasal infection could have been established via contact with the facial disease, leading to rhinosinusitis. Facial skin eruptions due to M. chelonae are reported to be unusual [15], which could explain why this combination is rare. On the other hand, rhinitis/sinusitis and disseminated cutaneous disease can also be

Fig. 3. Pathological findings. Severe inflammatory cell infiltration with neutrophils and lymphocytes in the dermis (a) and nasal mucosa (b) (Hematoxylin-Eosin;  50 and  400, respectively). Red rod-shaped microorganisms surrounded by inflammatory cells in the nasal mucosa (c) (Ziel-Neelsen;  630). There are approximately 20 bacilli in this field of view (arrow).

694

Y. Enomoto et al. / J Infect Chemother 21 (2015) 691e694

observed in fungal infections such as sporotrichosis and coccidioidomycosis [16e18]. In the differential diagnosis of this combination, opportunistic fungal infections should be incorporated. M. chelonae is widely acknowledged to be susceptible to CAM, tobramycin, linezolid, and imipenem, and their use in combination including CAM is a recommended treatment [2]. In our patient, we empirically started anti-mycobacterial combination chemotherapy with CAM, LVFX, and RFP, which was selected to target major pathogens such as M. marinum, M. chelonae, M. abscessus, M. fortuitum, M. avium complex, and Mycobacterium haemophilum as the possible cause of cutaneous and nasal/paranasal mycobacterial infection [1,2,8,11]. Susceptibility to our selected drugs appeared favorable; indeed, the rash showed some improvement. Nevertheless, we could not save the patient. That would be a reflection of his severe immunosuppression and/or poor general condition associated with old age and preexisting diseases, such as heart failure and renal dysfunction. In conclusion, this is the first case report of M. chelonae infection presenting as both disseminated cutaneous infection and rhinosinusitis in an immunocompromised patient. This valuable case suggests that disseminated cutaneous infection could spread to the other organs, possibly via contact infection. Conflict of interest None. References [1] Wentworth AB, Drage LA, Wengenack NL, Wilson JW, Lohse CM. Increased incidence of cutaneous nontuberculous mycobacterial infection, 1980 to 2009: a population-based study. Mayo Clin Proc 2013;88:38e45. [2] Griffith DE, Aksamit T, Brown-Elliott BA, Catanzaro A, Daley C, Gordin F, et al. An official ATS/IDSA statement: diagnosis, treatment, and prevention of nontuberculous mycobacterial diseases. Am J Respir Crit Care Med 2007;175: 367e416.

[3] Wallace Jr RJ, Brown BA, Onyi GO. Skin, soft tissue, and bone infections due to Mycobacterium chelonae chelonae: importance of prior corticosteroid therapy, frequency of disseminated infections, and resistance to oral antimicrobials other than clarithromycin. J Infect Dis 1992;166:405e12. [4] Singh N, Yu VL. Successful treatment of pulmonary infection due to Mycobacterium chelonae: case report and review. Clin Infect Dis 1992;14:156e61. [5] Pandita D, Carson PJ. Thyroid abscess caused by Mycobacterium chelonae. Clin Infect Dis 1999;28:1183e4. [6] Salmanzadeh S, Honarvar N, Goodarzi H, Khosravi AD, Nashibi R, Serajian AA, et al. Chronic mycobacterial meningitis due to Mycobacterium chelonae: a case report. Int J Infect Dis 2014;27:67e9. [7] Takekoshi D, Al-Heeti O, Belvitch P, Schraufnagel DE. Native-valve endocarditis caused by Mycobacterium chelonae, misidentified as polymicrobial gram-positive bacillus infection. J Infect Chemother 2013;19: 754e6. [8] Suh JD, Ramakrishnan VR, Tajudeen B, Reger C, Kennedy DW, Chiu AG. Identification and treatment of nontuberculous Mycobacterium sinusitis. Am J Rhinol Allergy 2011;25:421e4. [9] Mullin D, Jothi S, Healy D. Mycobacterium chelonae infections involving the head and neck. Ann Otol Rhinol Laryngol 2009;118:714e20. [10] Spring PM, Miller RH. Initial report of primary sinusitis caused by an atypical pathogen (Mycobacterium chelonae) in an immunocompetent adult. Ear Nose Throat J 1999;78:358e9. 362e4. [11] Solyar A, Lee AS, Przybyszewski B, Lanza DC. Atypical mycobacterium detection in refractory chronic rhinosinusitis. Otolaryngol Head Neck Surg 2012;146:1012e6. [12] Soto LE, Bobadilla M, Villalobos Y, Sifuentes J, Avelar J, Arrieta M, et al. Postsurgical nasal cellulitis outbreak due to Mycobacterium chelonae. J Hosp Infect 1991;19:99e106. [13] Nakanaga K, Hoshino Y, Yotsu RR, Makino M, Ishii N. Laboratory procedures for the detection and identification of cutaneous non-tuberculous mycobacterial infections. J Dermatol 2013;40:151e9. [14] Kennedy BS, Bedard B, Younge M, Tuttle D, Ammerman E, Ricci J, et al. Outbreak of Mycobacterium chelonae infection associated with tattoo ink. N Engl J Med 2012;367:1020e4. [15] Mali SA, Doctor TB, Doshi AP, Sharma R. Extensive infection of face by Mycobacterium chelonae: an unusual presentation. Indian J Dermatol 2014;59: 495e7. [16] Thompson 3rd GR, Patterson TF. Fungal disease of the nose and paranasal sinuses. J Allergy Clin Immunol 2012;129:321e6. [17] Yap FB. Disseminated cutaneous sporotrichosis in an immunocompetent individual. Int J Infect Dis 2011;15:e727e9. [18] Carpenter JB, Feldman JS, Leyva WH, DiCaudo DJ. Clinical and pathologic characteristics of disseminated cutaneous coccidioidomycosis. J Am Acad Dermatol 2010;62:831e7.

Rhinosinusitis and disseminated cutaneous infection caused by Mycobacterium chelonae in an immunocompromised patient.

Mycobacterium chelonae frequently involves the skin, and the disseminated form can be observed in immunocompromised patients. In contrast, rhinosinusi...
1MB Sizes 1 Downloads 8 Views