Mycobacterium Abscessus Cervical Lymphadenitis: an Immunocompetent Child

MYCOBACTERIUM ABSCESSUS CERVICAL LYMPHADENITIS: AN IMMUNOCOMPETENT CHILD

Jenn-Tzong Chang, Yung-Feng Huang, Yen-Ting Lin, Yung-Ching Liu,1 Lin-Hui Chiu,2 Hui-Zin Tu,2 and Kai-Sheng Hsieh Department of Pediatrics, 1Division of Infectious Disease, Department of Internal Medicine, and 2Division of Microbiology, Kaohsiung Veterans General Hospital, Kaohsiung, Taiwan.

Nontuberculous mycobacterium (NTM) is one of the well-known causes of cervicofacial lymphadenopathy in children under 5 years of age. Children often present with a painless cervical mass that fails to respond to conventional antibiotics. They are often referred under the suspicion of a neoplasm or bacterial adenitis rather than NTM cervical lymphadenitis. The lack of systemic symptoms, modest or negative purified protein derivative test and absence of exposure to active tuberculosis are characteristics of NTM lymphadenitis. The diagnosis usually requires the isolation of pathogen or pathologic proof. Complete excision is the choice of treatment by the majority of authors in the literature. This not only enables rapid diagnosis but ensures the lowest recurrence rate. Medical management is sometimes successful when complete resection is impossible or refused. To our knowledge, the incidence of NTM cervical lymphadenitis in children is increasing throughout the world. However, such reports of children in Taiwan is lacking. Clinicians should suspect a possible nontuberculous mycobacterial infection when a cervical lump is found in a child.

Key Words: immunocompetent, lymphadenitis, Mycobacterium abscessus, nontuberculous mycobacteria, Taiwan (Kaohsiung J Med Sci 2006;22:415–9)

Nontuberculous mycobacteria (NTM) are ubiquitous However, most NTM infections occur in seemingly organisms that are found in soil, water, and food healthy children [5]. [1–3]. NTM was first described as a pathogen of the Children often present with a painless cervical head and neck area in 1956 [1], and it was well mass, followed by a dusky/purplish discoloration, known to cause cervicofacial lymphadenopathy in and shining, thinning, and flaking of the overlying children [1,2,4]. Infections often occur in children skin [4]. Many of the lymph nodes suppurate and between 1 and 5 years of age, but they rarely cause this results in chronic draining sinuses without inter- systemic symptoms [4]. vention [4]. All nodes in the cervical region may Infections caused by NTM can be associated with be affected, especially in the submandibular and immunocompromised status or they may follow cer- submental chains [2,4,6]. Children are often referred tain procedures such as dialysis, acupuncture, local under the suspicion of a neoplasm, bacterial adenitis, injection, or placement of intravenous catheters. or reactive adenopathy, rather than NTM cervical lymphadenitis [7]. To our knowledge, the incidence of NTM cervical lymphadenitis in children is increasing worldwide Received: December 6, 2005 Accepted: March 30, 2006 [1–3]. However, it has yet to be reported in children Address correspondence and reprint requests to: Dr Yung- in Taiwan [3,8]. Clinicians should suspect a possible Feng Huang, Department of Pediatrics, Kaohsiung Veterans General Hospital, 386 Ta-Chung 1st Road, Kaohsiung, Taiwan. NTM infection when a cervical lump is found in E-mail: [email protected] a child.

Figure 1. An erythematous, tender and nonmovable mass was Figure 2. Only a small scar lesion remained after 6 months of noted over the right submandibular area at admission. treatment.

CASE PRESENTATION purified antigen (PPD) test was negative with a 13-mm induration judged at 72 hours. A 6-year-old boy presented with a neck mass of 1 His condition failed to improve clinically with clin- month’s duration over the right submandibular region. damycin treatment. Fever persisted and the cervical No trauma or insect bite history could be traced. He mass became erythematous with limited whitish dis- had received local treatment of the nose and throat at charge. Another fine needle aspiration was performed an otolaryngology clinic. One day later, the neck mass 2 weeks after admission, and acid-fast stain was posi- was diagnosed. He was brought to a local hospital for tive. A rapid-growing bacterium was cultured, and it treatment, where oxacillin and then augmentin were was finally identified as Mycobacterium abscessus. prescribed. Unfortunately, fever, tenderness, and pro- Surgical resection of the cervical mass was sug- gressive enlargement of the mass were noted 2 weeks gested, but the family refused because they were after its onset. Subsequently, he was referred to our worried about the possible complications. We pre- hospital. scribed azithromycin and pyrazinamide as treatment. He had a past history of congenital heart disease The mass shrunk and ulcerated with discharge dur- with membranous type ventricular septal defect, ing the medical management. After 6 months of treat- which had closed spontaneously during infancy. He ment, only a small lesion remained over the right had been vaccinated with Bacillus Calmette–Guerin submandibular region (Figure 2). (BCG) at the age of 2 months. The parents had no significant medical histories. Physical examination showed a tender, non- DISCUSSION movable mass, about 5 × 3 cm, over the right submandibular area (Figure 1). The leukocyte count was 13.59 × NTM is the most common cause of chronic lym- 109/L, hemoglobin was 12.2 g/dL, and platelets were phadenitis in healthy children under the age of 5 years 314 × 109/L. C-reactive protein was 0.7 mg/dL and [1,2]. Children often present with a painless cervical the erythrocyte sedimentation rate was 70 mm/hour. mass that fails to respond to conventional antibiotics. Immunologic studies including IgG (975 mg/dL), IgA A high index of suspicion is needed to rule out such (177 mg/dL), IgM (195 mg/dL), and lymphocyte sub- an infection. However, there is often a delay of 8 populations were all within normal limits. weeks or more before the diagnosis is made in the Fine needle biopsy was done 2 days after admis- majority of children [9]. sion. It showed a picture of lymph node with suppu- Although NTM is a well-known cause of cervical rative and granulomatous inflammation. Gram stain, lymphadenitis of children in Western countries, there acid-fast stain, and cultures were all negative. Protein have been no reports in Taiwanese children till now.

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Initially, we tried to rule out lymphoma since there was skin and soft tissue infections, especially after trauma poor evidence of infection in our case, but fine needle or a surgical procedure, are the most common infec- biopsy revealed suppurative and granulomatous tions caused by M. abscessus [13]. The hosts are usu- inflammation of lymph node instead of malignant ally immunocompetent. RGM might be important change. Suppurative granulomas are the most charac- pathogens in Taiwan, since they accounted for 56.2% teristic feature in skin biopsy specimens from cuta- of NTM diseases in one series [3]. About 1/5 of these neous NTM infections [10]. Langerhans giant cells, were due to M. abscessus, and all occurred in adults. caseating necrosis, granulomatous inflammation, and We believe that this is the first report of M. abscessus calcification can be seen in mycobacterial cervical cervical lymphadenitis in a child in Taiwan. lymphadenitis [6]. Therefore, physicians should sus- Surgery is the choice of treatment for NTM lym- pect NTM infection if the above histopathologic find- phadenitis by most authors in the literature for several ings are found. reasons: (1) it provides rapid tissue diagnosis; (2) Definite diagnosis of NTM lymphadenitis is made it increases the cure rate with the best cosmetic out- by positive culture from a surgical specimen, but the come and a low complication rate when performed yield rate varies greatly from 20% to 80% [1,2,4–6]. early; and (3) it avoids the costs and toxicity associated However, a negative result should not exclude its with the long-term use of antibiotics. Within the vari- diagnosis, since the organism has fastidious growth ous surgical options, complete excision has the lowest requirements [2,6]. Further, it usually takes several recurrence rate, especially in advanced cases [1–6]. weeks to obtain the culture results. Thus, the institu- Medical treatment is sometimes successful when tion of treatment should be based on the clinical diag- complete excision is impossible or refused [4]. The nosis and not on the cultures. Although the NTM newer macrolides, such as clarithromycin or azithro- infection of our patient was confirmed eventually, it mycin, show effective in vitro activity against most was not found through fine needle biopsy in the begin- NTM [14–17]. In general, the choice of antibiotic ning. We suggest that diagnostic procedures should should be based on susceptibility tests. Antimicrobial be repeated regardless of previous negative results if therapy may vary depending on the species of NTM mycobacterial lymphadenitis is still highly suspected. and the nature of the disease. For example, the treat- The demonstration of acid-fast staining depends ment of localized infections due to M. abscessus is cur- on the number of bacilli in the sample studied, which rently managed by using clarithromycin alone, while is related mainly to the immunology status of the tobramycin and imipenam are suggested to be added host, and it is usually smaller in normal hosts [10]. In for serious disseminated infections [15,17]. However, a immunocompetent hosts, acid-fast bacilli staining was well-designed controlled study is required to support positive in only 11–30% of cases [5,10,11]. The presence their clinical usefulness. of acid-fast bacilli on a smear is helpful in determin- The role of antituberculous drugs for the treatment ing whether or not the NTM isolates represent a sig- of NTM is controversial as well, especially in children. nificant infection or just a contamination [3]. Loeffler had suggested a 2–3 drug regimen including The tuberculin test is used to show delayed-type either clarithromycin or azithromycin, rifampin with hypersensitivity reactions against mycobacterial anti- or without ethambutol [4]. Nolt et al also stated that it gen, in which the reagent is mostly PPD. The diagnosis unclear as to whether or not antituberculous drugs tic criteria of PPD test differ between countries. In hasten recovery from NTM infection [14]. However, Taiwan, a positive reaction means ≥ 18 mm induration most NTM are considered to be resistant to the major- with BCG vaccination history or ≥ 10 mm induration ity of antituberculous drugs according to susceptibil- without BCG vaccination history at 72 hours after ity testing, particularly in the case of RGM [1,3,14–16]. injection of PPD. This is considered to be the principal In conclusion, the prevalence of NTM cervical diagnostic tool in tuberculous infections in non- lymphadenitis in children has increased worldwide, endemic areas, whereas the result is mostly negative but not in Taiwan. Its diagnosis is sometimes difficult or intermediate in nontuberculous infections [5,6]. and requires a high degree of suspicion. We share our Mycobacterium abscessus is one of the species of clinical experience on the distinction of a cervical rapidly growing mycobacteria (RGM) [12]. Localized mass in children through this report.

Kaohsiung J Med Sci August 2006 • Vol 22 • No 8 417 J.T. Chang, Y.F. Huang, Y.T. Lin, et al

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