|Year : 2016 | Volume
| Issue : 3 | Page : 351-353
Nontuberculous mycobacterial adenitis outside of the head and neck region in children: A case report and systematic review of the literature
Diego Heraud1, Richard D Carr2, Jason McKee3, Walter Dehority4
1 Department of Pediatrics, University of New Mexico Health Sciences Center, Albuquerque, NM, USA
2 University of New Mexico Health Sciences Center Health Informatics Center, Albuquerque, NM, USA
3 Department of Surgery, University of New Mexico Health Sciences Center, Albuquerque, NM, USA
4 Department of Pediatrics, Division of Infectious Diseases, University of New Mexico Health Sciences Center, Albuquerque, NM, USA
|Date of Web Publication||13-Feb-2017|
University of New Mexico, Department of Pediatrics, Division of Infectious Diseases, MSC10 5590, 1 University of New Mexico, Albuquerque, NM 87131-0001
Source of Support: None, Conflict of Interest: None
Nontuberculous mycobacterial (NTM) adenitis of the head and neck region is well-described in healthy children, most commonly presenting under the age of 5 years. Extracervicofacial NTM adenitis is less common. We present a case of NTM inguinal adenitis in a 2-year-old girl, followed by a systematic review of the literature.
Keywords: Adenitis, Inguinal, Pediatric, Mycobacterium avium intracellulare, Nontuberculous mycobacteria
|How to cite this article:|
Heraud D, Carr RD, McKee J, Dehority W. Nontuberculous mycobacterial adenitis outside of the head and neck region in children: A case report and systematic review of the literature. Int J Mycobacteriol 2016;5:351-3
|How to cite this URL:|
Heraud D, Carr RD, McKee J, Dehority W. Nontuberculous mycobacterial adenitis outside of the head and neck region in children: A case report and systematic review of the literature. Int J Mycobacteriol [serial online] 2016 [cited 2021 Apr 20];5:351-3. Available from: https://www.ijmyco.org/text.asp?2016/5/3/351/200079
| Case report|| |
A previously healthy girl, aged 2 years and 9 months, who lived on a ranch in northern New Mexico was seen by her primary care provider with a 3-week history of a right-sided inguinal swelling. She had cut her right foot several weeks prior and frequently played in the animal pastures barefoot. No erythema, fever, or pain was present. Initial examination was concerning for an inguinal adenitis. Five-day courses of amoxicillin–clavulanate, followed by trimethoprim-sulfamethoxazole failed to improve her condition. Ultrasonography at this time revealed matted lymphadenopathy. Six and 10 days after her ultrasound, she underwent fine-needle aspiration (FNA) of the involved lymph nodes, which produced negative Gram stains and sterile bacterial cultures. Following the last FNA, a thick, cream-colored discharge was noted from the aspiration site, and she developed violaceous discoloration over the region. The site remained nontender, and she was otherwise well. Two weeks after the last aspiration, she underwent a third FNA, which was sent for acid-fast bacillus (AFB) stain and culture. AFB stains were positive, and her culture ultimately grew Mycobacterium avium intracellulare (MAC). The patient was observed carefully off of antimycobacterial therapy, with slow improvement of the lesions. One month later, the patient was admitted with acute onset of fever and new inguinal discharge, pain, and erythema. Wound cultures of the discharge demonstrated monomicrobial growth of methicillin-susceptible Staphylococcus aureus (MSSA), concerning for a bacterial superinfection. The patient underwent operative debridement at this time, which demonstrated necrotic tissue and extensive purulence, cultures of which again demonstrated MSSA. Given the extensive necrotic tissue present, a formal lymphadenectomy could not be performed, though the area was widely debrided. The patient improved after surgery, and was discharged on cephalexin for treatment of a presumed MSSA superinfection, along with clarithromycin and rifampin for treatment of MAC. She completed 2 weeks of antistaphylococcal therapy and 4 months of antimycobacterial therapy, with resolution of her adenitis.
| Discussion|| |
Given the lack of published literature on this topic, we performed a systematic literature search on PubMed and Web of Knowledge on 19 November 2014. A search of PubMed utilizing medical subject heading terms “Mycobacterium Infections, Non-Tuberculous” and “Lymphadenitis” was undertaken, and the results were combined with a keyword search of titles and abstracts using the phrase “non-tuberculous mycobacterial” and the keyword “lymphadenitis” and limited to the child age group (birth to 18 years). Given the long time between the original literature search and manuscript completion, the search was repeated on 31-Jul-2015 with no new reports identified. Web of Knowledge was searched with the same keywords and was limited to the pediatric research area. The references from two Pediatric Infectious Disease textbooks were also reviewed. Reports were limited to the English language, with no restriction on publication date. Inclusion criteria included an age ≤18 years, no documentation of an immunosuppressive condition, and microbiologic [culture or polymerase chain reaction (PCR)] confirmation of a NTM isolated from lymphatic tissue. For manuscripts meeting inclusion criteria, references were sequentially searched for additional cases. Cases of invasive mycobacterial disease (e.g., mycobacteremia) were excluded, given the desire to focus on immunocompetent children and the rarity with which these organisms disseminate in such patients. Cases of lymphadenitis outside the head and neck region were then separated from those involving the head and neck region and analyzed.
The original search produced 246 individual articles. Review of abstracts eliminated 31 articles, while 13 were eliminated due to publication in a foreign language. Review of these remaining articles eliminated another 104, while sequential review of references produced 43 additional articles, for a total of 141. The most common reason for study ineligibility was failure to document a microbiologically confirmed NTM infection.
Altogether, 2194 individual cases of NTM adenitis meeting our inclusion criteria were reported from 1956 to 2014. Thirty-five cases were documented outside the head and neck region (1.6% of all cases) and were published between 1966 and 2008 ,,,,,,,,,. Many cases lacked specific clinical details, and though verification of inclusion criteria was possible, some data (e.g., gender, specific extracervical location) were unavailable.
The 35 cases in the literature were combined with our case to help characterize the clinical course of this process. The inguinal region was most frequently involved in the 15 cases with available data on specific location (13 cases, 86.7%). Only one case was bilateral. Ages ranged from 18 months to 5 years, with a mean age of 3.2 years (median of 3.0 years). Girls predominated (six of the eight with gender information available). Contiguous trauma prior to disease onset was identified in two subjects. Five subjects were treated with antimycobacterial therapy for an average duration of 87 days. Twenty-four cases were a result of infection with MAC, with three cases due to NTM with no identifiable species, two cases each of Mycobacterium fortuitum and Runyon class III NTM, and one case each of Mycobacterium malmoense, Mycobacterium scrofulaceum, Mycobacterium terrae, Mycobacterium marinum, and Mycobacterium haemophilum. Three of six patients with data on overlying skin discoloration presented with erythema. Seven subjects underwent surgical debridement, with four undergoing complete excision and three undergoing partial debridement. Sinus formation complicated two of the three patients with partial debridement, and one of the four undergoing complete excision. Two patients required two surgeries.
NTM adenitis outside the head and neck region appears to be relatively uncommon, with only 35 cases identified in our review. This represents only 1.6% of all cases of microbiologically confirmed NTM adenitis in otherwise healthy children in the world literature, which is lower than previously reported . This may be due to the strict inclusion criteria we utilized, given the requirement for a microbiological or PCR-confirmed NTM infection. Nonetheless, the presentation of NTM adenitis outside the head and neck region appears to mirror that of head and neck disease. The mean age of children in our review was 3.2 years, with a female predominance as seen with cervicofacial NTM adenitis . Mortality was not reported, and bilateral disease was rare. Skin involvement was noted in half of cases. MAC was the predominant organism involved (present in two-thirds of cases), as is typically described with NTM adenitis of the head and neck region . However, eight different species of NTM were reported in keeping with the broad distribution of species involvement seen in head and neck disease .
Our case had antecedent trauma to her ipsilateral foot prior to the onset of infection and lived on a farm with ample exposure to soil, through which she may have been infected. Contiguous trauma was reported in one other case in our review. Such trauma may provide a portal of entry for infection and facilitate dissemination to regional lymph nodes. Limited data was available in our review on treatment options, though excisional surgery was reported to be successful, as it has been in NTM adenitis of the head and neck region . Notably, lymphedema was reported in one case following surgical excision . This was a concern in our patient, as well, given the need to resect such a large volume of lymphatic tissue in the involved leg. As is seen with NTM adenitis of the head and neck region, sinus formation and poor wound healing may be seen with NTM adenitis outside of the head and neck region . Though limited by the small number of reports, sinus formation was more likely following nonexcisional surgery in our review. It should be noted that our patient underwent three FNA procedures prior to her ultimate diagnosis and eventually developed a draining sinus. This has also been well described in NTM adenitis of the head and neck region .
Our review had several limitations. It was subject to publication biases, as well as informational biases inherent to the analysis of retrospective data. The quality of data reported was variable between reports, and many cases were presented as aggregate data, thereby limiting our ability to abstract clinical details. Our requirement for microbiologic or PCR confirmation of infection may have excluded several cases from our review; however, we felt that given the anecdotal nature of this literature and the variety of pathologic processes that may cause granulomatous inflammation in lymphatic tissue, use of less stringent inclusion criteria (e.g., histological findings or clinical presentation alone) may have resulted in a lack of specificity. Additionally, this facilitated an accounting of the species distribution of NTM involved in extracervicofacial infections.
Extracervicofacial adenitis due to NTM is a rare finding in children, comprising 1.6% of all reported cases of NTM adenitis in children in the world literature. However, it appears to present in a similar fashion to NTM adenitis of the head and neck region. Data on therapy is limited, but surgical excision with or without postoperative antimycobacterial therapy appears to be an acceptable option.
| Conflicts of interest|| |
All contributing authors declare no conflicts of interest.
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