Mycobacterial Cervicofacial Lymphadenitis In Children Research Articles

Comparison of treatment modalities for non-tuberculous mycobacterial cervicofacial lymphadenitis in children.

We aim to compare the different treatment modalities of non-tuberculous cervicofacial lymphadenitis in children, by means of a retrospective study conducted in the University Hospitals of Leuven of patients treated between 2012 and 2022. For this retrospective cohort study, data were collected and pseudonimised from 52 patients with non-tuberculous cervicofacial lymphadenitis, who were treated in our hospital between January 2012 and December 2022, either conservatively, antibiotically, surgically, or with a combination of these options. We only included patients who were considered immunocompetent. All of the included patients were below 10 years at time of treatment. We collected data regarding time to resolution and adverse effects, i.e., skin discoloration, excessive scar formation, fistula formation, persistence of adenopathies after treatment, need for additional treatment, facial nerve paresis/paralysis, or systemic side-effects due to antibiotic treatment. The mean time to resolution (in days) when looking at primary treatments, was shortest in partial excisions (16), followed by complete excisions (19), antibiotic therapy (129), incision and drainage (153), curettage (240), and finally conservative management (280). Taking into account isolated treatments (i.e., both primary and adjuvant), we also observed consistently faster time to resolution in surgical and antibiotic treatments when compared to conservative treatment. Antibiotic therapy (p = 0.003), incision and drainage (p = 0,004) were associated with a significantly higher need for adjuvant treatment. Curettage was associated with a higher incidence of fistula formation (p = 0,006) and higher number of adjuvant treatments (p = 0,002). This study shows a faster resolution of nontuberculous mycobacterial cervicofacial lymphadenitis in children when treated surgically, more specifically when treated with partial or complete lymph node excision. Antibiotic treatment also leads to faster resolution than conservative management. There was a low rate of complications, and no permanent facial nerve damage was reported.

Long-Term Outcome of Surgical Treatments for Nontuberculous Mycobacterial Cervicofacial Lymphadenitis in Children

Information on long-term treatment outcome for nontuberculous mycobacterial (NTM) cervicofacial lymphadenitis in children is scarce. The purpose of this study is to evaluate long-term outcome for surgical treatment, which is the mainstay treatment modality. This case series describes recurrence rates of surgically treated NTM cervicofacial lymphadenitis patients with a follow-up of at least 10 years. The current study data were partially collected from a randomized, prospective, multicenter, multidisciplinary trial (CHIMED study), which was conducted between 2000 and 2006 to determine the optimal treatment for NTM cervicofacial lymphadenitis in children. After the CHIMED trial inclusion ended, our institute continued to serve as a referral center. This enabled us to enlarge the surgical CHIMED cohort by adding patients who were treated during 2007 to 2010 in our center and collect the rest of the current study data. About 427 children with chronic cervicofacial lymphadenopathy were analyzed. Among these, 290 had microbiologically confirmed cervicofacial mycobacterial infections (n=3 Mycobacterium tuberculosis, n=1 Mycobacterium bovis, n=286 NTM). Of these 286 children with NTM cervicofacial lymphadenitis, 189 were treated surgically (median age: 41 months, range: 9-144, 46.0% males). The affected lymph nodes were excised in 151 children (79.9%), and curettage was performed in 38 children (20.1%). One patient (0.07%) experienced a reactivation/recurrence 2 years after surgical excision and required another surgical excision. Three children (7.9%) experienced infection reactivation/recurrences after curettage, confirmed by redness or a draining fistula, within the first year after healing. Two of these 3 patients were treated with additional surgical excisions. The long-term outcome of surgical excision for NTM cervicofacial lymphadenitis is favorable with a low recurrence rate. Curettage or a conservative wait-and-see approach can be considered an alternative in advanced and surgically challenging cases. However, healing will take longer, and late recurrences are possible.

Long-term outcome of nonsurgical treatment of nontuberculous mycobacterial cervicofacial lymphadenitis in children

Chronic cervicofacial lymphadenitis in children is often caused by nontuberculous mycobacteria (NTM). Children with NTM infection who were not surgically treated were evaluated for long-term outcome with a follow-up of at least 10 years. Among the 117 nonsurgically treated children, the median age was 46 months (range, 9-155 months), 56 were male (47.9%), and 61 were female (52.1%). Of the overall group, 75 received antibiotic therapy consisting of clarithromycin and rifabutin (64.1%), and for 54, observation (a wait-and-see approach) was chosen (46.2%). In 100 patients, treatment was considered successful (85%), with a median resolution of 24 (range, 11-134) weeks in the antibiotic group compared to 44.5 (range, 18-130) weeks in the wait-and-see group (P < .05). After 6 months, 58 patients in the antibiotic group were successfully treated (77%), whereas 42 patients of the wait-and-see group demonstrated complete resolution after a median observation time of 44.5 weeks (100%). In 10 patients who experienced complete resolution of the lymphadenitis, infection recurrence developed years later (10%). Nonsurgical treatment of NTM infection can be considered an alternative in advanced and surgically challenging cases. However, healing will take months to years, and late recurrences are possible.

Nontuberculous mycobacterial cervicofacial lymphadenitis in children from the multicenter, randomized, controlled trial in The Netherlands: Relevance of polymorphisms in candidate host immunity genes

Objective The annual incidence of nontuberculous mycobacterial (NTM) cervicofacial lymphadenitis in otherwise healthy children is unexpectedly high (8 per million). It mostly arises as localized cervicofacial lymphadenitis. Previous research has suggested environmental risk factors for oral exposure to NTM and a temporal association with eruption of teeth. We studied 22 polymorphisms in relevant candidate genes, some related to periodontitis, in children with NTM lymphadenitis. We also tested for the most common mutation in IFNGR1. Methods We analyzed DNA from 81 Dutch children with NTM from a nationwide surveillance study and 215 community controls for 22 polymorphisms in CD209, IL1B, IL8, IL10, IL12B, IL12RB1, IL18, PTX3, TLR4, TNF, VDR and SLC11A1 by MassArray platform (Sequenom) and CONTING. We screened for 818del4 in IFNGR1 by PCR and VspI restriction enzyme cleavage. Results We found a positive association between NTM lymphadenitis and +3953TT in IL1B (OR 2.9; 95%-CI: 1.2–7.2). Furthermore, our results showed that −592C/A heterozygosity in IL10 is linked to protection from disease (OR 0.54; 95%-CI: 0.3–0.95), but that other polymorphisms were unrelated to localized NTM disease. However, these associations were not robust to Bonferroni's correction for multiple testing. None of the children carried the IFNGR1 818del4 mutation. Conclusions Dominance of environmental factors over genetic ones and insufficient sample size might explain the fragility of this study's results. Nevertheless, the association between NTM lymphadenitis and 3953C>T, a polymorphism previously linked to periodontitis, supports our hypothesis that oral exposure to mycobacteria during eruption of teeth plays a role in the etiology of cervical NTM lymphadenitis.

Treatment of non‐tuberculous mycobacterial cervicofacial lymphadenitis in children: critical appraisal of the literature

Surgical excision has historically been the treatment of choice for non-tuberculous mycobacterial cervicofacial lymphadenitis. Emerging data suggests antibiotic treatment alone could be an attractive alternative to surgery. We questioned (1) what treatment offers best cure rates in children with this condition and (2) the evidence for antibiotic treatment alone. Structured literature search according to the Evidence Based Medicine guidelines. SEARCH STRATEGY/METHODS: A structured search was conducted in PubMed, Embase and the Cochrane Library from 1966 up to November 2007. Relevant papers were critically appraised. Six papers were included, one of which was a randomised controlled clinical trial (RCT). In general, treatment sequencing was not consistent. Surgery and medical treatment were often used in combination, resulting in high overall cure rates. In the RCT surgery and antibiotic treatment were compared as single modalities. The outcome of surgery was far superior (96%versus 66% cure rate, respectively). Other studies indicated that substantial cure rates (up to 67%) were achieved with medical management alone, but failed to identify factors predicting response. Surgical excision still is the backbone in the management of cervicofacial non-tuberculous mycobacterial lymphadenitis, although a considerable number of children can be cured with antibiotics alone or combined modality treatment. To date it is unclear which subset of patients can benefit from antibiotic treatment only.

Treatment outcomes for nontuberculous mycobacterial cervicofacial lymphadenitis in children based on the type of surgical intervention

To report treatment outcomes of nontuberculous mycobacteria (NTM) cervicofacial lymphadenitis. A retrospective study from 1995 to 2006. Fifty-one patients (mean age, 30.3 months). All but one patient presented with lymphadenopathy. The mean time from symptoms to diagnosis was 42.3 days. Tuberculin skin testing (TST) was performed on 21 of 51 patients and was positive on 14 of 21 patients. Imaging was ordered in 23; 19 had CT scans. Pathology confirmed caseating granuloma in 45 (88%); cultures grew Mycobacterium avium-intracellulare complex in 46 of 51. Interventions included fine-needle aspiration (5, 10%), incision and drainage +/- curettage (24, 46%), and complete excisional biopsy (20, 38%). Of those who underwent complete excisional biopsy initially, 95 percent were cured compared with 32 of 51 (63%) with nonexcisional surgery. Macrolides and rifampin were more likely to be recommended in referred cases and when nonexcisional surgical treatment was not feasible or had failed. The average total duration of antibiotic therapy was 89.1 days. NTM infections require high levels of suspicion for timely diagnosis, and complete excisional biopsy results in least likelihood of persistent/recurrent disease.

Surgical Excision versus Antibiotic Treatment for Nontuberculous Mycobacterial Cervicofacial Lymphadenitis in Children: A Multicenter, Randomized, Controlled Trial

The optimal treatment of nontuberculosis mycobacterial cervical lymphadenitis in children has not been established. Until recently, surgical excision was the standard treatment, but the number of reports of successful antibiotic treatment is increasing, which questions whether surgery is the preferred treatment. In this randomized, multicenter trial, we compared surgical excision with antibiotic treatment. One hundred children with microbiologically proven nontuberculous mycobacterial cervicofacial lymphadenitis were randomly assigned to undergo surgical excision of the involved lymph nodes or to receive antibiotic therapy with clarithromycin and rifabutin for at least 12 weeks. The primary end point was cure, defined as regression of the lymph node enlargement by at least 75%, with cure of the fistula and total skin closure without local recurrence or de novo lesions after 6 months, as assessed by clinical and ultrasound evaluation. Secondary end points included complications of surgery and adverse effects of antibiotic therapy. Intention-to-treat analysis revealed that surgical excision was more effective than antibiotic therapy (cure rates, 96% and 66%, respectively; 95% confidence interval for the difference, 16%-44%). Treatment failures were explained neither by noncompliance nor by baseline or acquired in vitro resistance to clarithromycin or rifabutin. Surgical complications were seen in 14 (28%) of 50 patients; staphylococcal wound infection occurred in 6 patients, and a permanent grade 2 facial marginal branch dysfunction occurred in 1 patient. The vast majority of patients who were allocated to antibiotic therapy reported adverse effects (39 [78%] of 50 patients), including 4 patients who had to discontinue treatment. Surgical excision is more effective than antibiotic treatment for children with nontuberculous mycobacterial cervicofacial lymphadenitis.

Tuberculin Skin Testing Is Useful in the Screening for Nontuberculous Mycobacterial Cervicofacial Lymphadenitis in Children

We evaluated the diagnostic usefulness of tuberculin skin testing in the screening for nontuberculous mycobacterial (NTM) infection in children. We enrolled 180 children who had chronic cervicofacial lymphadenitis in our study. Skin testing was done using antigens of Mycobacterium tuberculosis, Mycobacterium avium, Mycobacterium kansasii, and Mycobacterium scrophulaceum. The reference standard for NTM infection was a positive culture result, identification by PCR, or both. Receiver operating characteristic analysis was used to identify the optimal cutoff point in skin induration for the detection of NTM infection. Accuracy of the mycobacterial skin tests was quantified using sensitivity and specificity rates and positive and negative predictive values at the optimal skin induration cutoff. A total of 112 NTM infections were identified, of which 83 were caused by M. avium, 21 by Mycobacterium haemophilum, and 8 by other NTM species. At the optimal cutoff for a positive test (5 mm), tuberculin skin testing had a sensitivity and specificity of 70% and 98%, respectively, and a positive predictive value and a negative predictive value of 98% and 64%, respectively, compared with a sensitivity and a specificity of 93% and 97%, respectively; M. avium sensitin, the best-performing skin test, had positive and negative predictive values of 98% and 90%, respectively. Tuberculin skin testing could be valuable as a first step in the diagnostic analysis of cervicofacial lymphadenitis in children without a history of TB exposure or bacille Calmette-Guérin vaccination.

Atypical mycobacterial cervicofacial lymphadenitis in children: A disease as old as mankind, yet a persistent challenge

The diagnosis of atypical cervicofacial lymphadenitis is often not readily clinically apparent. Parents may bring a child to the physician's attention long before the development of the classic findings. When assessing the child with acute-onset cervicofacial lymphadenitis, there are 2 imperative clinical determinations: Is the lymphadenitis of viral, bacterial, or mycobacterial etiology? If of mycobacterial etiology, is it an atypical or tuberculous mycobacterial process? The goal in management is to avoid misdiagnosis and lengthy duration of symptoms before appropriate therapy is instituted. This paper includes a case study–based review of the evaluation, diagnosis, and management of cervicofacial lymphadenitis in children due to atypical mycobacterial organisms.