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

PURPOSE: 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. METHODS: 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. RESULTS: 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). CONCLUSIONS: 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.

The diagnosis of nontuberculous cervicofacial lymphadenitis: A systematic review.

BACKGROUND: Nontuberculous mycobacterial cervicofacial lymphadenitis (NTMCL) is an uncommon condition detected in young immunocompetent children who typically present with a nontender neck mass. Various tests have been proposed to assist in the work-up of suspected NTMCL, with varying diagnostic utility. This systematic review investigates the sensitivity of the various diagnostic methods used in the work-up of pediatric NTMCL. METHODS: A systematic review in accordance with PRISMA guidelines was performed using the Pubmed, EMBASE, and Web of Science databases. Searches were filtered for English language studies published prior to 05/10/22. Studies meeting criteria included studies featuring 15+ pediatric patients with confirmed or suspected NTMCL. Studies with any reported diagnostic methodology used in the workup of NTMCL were included. RESULTS: Of 836 abstracts/articles reviewed, 21 studies met inclusion criteria. Diagnostic methods included culture(n = 11 studies), PPD-Tb(Tuberculin)(n = 12), PPD-Scrofulaceum, -Avium, or -Kansasii(n = 6), staining techniques(n = 4), IGRA(n = 3), and ultrasound(n = 2). All studies had an overall low risk of bias. Among patients confirmed to have NTMCL based on PCR and/or culture, the most sensitive tests were PPD-A(0.94, 95 % CI 0.91 to 0.97; n = 210 patients) and PPD-S(0.75, 95 % CI 0.68 to 0.81; n = 171). Auramine and Ziehl-Neelsen staining techniques had moderately high sensitivity(0.85 and 0.60 respectively), though were limited by low patient numbers(n = 20). PPD-Tb(0.45, 95 % CI 0.39 to 0.50; n = 300) and IGRA(0.02; 95 % CI 0 to 0.06; n = 48) demonstrated poor sensitivity. Among patients suspected to have NTM lymphadenitis based on global assessment, the most sensitive tests included combined PPD-S + A + K(0.92, 95 % CI 0.86 to 0.98; n = 85), PCR(0.82, 95 % CI 0.75 to 0.88; n = 136), and PPD-A(0.72, 95 % CI 0.62 to 0.81; n = 84). Culture showed a sensitivity of 0.54(95 % CI 0.50 to 0.58; n = 494). PPD-K, PPD-S, IGRA, and staining techniques demonstrated lower sensitivity. CONCLUSIONS: This systematic review is the largest study investigating the sensitivity of the various diagnostic methods used in the work-up of pediatric NTMCL. Patients with clinical suspicion for NTMCL and a positive PPD-Tb should first have tuberculous lymphadenitis ruled out with IGRA. Patients with a positive PPD-Tb and negative IGRA and high clinical suspicion for NTMCL can undergo presumptive surgical intervention. Patients with a negative PPD-Tb can undergo NTM antigen skin testing if available, or if high clinical suspicion exists, surgical intervention to reduce tissue burden and elicit additional tissue data.

Successful addition of topical antibiotic treatment after surgery in treatment-refractory nontuberculous mycobacterial skin and soft tissue infections.

Treatment of skin and soft tissue infections with nontuberculous mycobacteria sometimes fails despite repeated debridements and long-term systemic antibiotic therapy. These treatment-refractory infections can cause significant morbidity and pose a treatment challenge. Following surgery, we treated three patients with negative pressure wound therapy with the instillation and dwell time of topical antibiotics, in addition to systemic antibiotic treatment. Treatment was successful and well tolerated, except for some local irritation.

Atypical mycobacterium infection following anterior cruciate ligament reconstruction.

A man in his 20s presented with instability of the right knee following an incident of fall from a height. He was clinicoradiologically diagnosed to have an anterior cruciate ligament (ACL) tear for which he underwent ACL reconstruction. Postoperatively, he was started on an accelerated ACL rehabilitation protocol. Six weeks postoperatively, he developed features of subclinical septic arthritis for which he underwent arthroscopic debridement. Intraoperative samples cultured Mycobacterium abscessus complex on MGIT 960 system. The patient subsequently had to undergo another debridement after 1 month as there were clinical signs of persisting infection. The graft was intact even on the second debridement and after removing the implants. This case reports a rare complication of ACL reconstruction with infection by atypical mycobacterium and the clinical outcome. It also emphasises that prompt surgical intervention can save the graft.

Surgical Resection in Nontuberculous Mycobacterial Pulmonary Disease.

Rates of nontuberculous mycobacterial pulmonary disease are increasing worldwide, particularly in the United States and other developed countries. While multidrug antimicrobial therapy is the mainstay of treatment, surgical resection has emerged as an important adjunct. In this article, we will review the indications for surgery, preoperative considerations, surgical techniques, and postoperative outcomes.

Long-Term Esthetic Outcome of Different Treatment Modalities for Nontuberculous Mycobacterial Cervicofacial Lymphadenitis.

BACKGROUND: Nontuberculous mycobacterial (NTM) cervicofacial lymphadenitis is a rare infection which almost exclusively occurs in children, most commonly children 0-5 years old. It can leave scars in highly visible areas. The present study aimed to evaluate the long-term esthetic outcome of different treatment modalities for NTM cervicofacial lymphadenitis. METHODS: This retrospective cohort study included 92 participants with a history of bacteriologically proven NTM cervicofacial lymphadenitis. All patients were diagnosed at least 10 years prior and were aged >12 years upon enrollment. Based on standardized photographs, the scars were assessed by subjects with the Patient Scar Assessment Scale, and by five independent observers with the revised and weighted Observer Scar Assessment Scale. RESULTS: The mean age at initial presentation was 3,9 years and the mean follow-up time was 15.24 years. Initial treatments included surgical treatment (n = 53), antibiotic treatment (n = 29) and watchful waiting (n = 10). Subsequent surgery was performed in two patients, due to a recurrence after initial surgical treatment, and in 10 patients initially treated with antibiotic treatment or watchful waiting. Esthetic outcomes were statistically significantly better with initial surgery, compared to initial non-surgical treatment, based on patient scores of scar thickness, and based on observer scores of scar thickness, surface appearance, general appearance and the revised and weighted sum score of all assessment items. CONCLUSIONS: The long-term esthetic outcome of surgical treatment was superior to non-surgical treatment. These findings could facilitate the process of shared decision making. LEVEL OF EVIDENCE: Level III.

Surgical Treatment of Nontuberculous Mycobacterial Pulmonary Disease and a Combination of Nontuberculous Mycobacterium Pulmonary Disease and Pulmonary Tuberculosis.

OBJECTIVES: Nontuberculous mycobacterial pulmonary disease (NTMPD) is an important health system problem worldwide. Surgical treatment experience is limited, and the safety of such operations is still unclarified. Therefore, improving our knowledge of NTMPD is important. DESIGN: In this retrospective observational study, we analyzed the medical records of 2,432 patients operated on from January 2011 through December 2017 in our hospital. There were 20 patients with NTMPD in group 1 and 23 patients with combined NTMPD and pulmonary tuberculosis in group 2. Patients received antibiotic treatment before and after surgery as per Russian Federal clinical guidelines and individual drug susceptibility. RESULTS: Postoperative complications occurred in 2 (10%) cases in group 1 and 4 (17.4%) in group 2, and intraoperative complications occurred in 2 (8.7%) cases in group 2. There was no 30-day mortality in both groups. After 1 year, all 40 traced patients had neither bacterial excretion nor cavities in the lungs. After 3 years and 5 years, the efficacy among 32 and 13 traced patients was 100%, respectively. CONCLUSIONS: Modern surgery for small NTMPD forms is safe - helping improve outcomes for ineffective antibiotic treatment. Combined surgery and antibiotic treatment are acceptable in both the short and long term.

Outcomes of Surgical Treatment for Mycobacterium abscessus Complex Pulmonary Disease.

BACKGROUND: Mycobacterium abscessus complex pulmonary disease is notoriously difficult to treat by medication alone. We report our experience with resectional surgery combined with preoperative and postoperative multidrug chemotherapy for the treatment of patients with M. abscessus complex pulmonary disease. METHODS: This is a retrospective review of 33 patients undergoing lung resection for M. abscessus complex pulmonary disease at a single center in Japan between January 2008 and December 2019. RESULTS: The median age of patients was 54.0 (interquartile range [IQR], 49.0-66.0) years; 27 (81.8%) were female. Nodular-bronchiectatic was the most common disease type (n = 24, 72.7%). Disease was limited in 18 (54.5%) patients and extensive in 15 (45.5%). The median duration of preoperative multidrug chemotherapy employing oral and parenteral antibiotics was 10.0 (IQR, 3.0-18.0) months. A total of 34 anatomical lung resections were performed as follows: 22 lobectomies, 5 segmentectomies, 4 combined resections, 2 bilobectomies, and 1 pneumonectomy. No operative mortalities and 4 (13.3%) morbidities occurred. The median duration of multidrug chemotherapy after the surgery was 18.0 (IQR, 12.0-31.0) months. Postoperative sputum-negative status was achieved in 31 (93.9%) patients; all 23 patients obtaining preoperative negative conversion remained negative, and 8 (80.0%) of 10 patients with preoperative positive sputum became negative postoperatively. Recurrence was observed in 2 (6.5%) patients. The recurrence-free probabilities were 96.3%, 96.3%, and 80.2% at 1 year, 3 years, and 5 years, respectively. CONCLUSIONS: Combined with preoperative and postoperative multidrug chemotherapy, resectional surgery can be performed safely and achieve favorable outcomes for patients with M. abscessus complex pulmonary disease.

Outcomes of adjunctive surgery for nontuberculous mycobacterial pulmonary disease.

BACKGROUND: Owing to the unsatisfactory results of antibiotic treatment alone, surgical resection is currently considered as adjunctive therapy in patients with nontuberculous mycobacterial pulmonary disease (NTM-PD). However, reports regarding the outcomes of surgery vary considerably by institution. Here, we investigated the surgical outcomes and risk factors associated with unfavorable outcomes after surgery. METHODS: We analyzed patients with NTM-PD who underwent pulmonary resection at Seoul National University Hospital between January 1, 2006, and December 31, 2020, and assessed the types of surgical procedures, complications, and long-term outcomes. Multivariate logistic regression analysis was used to identify the risk factors associated with treatment refractoriness or recurrence after surgery. RESULTS: Among 67 patients who underwent surgery during the study period, the most common indication for surgery was persistent culture positivity despite rigorous medical treatment (80.6%), followed by longstanding cavitary lesions or radiographic aggravation (10.4%) and massive hemoptysis (4.5%). Among 53 patients with positive mycobacterial cultures at the time of surgery, 38 (71.7%) achieved initial negative culture conversion, 9 (17.0%) of whom experienced recurrence. Nine (13.4%) patients experienced postoperative complications, which were managed without lasting morbidity and mortality. Female sex (adjusted odds ratio [aOR] 6.63; 95% confidence interval [CI] 1.04-42.4; P = .046), preoperative positive mycobacterial culture (aOR 5.87; 95 %CI 1.04-33.08; P = .045), and residual lesions (aOR 6.86; 95 %CI 1.49-31.56; P = .013) were associated with refractoriness or recurrence. CONCLUSIONS: Pulmonary resection is a reasonable treatment modality for patients with refractory NTM-PD or major complications such as massive hemoptysis. The potential risk factors associated with unfavorable outcomes included female sex, preoperative positive mycobacterial culture, and residual lesions after surgery.

Arthroscopic treatment of Mycobacterium massiliense septic arthritis outbreak after intra-articular injection: A case-series report and literature review.

ABSTRACT: Non-tuberculous mycobacteria (NTM) comprise mycobacteria, with the exceptions of Mycobacterium (M.) leprae and the M. tuberculosis complex. Septic arthritis caused by NTM is so rare that there is no standardized treatment.Between April and September 2012, 27 patients were infected with M. massiliense in a single clinic following injection of steroid in the knee joint. Clinical data of 9 patients who received arthroscopic treatment in Seoul Hospital of Soonchunhyang University were analyzed retrospectively.Arthroscopic irrigation and debridement were performed average 2.6 times (1-3 times). As 6 out of 9 cases (67%) had joint contracture of the knee joint, arthroscopic adhesiolysis, and brisement were performed. After surgical procedures, Hospital for Special Surgery and Lysholm knee score showed improvement compared before the surgery, but a radiographic result evaluated by Kellgren-Lawrence revealed that 6 cases got deteriorated to stage 4 in the 4-year follow-up.NTM septic arthritis had a higher recurrence and a higher contracture incidence than septic arthritis caused by tuberculous mycobacteria or other bacteria. Treatment was possible with repeated arthroscopic debridement and intravenous antibiotics.

Chest wall pseudotumor: a case of non-tuberculous mycobacterial infection.

BACKGROUND: Non-tuberculous mycobacterial (NTM) infections are increasing worldwide, making them an international public health problem. Surgical management is often indicated for localized infectious disease; however, most surgeons are unaware of the potential risks of transmission during surgery. CASE PRESENTATION: An 88-year-old Asian female was referred to our hospital for a tumor in the right lateral thoracic region. One month prior, she had a feeling of fullness and complained of localized pain and warmth in the right lateral thoracic wall. Pain and warmth gradually resolved without intervention; however, the fullness was getting worse. Computed tomography (CT) scan showed a mass of approximately 65 × 30 mm with an osteolytic change, involving the right 8th rib. Based on the rapid growth rate and CT findings, we strongly suspected a malignant chest wall tumor, and en bloc tumor resection with the 8th rib was performed. When the specimen was cut, a large amount of viscous pus was drained and its culture showed growth of Mycobacterium avium. Microscopically, the non-caseating epithelioid cell granuloma extended into the rib, infiltrating the bone cortex. On follow-up 1 month after discharge, there were no signs of infection or other adverse events associated with the surgery. CONCLUSIONS: Herein, we report about a patient with a mass diagnosed as an NTM abscess involving the rib cage, which was confused with a malignant tumor and eventually diagnosed following surgical excision. This report emphasizes the need to be aware of the possibility of NTM infection and take appropriate precautions if the patient has a rapidly growing mass in the chest wall.

Thoracoscopic right middle lobectomy for Mycobacterium abscessus in a young patient suspected of having congenital immunodeficiency.

Mycobacterium abscessus (M. abscessus) infection is resistant to multi-antibacterial treatment, and surgical resection is often recommended. We report a case of M. abscessus infection in a young patient suspected of having a GATA2 mutation. A 19-year-old woman with a medical history of severe sinusitis and a family history of non-tuberculous mycobacteriosis presented at our hospital. M. abscessus was confirmed by sputum culture. The patient received multidrug therapy, including clarithromycin. CT scan demonstrated bronchodilation and capacity decrease due to non-obstructive atelectasis in the middle lobe. We performed thoracoscopic resection without complications. Congenital immunodeficiency was suspected given the patient's past medical and family history. The result of lymphocyte subset analysis revealed a GATA2 mutation, but no genetic mutation was detected by a next-generation sequencer. The patient followed a good clinical course. This paper reports the successful treatment of an M. abscessus infection and the importance of checking the genetic background of young patients.

Extensive Lung Resection for Nontuberculous Mycobacterial Lung Disease With Multilobar Lesions.

BACKGROUND: Nontuberculous mycobacterial lung disease often spreads to multiple lobes, and extensive lung resection (ELR) is sometimes required to control the disease. The safety and feasibility of ELR for nontuberculous mycobacterial lung disease remain unclear, however. METHODS: This retrospective study included patients with nontuberculous mycobacterial lung disease who underwent adjuvant lung resection. Characteristics were compared between patients who underwent ELR and those who underwent simple anatomic lung resection (SALR). The outcome data were analyzed by a Cox regression analysis. RESULTS: A total of 146 patients underwent ELR (n = 54) or SALR (n = 92). ELR was associated with a longer operative time (306 vs 237 minutes; P < .001) and higher incidence of prolonged air leak (17% vs 3.3%; P = .016) than SALR. Rates of mortality, sputum culture conversion (positive to negative), and microbiological recurrence did not differ markedly between the groups. In the multivariate analysis, ELR was not a significant risk factor for an unfavorable outcome after nontuberculous mycobacterial lung disease surgery (hazard ratio, 2.23; 95% confidence interval, 0.82-6.03; P= .11). CONCLUSIONS: ELR for nontuberculous mycobacterial lung disease has some drawbacks compared with SALR but seems as safe and feasible as SALR. ELR may provide improved disease control in some cases of nontuberculous mycobacterial lung disease with multilobar lesions.

The impact of adjuvant surgical treatment of nontuberculous mycobacterial pulmonary disease on prognosis and outcome.

BACKGROUND: Lung resection in patients with nontuberculous mycobacterial pulmonary disease (NTM-PD) has been reported to be associated with favorable outcomes. However, little is known regarding the risk and prognostic factors for refractory and recurrent cases. We aimed to evaluate the overall impact and benefit of adjuvant lung surgery by comparing NTM-PD patients who underwent adjuvant lung resection with those treated exclusively with antibiotics. We also investigated the efficacy of serum IgA antibody against glycopeptidolipid (GPL) core antigen (GPL core antibody) to monitor disease activity and predict the recurrence of disease after adjuvant lung resection. METHODS: We retrospectively evaluated the clinical characteristics and surgical outcomes of 35 patients surgically treated for NTM-PD. Furthermore, we compared surgically treated patients and control patients treated exclusively with antibiotics who were matched statistically 1:1 using a propensity score calculated from age, sex, body mass index, and radiologic features of disease. RESULTS: In the surgically treated patients, the median age was 58 (interquartile range, 47-65) years and 65.7% were female. Twenty-eight patients had Mycobacterium avium complex. Operations comprised four pneumonectomies, two bilobectomies, one bilobectomy plus segmentectomy, 17 lobectomies, two segmentectomies, and nine lobectomies plus segmentectomies. Postoperative complications occurred in seven patients (20%), there were no operative deaths, and 33 (94.3%) patients achieved negative sputum culture conversion. Refractory and recurrent cases were associated with remnant bronchiectasis, contralateral shadows, and positive acid-fast bacilli staining or culture. Of 28 statistically matched pairs, long-term sustained negative culture conversion was observed in 23 (82.2%) surgical group patients and in 14 (50.0%) non-surgical group patients (0.0438). The mortality rate was lower in the surgical group, but did not reach statistical significance (one in the surgical group and four in the non-surgical group, p = 0.3516). GPL core antibody was correlated with disease activity and recurrence. CONCLUSIONS: NTM-PD patients who underwent adjuvant lung resection experienced overall favorable outcomes and achieved sputum culture conversion more frequently. Long-term mortality may have been reduced by this procedure, and the level of GPL core antibody was shown to be a good clinical indicator of disease activity after surgery.

Recent advances and controversies in surgical intervention of nontuberculous mycobacterial lung disease: A literature review.

The prevalence of nontuberculous mycobacterial lung disease (NTM-LD) has increased in Western and Asian nations in recent decades. While surgery may improve the outcome of more complex cases, many inconsistencies exist in the current literature regarding the management, growing emergence, and challenges of drug-resistant forms of NTM-LD, the indications and timing of surgical treatment, and perioperative multimodal therapy of NTM-LD. Moreover, data regarding the comparative treatments, risk factors of pulmonary resection for NTM-LD, and the long-term outcomes of microbiological recurrence are limited. This review will focus on outlining the outcomes of recently optimized surgical approaches, as well as providing an overview of the roles of perioperative multimodalities therapies in the treatment of NTM-LD.

Submandibular lymphadenitis due to Mycobacterium malmoense: first reported case from Greece.

An 8-year-old girl with subacute submandibular lymphadenitis and no other complaints is described. After failure of parenteral antistaphylococcal therapy, she underwent incision and drainage of the involved lymph node. The responsible pathogen was identified as Mycobacterium malmoense by GenoType CM assay and sequencing of the 16S ribosomalRNA (rRNA) gene. The patient remains healthy, 11 months after surgery, even though it took approximately 4 months for the surgical incision to heal completely. While M. malmoense is a relatively common cause of non-tuberculous mycobacteria (NTM) lymphadenitis in Northern Europe, this is the first reported case from Greece. We conclude that in a young child with lymphadenitis without systemic symptoms, the microbiology laboratory should be notified in advance in order to extend the duration of mycobacterial cultures. Application of molecular methods will increase the number of reported cases of rare NTM in the future.

Pulmonary resection for nontuberculous mycobacterial pulmonary disease: outcomes and risk factors for recurrence.

OBJECTIVES: As the number of patients with nontuberculous mycobacterial pulmonary disease (NTMPD) increases, surgical treatment to control disease becomes more important. However, postoperative outcomes and predictors of recurrence have been insufficiently evaluated. METHODS: We retrospectively investigated 100 patients with NTMPD who underwent pulmonary resection from 2009 to 2016 at our institution. Clinical data of patients with and without postoperative recurrence were statistically compared. Recurrence was defined as microbiological re-identification or computed tomography findings highly suspicious for relapse after excluding other diseases. Recurrence-free survival was calculated using the Kaplan-Meier method. Survival curves were compared using the log-rank test. Predictive factors were evaluated using univariate and multivariate analyses. RESULTS: Nine patients experienced recurrence. A significant difference in recurrence-free survival was detected between patients with and without a positive preoperative sputum culture (P = 0.000942). Moreover, patients with a positive preoperative sputum smear (≥ 2 +) had a significantly higher recurrence rate than those who did not (P = 0.000216). Multivariate analysis revealed that preoperative sputum smear (≥ 2 +) is an independent risk factor for recurrence after pulmonary resection for NTMPD (odds ratio, 7.38; 95% confidential interval, 1.29-42.2; P = 0.024). CONCLUSIONS: NTM discharge might have an impact on postoperative recurrence of NTMPD patients without residual cavitary lesions. Preoperative NTM discharge should be minimized by optimizing medical therapy before surgical treatment to improve the postoperative course. Intensive follow-up and prolonged postoperative medical therapy should be considered for patients without a sufficient reduction in bacterial discharge before pulmonary resection.