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Nontuberculous mycobacterial lymphadenitis [keywords]
- [Surgical treatment of children with nontuberculous mycobacteria cervical lymphadenitis.] [JOURNAL ARTICLE]
- HNO 2014 Jul 11.
Persistent cervical lymphadenopathy is the typical clinical manifestation of nontuberculous mycobacterial (NTM) infection in otherwise healthy children 1-5 years of age. A positive NTM culture or PCR is necessary to proof the diagnosis. In the case of localized disease, cervical lymphadenectomy simultaneously serves both diagnosis and therapy. A typical complication of surgical treatment, i.e. incision, puncture or excision, is the formation of a fistula, which then requires further surgical intervention. In the case of an unconfirmed diagnosis, the extent of the initial surgical intervention remains unclear.On the basis of this diagnosis, 17 operations were performed in 10 children under the age of 7 years (8 female, 2 male; age 17 months to 5 years, median 36 months) in the Charité ENT clinics between 2009 and 2012. Clinical course and diagnostics, as well as the results of therapies and treatments were retrospectively analysed.Duration of anamnesis prior to initial surgery was 2-30 weeks (mean 10.4 weeks). A second intervention was performed in 7 out of 10 patients. No patient developed recurrent disease after selective cervical lymphadenectomy. The clinical course of 1 patient was complicated by a cefuroxime-responsive Staph. aureus superinfection. A second patient experienced transient accessory nerve paresis after lymphadenectomy, which resolved 2 months after the second surgery.In case of persistent cervical lymphadenopathy a complete diagnostic workup is necessary. If lymphadenopathy continues to persist 1 month after a 10-day course of broad-spectrum antibiotics, a selective cervical lymphadenectomy should be performed. In order to avoid the development of fistulae and avoid secondary surgical procedures, incision, drainage and puncture should be deferred.
- Histology of solid lateral cervical masses biopsied in children. [Journal Article]
- Int J Pediatr Otorhinolaryngol 2014 Jan; 78(1):39-45.
Solid cervical lateral neck masses in children may require surgical biopsy to confirm appropriate diagnostic and begin a directed therapeutic treatment. We aimed to describe the contribution of pathological results and compare them with the clinical diagnosis and the paraclinical tools.A retrospective review of surgical biopsies for solid lateral neck masses in children over a ten year period in a pediatric tertiary center was conducted. Demographic, imaging, laboratory analysis, surgical and pathological data were collected and analyzed using descriptive statistics with SPSS 17.0.44 biopsies were done between 2002 and 2012. Inflammatory masses were found in 26/44 biopsies with half of them (13/26) being nontuberculous mycobacterial (NTM) lymphadenitis. Non-inflammatory/benign masses represented 9/44 biopsies and 5/44 masses were of malignant etiology. Malignant masses imaging had a sensitivity and specificity of 33% and 75%, respectively, for ultrasound, whereas Neck CT scan had 33% and 77%, respectively. The contribution of pathological results to the clinical management was questionable in 39% (17/44) of biopsies.Inflammatory masses with NTM lymphadenitis were the most common diagnosis. Imaging was not helpful in establishing the diagnosis. Heterogeneity in the management of solid lateral neck masses between clinicians was important and indicates the need for guideline approach.
- Comparison of Mycobacterium lentiflavum and Mycobacterium avium-intracellulare complex lymphadenitis. [Journal Article]
- Pediatr Infect Dis J 2014 Jan; 33(1):28-34.
Mycobacterium lentiflavum is considered a rare pathogen causing nontuberculous mycobacterial (NTM) lymphadenitis.A multicenter, retrospective study was performed in immunocompetent children <14 years of age with microbiologically confirmed NTM lymphadenitis treated at 6 hospitals in Madrid, Spain, during 2000-2010. We compared children with M. lentiflavum and Mycobacterium avium-intracellulare complex infection.Forty-five microbiologically confirmed NTM lymphadenitis patients were identified: 19 (45.2%) caused by M. avium-intracellulare complex, 17 (40.5%) by M. lentiflavum, 1 by both and 5 by other mycobacteria. Out of 17 M. lentiflavum cases, 14 were diagnosed in the past 5 years. Regarding M. lentiflavum cases, median age was 23 months. Submandibular nodes were the most frequently involved (76.5%), with multiple locations seen in 41% of the children and spontaneous drainage in 41% of them. Drug susceptibility tests were performed in 14 isolates and showed a complete susceptibility to clarithromycin and cycloserine, whereas 93% were resistant to rifampin, 33% to quinolones and full resistance to other tested antimycobacterial drugs was detected. All but 1 child required surgery and 11 were treated additionally with various drug combinations. Total resolution was achieved in 50% of children within 6 months.Compared with M. avium-intracellulare complex cases, children were younger and laterocervical nodes were significantly less frequently involved. No statistically significant differences were found related to clinical characteristics, treatment and outcome.M. lentiflavum is an emerging pathogen producing NTM lymphadenitis in Madrid.
- A novel homozygous p.R1105X mutation of the AP4E1 gene in twins with hereditary spastic paraplegia and mycobacterial disease. [Case Reports, Journal Article, Research Support, N.I.H., Extramural, Research Support, Non-U.S. Gov't]
- PLoS One 2013; 8(3):e58286.
We report identical twins with intellectual disability, progressive spastic paraplegia and short stature, born to a consanguineous family. Intriguingly, both children presented with lymphadenitis caused by the live Bacillus Calmette-Guérin (BCG) vaccine. Two syndromes - hereditary spastic paraplegia (HSP) and mycobacterial disease - thus occurred simultaneously. Whole-exome sequencing (WES) revealed a homozygous nonsense mutation (p.R1105X) of the AP4E1 gene, which was confirmed by Sanger sequencing. The p.R1105X mutation has no effect on AP4E1 mRNA levels, but results in lower levels of AP-4ε protein and of the other components of the AP-4 complex, as shown by western blotting, immunoprecipitation and immunofluorescence. Thus, the C-terminal part of the AP-4ε subunit plays an important role in maintaining the integrity of the AP-4 complex. No abnormalities of the IL-12/IFN-γ axis or oxidative burst pathways were identified. In conclusion, we identified twins with autosomal recessive AP-4 deficiency associated with HSP and mycobacterial disease, suggesting that AP-4 may play important role in the neurological and immunological systems.
- Management of extrapulmonary nontuberculous mycobacterial infections. [Journal Article, Review]
- Semin Respir Crit Care Med 2013 Feb; 34(1):143-50.
Nontuberculous mycobacteria represent a vast group of environmental organisms that have the potential to cause disease in humans. Unlike tuberculosis, these organisms are not known to be transmitted from human to human. The most common clinical presentation is pulmonary disease. Approximately 10% of infections manifest as extrapulmonary disease. The portals of entry are the respiratory tract, gastrointestinal tract, or direct inoculation via trauma or an invasive procedure. Like tuberculosis, the nontuberculous mycobacteria have the potential to infect any organ system given the opportunity in an immunocompromised host. The spectrum of disease is extensive ranging from self-limited furunculosis to life-threatening disseminated infection. Common extrapulmonary manifestations include lymphadenitis, disseminated disease, skin, soft tissue, and bone infection. Less common manifestations include keratitis, catheter-related bloodstream infections, septic arthritis, central nervous system infection, and peritonitis. The incidence of extrapulmonary infections is unknown. Outbreaks have been reported due to inadequate disinfection of surgical equipment or contamination of injected solutions or medications. A high index of suspicion is required when patients present with subacute or chronic complaints of extrapulmonary infection. This review addresses the management of the common extrapulmonary nontuberculous infections.
- Diagnosis of nontuberculous mycobacterial infections. [Journal Article, Review]
- Semin Respir Crit Care Med 2013 Feb; 34(1):103-9.
The nontuberculous mycobacteria (NTM) are typically environmental organisms residing in soil and water. Although generally of low pathogenicity to humans, NTM can cause a wide array of clinical diseases; pulmonary disease is most frequent, followed by lymphadenitis in children, skin disease by M. marinum (particularly in fish tank fanciers), and other extrapulmonary or disseminated infections in severely immunocompromised patients. Of the >140 NTM species reported in the literature, 25 species have been strongly associated with NTM diseases; the remainder are environmental organisms rarely encountered in clinical samples. Correct species identification is very important because NTM species differ in their clinical relevance. Further, NTM differ strongly in their growth rate, temperature tolerance, and drug susceptibility. The diagnosis of NTM disease is complex and requires good communication between clinicians, radiologists, and microbiologists. Isolation of M. kansasii and (in northwestern Europe) M. malmoense from pulmonary specimens usually indicates disease, whereas Mycobacterium gordonae and, to a lesser extent, M. simiae or M. chelonae are typically contaminants rather than causative agents of true disease. Mycobacterium avium complex (MAC), M. xenopi, and M. abscessus form an intermediate category between these two extremes. This review covers the clinical and laboratory diagnosis of NTM diseases and particularities for the different disease types and patient populations. Because of limited sensitivity and specificity of symptoms, radiology, and direct microscopy of clinical samples, culture remains the gold standard. Yet culture is time consuming and demands the use of multiple media types and incubation temperatures to optimize the yield. Outside of reference centers, such elaborate culture algorithms are scarce.
- A case of Mycobacterium goodii prosthetic valve endocarditis in a non-immunocompromised patient: use of 16S rDNA analysis for rapid diagnosis. [Case Reports, Journal Article]
- BMC Infect Dis 2012.:301.
Mycobacterium goodii is a rare cause of significant infection. M. goodii has mainly been associated with lymphadenitis, cellulitis, osteomyelitis, and wound infection.A case of a 76-year-old Caucasian female is presented. The patient developed a prosthetic valve endocarditis caused by M. goodii. She had also suffered from severe neurological symptoms related to a septic emboli that could be demonstrated as an ischemic lesion found on CT of the brain. Transesophageal echocardiography verified a large vegetation attached to the prosthetic valve. Commonly used blood culture bottles showed growth of the bacteria after 3 days.Although M. goodii is rarely involved in these kinds of severe infections, rapidly growing mycobacteria should be recognized during conventional bacterial investigations and identified by molecular tools such as analysis of 16S rDNA. Species identification of nontuberculous mycobacteria is demanding and is preferably done in collaboration with a mycobacterial laboratory. An early diagnosis provides the opportunity for adequate treatment. In the present case, prolonged antimicrobial treatment and surgery with replacement of the prosthetic valve was successful.
- Epithelioid histiocytic infiltrate caused by Mycobacterium scrofulaceum infection: a potential mimic of various neoplastic entities. [Case Reports, Journal Article]
- Am J Dermatopathol 2013 Apr; 35(2):266-9.
Mycobacterium scrofulaceum is a slow-growing atypical mycobacterium that is ubiquitous within our environment found in the water and soil. Most commonly, it manifests as the organism responsible for lymphadenitis in children. In adults, infection by this organism is rare and usually occurs in the setting of local or systemic host immunosuppression. We herein report a case of a 45-year-old woman who presented with a large subcutaneous nodule over her right upper arm. She had been on low dose oral prednisone for 17 years for systemic lupus erythematosus without complication. A biopsy of the nodule revealed a diffuse dermal infiltrate of epithelioid histiocytes laden with acid-fast bacilli mimicking a fibrohistiocytic neoplasm. Treatment with clarithromycin monotherapy resulted in clinical remission. Consideration of atypical mycobacterial infection is warranted in fibrohistiocytic proliferations of the skin especially within immunosuppressed patients.
- [Nontuberculous mycobacterial epitrochlear adenitis]. [Case Reports, English Abstract, Journal Article]
- Arch Pediatr 2012 Oct; 19(10):1070-3.
We report the case of a 2-year-old girl referred for unilateral epitrochlear lymphadenitis caused by Mycobacterium avium. Adenitis is the most frequent presentation of non tuberculous mycobacteria in children. Typical locations are the cervical, submandibular, axillar, inguinal, mediastinal, and parotid regions. To our knowledge, this is the first observation of an epitrochlear location. The diagnosis was made by evidencing the causal bacterium but also by the exclusion of other causes such as Bartonella henselae and Mycobacterium tuberculosis infections. Treatment is based on surgical excision, which provides a cure rate of 90%. Macrolides are reserved for extended lesions and/or relapsing lesions despite surgical management.