Azzali, A

de Martino, M

Galli, L.

Montagnani, C

Simonetti, Mt

Spinelli, G

English

Florence Research

CASE REPORT Open AccessFirst case of Mycobacterium marseillenselymphadenitis in a childA. Azzali1, C. Montagnani2, M. T. Simonetti3, G. Spinelli4, M. de Martino1 and L. Galli1,2*AbstractBackground: Nontuberculous mycobacteria (NTM) are pathogens that commonly affect the paediatric populationand its most frequent manifestation is a cervicofacial lymphadenopathy. With the improvement of technologies,new species have been recently identified.Case presentation: We report the first case of NMT lymphadenitis in a child caused by Mycobacterium marseillense,a newly described species belonging to Mycobacterium avium complex.Conclusions: Improving the identification of these newly discovered mycobacteria, further information will be availableabout their clinical involvement and their best treatment.Keywords: Lymphadenitis, Children, Mycobacterium marseillense, Nontuberculous mycobacteriaBackgroundNontuberculous mycobacteria (NTM) are pathogenscausing different patterns of diseases, ranging from anasymptomatic colonization to a widespread dissemin-ation, depending on the host immune competence [1].NMT can be found in soil, water, milk, eggs, vegetablesand animals. In immunocompetent children, they usuallycause an unilateral, painless, progressive lymphadenop-athy that lacks constitutional symptoms. Submandibularand cervical lymph node stations are mainly involved,while the incidence of preauricolar, inguinal and axillaryarea affection is lower [2].Cervicofacial NTM lymphadenitis affects mainlychildren below 4 years of age, while it is rarely diagnosedin adolescents. The estimated incidence of NTM diseaseis of 0.6 to 1.6 cases per 100,000 children per year and itincreases during spring and late winter [2].More than 150 species of NTM have been recognized,but less than 20 species have been identified in humaninfections [3]. Members of Mycobacterium avium complex(MAC) are the causative agents of more than 75% of casesof NTM lymphadenitis, followed by Mycobacteriumhaemophilum (6%) [4].Mycobacterium marseillense has recently been de-scribed as a new species belonging to the MAC complex[5]. We report on the first case of NMT lymphadenitiscaused by Mycobacterium marseillense in a child.Case presentationWe evaluated a 4 years old girl, previously healthy, withno relevant family medical history. She was conductedto our clinic because of a submandibular lymphadenitis.A month before, during a 3 days fever period, a painfulsubmandibular swelling was noted. Neck ultrasound(US) showed a 2-cm diameter lymphadenopathy with aconserved structure. Therapy with ibuprofen was thensuggested. Because of symptoms persistence, a controlUS was repeated after 5 days. The lymph node was en-larged and presented a subverted structure, with hypoe-chogenic areas and an absent roundness index (Fig. 1).Therapy with co-amoxiclav was immediately started at adosage of 80 mg/kg/day for 6 days. Blood tests showed aslight increase in C-reactive protein values (53 mg/L;normal value <50 mg/L) and erythrocyte sedimentationrate (31 mm/h); serology for Cytomegalovirus confirmeda previous infection, while IgM for Epstein Barr Virus(EBV) were positive. Blood smear was normal. Tuberculinskin test resulted positive (10 mm infiltration diameter).* Correspondence: luisa.galli@unifi.it1Department of Health Sciences, University of Florence, Anna MeyerChildren’s University Hospital, Viale Pieraccini, 24, I-50139 Florence, Italy2Pediatric Infectious Diseases Unit, Anna Meyer Children’s University Hospital,Florence, ItalyFull list of author information is available at the end of the article© The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.Azzali et al. Italian Journal of Pediatrics  (2017) 43:92 DOI 10.1186/s13052-017-0413-5QuantiFERON-TB Gold In-tube test and chest X-rayresulted negative.Neck US performed after 20 days was unchanged.Therefore the girl was referred to our infectious diseaseunit for further investigations. At physical examination,a fixed right submandibular lymphadenopathy, with anelastic consistency, painless and with no superficial skinalterations was detected. No other significant clinicalfeatures were described. Blood tests showed noalteration in blood count, inflammatory markers, liverfunction, lactic dehydrogenase, coagulation. Francisellatularensis and Toxoplasma gondii serology were nega-tive. EBV serology showed evidence of a past infection.In the suspicion of an NTM lymphadenitis, antimicro-bial therapy with clarithromycin (15 mg/kg/die), andrifampicin (20 mg/kg per day) was started. A magneticresonance imaging showed a confluent lymphonodalpack inside the inferior pole of the right parotid gland(4 × 4.2 × 2.9 cm) that was enhanced after contrastmedium infusion. The enhancement involved also thewhole parotid gland parenchyma and the ipsilateralsternocleidomastoid muscle.Due to sudden enlargement of swelling, exeresis of thelymph node was performed. Bacterioscopic examshowed a positivity for acid-fast bacilli, and the histo-logical examination evidenced a granulomatous inflam-mation. Culture from the biopsy grew a M. marseillensestrain, identified using a commercial kit (Hain GenotypeMycobacteria CM, Hain Lifescience, Germany) andsequencing the spacer region interposed between 16Sand 23S rRNA genes, as described previously [6].Because of a slow healing up of the surgical scar, etham-butol (25 mg/kg/die) was added to the previous therapy.Antimicrobial treatment was well tolerated by thepatient and no adverse effect were reported. Blood examsand clinical assessment were periodically performed inorder to evaluate potential drug-induced toxicity. Therapywas finally stopped 2 months after surgery.Periodical blood tests and neck US were performed.At 10 months follow up visit, no relapse was found andthe surgical scar was completely healed.Discussion and conclusionTo the best of our knowledge, this is the first reportedcase of lymphadenitis due to M. marseillense in a child,moreover immunocompetent. NTM infection shouldalways be considered in the differential diagnosis ofchronic lymphadenopathies, especially in children below4 years of age. MAC is the most common cause of NTMlymphadenitis, with M. avium being the species mostfrequently isolated. However, mycobacterial taxonomy isoften a challenge for microbiologists, because of the newspecies that are continuously described due to the appli-cation of molecular techniques [3].M. marseillense was firstly described in 2009. It is aslowly growing, small, acid-fast bacillus, belonging to theMAC complex, showing the closest similarity withMycobacterium chimaera [5].Clinical features of M. marseillense infections are notyet clearly known due to the extreme rarity of these con-ditions. Up to now, only two cases of pulmonary infec-tion and two cases of isolation in the sputum of patientswith cystic fibrosis have been described [7]. One reportwas about a 56 years old woman with systemic lupuserythematosus who presented a story of cough andpurulent sputum with chest consolidation of the rightmiddle lobe at computed tomography (CT) scan,unresponsive to multiple courses of antibiotics. AfterFig. 1 Preoperative ultrasound of patient’s lymph nodeAzzali et al. Italian Journal of Pediatrics  (2017) 43:92 Page 2 of 3bronchoscopy, M. marselleinse was isolated in broncho-alveolar lavage [8]. The other report was about a 65 yearsold immunocompetent man who presented at chestradiographs a diffuse nodular opacity and bronchialthickening, confirmed by high-resolution CT. On bron-choalveolar lavage, a nontuberculous Mycobacteriumstrain was isolated by culture and preliminarily identifiedas M. intracellulare. Rifampin, isoniazid and amikacinwere initially prescribed, with clinical improvement. Inthe subsequent 4 years, the patient presented intermit-tently positive culture despite the various therapeuticcycles. The last drug regimen was with ethambutol,rifampin and azithromycin. Anyway, cough and sputumproduction persisted despite negative microbiologicaltest results. Moreover, radiological follow up evidenced aprogressive pulmonary involvement [9].As no other cases of lymphadenopathy due to M.marseillense were described, no specific indications forthe treatment of this kind of infection were present inthe literature. Excision was performed as indicated inNTM lymphadenitis and allowed the identification ofthe germ. Therapy for NTM infections with macrolideand rifampin was given as suggested by American ThoracicSociety and the Infectious Diseases Society of Americaguidelines [10], and ethambutol was later added due todelay in the healing of the surgical scar. Clinical andradiological follow up showed a decrease of lymph nodedimension with an almost complete recovery at 6 months.M. marseillense role in lymphadenitis could be under-estimated given the similarities to M. intracellulare.Further studies are needed to understand the clinicalinvolvement of these newly discovered mycobacteria inNTM lymphadenitis and their best treatment.AbbreviationsEBV: Epstein Barr Virus; M. marseillense: Mycobacterium marseillense;MAC: Mycobacterium avium complex; NTM: Nontuberculous mycobacteriaAcknowledgementsNot applicable.FundingNo funding to declare.Availability of data and materialsNot applicable.Authors’ contributionsSMT performed the microbiological analysis; SG established the surgicalprocedure. All authors read and approved the final manuscript.Ethics approval and consent to participateNot applicable.Consent for publicationObtained from parents.Competing interestsThe authors declare that they have no competing interests.Publisher’s NoteSpringer Nature remains neutral with regard to jurisdictional claims inpublished maps and institutional affiliations.Author details1Department of Health Sciences, University of Florence, Anna MeyerChildren’s University Hospital, Viale Pieraccini, 24, I-50139 Florence, Italy.2Pediatric Infectious Diseases Unit, Anna Meyer Children’s University Hospital,Florence, Italy. 3Tuscany Regional Reference Centre for Mycobacteria,Microbiology and Virology Unit, Careggi Hospital, Florence, Italy.4Maxillo-Facial Surgery Unit, Neurosensorial Department, AziendaOspedaliero-Universitaria Careggi, Florence, Italy.Received: 9 August 2017 Accepted: 3 October 2017References1. Naselli A, Losurdo G, Avanzini S, Tarantino V, Cristina E, Bondi E, et al.Management of nontuberculous mycobacterial lymphadenitis in a tertiarycare children's hospital: a 20year experience. J Pediatr Surg. 2017;52:593–7.2. Tebruegge M, Pantazidou A, MacGregor D, Gonis G, Leslie D, Sedda L, et al.Nontuberculous mycobacterial disease in children - epidemiology, diagnosis& management at a tertiary center. PLoS One. 2016;11:e0147513.3. Tortoli E. Microbiological features and clinical relevance of new species ofthe genus mycobacterium. Clin Microbiol Rev. 2014;27:727–52.4. Zimmermann P, Tebruegge M, Curtis N, Ritz N. The management of non-tuberculous cervicofacial lymphadenitis in children: a systematic review andmeta-analysis. J Inf Secur. 2015;71:9–18.5. Ben Salah I, Cayrou C, Raoult D, Drancourt M. Mycobacterium marseillensesp. nov., mycobacterium timonense sp. nov. and mycobacteriumbouchedurhonense sp. nov., members of the Mycobacterium AviumComplex. Int J Syst Evol Microbiol. 2009;59:2803–8.6. Tortoli E, Pecorari M, Fabio G, Messino M, Fabio A, Commercial DNA. Probesfor mycobacteria incorrectly identify a number of less frequentlyencountered species. J Clin Microbiol. 2010;48:307–10.7. Candido PH, Nunes Lde S, Marques EA, Folescu TW, Coelho FS, de MouraVC, et al. Multidrug-resistant nontuberculous mycobacteria isolated fromcystic fibrosis patients. J Clin Microbiol. 2014;52:2990–7.8. Kim SY, Yoo H, Jeong BH, Jeon K, Ha YE, Huh HJ, et al. First case ofnontuberculous mycobacterial lung disease caused by mycobacteriummarseillense in a patient with systemic lupus erythematosus. DiagnMicrobiol Infect Dis. 2014;79:355–7.9. Grottola A, Roversi P, Fabio A, Antenora F, Apice M, Tagliazucchi S, et al.Pulmonary disease caused by mycobacterium marseillense, Italy. EmergInfect Dis. 2014;20:1769–70.10. Griffith DE, Aksamit T, Brown-Elliott BA, Catanzaro A, Daley C, Gordin F, et al.An official ATS/IDSA statement: diagnosis, treatment, and prevention ofnontuberculous mycobacterial diseases. Am J Respir Crit Care Med. 2007;175:367–416.•  We accept pre-submission inquiries •  Our selector tool helps you to find the most relevant journal•  We provide round the clock customer support •  Convenient online submission•  Thorough peer review•  Inclusion in PubMed and all major indexing services •  Maximum visibility for your researchSubmit your manuscript atwww.biomedcentral.com/submitSubmit your next manuscript to BioMed Central and we will help you at every step:Azzali et al. Italian Journal of Pediatrics  (2017) 43:92 Page 3 of 3

First case of Mycobacterium marseillense lymphadenitis in a child

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First case of Mycobacterium marseillense lymphadenitis in a child

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