Abstract
Mycobacterium triplex was first described in 1996. This nontuberculous Mycobacterium causes a severe pulmonary disease in immunocompromised patients but it can involve also healthy patients. A literature search was made on the PubMed database and it produced only few cases of children with cervical lymphadenitis due to this Mycobacterium Triplex. We are describing a case of M. triplex cervical lymphadenitis in an immunocompetent child.
Keywords: Mycobacterium triplex, Cervical lymphadenitis, Immunocompetent child
Case report
A 4 years old boy was admitted to the Pediatric Surgery of University of Siena with cervical lymphadenopathy since about one month. Background showed a single episode of fever spontaneously regressed in about 48 h five days before the onset of enlargement of the cervical nodes. He underwent to antibiotic therapy with amoxicillin clavulanic acid and rifampicin for 15 days, without clinical remission. For this reason was reffered to our Clinic. A physical examination revealed a right swollen submandibular lymphnode (3 × 2 cm), feel rubbery, movable and painful. Overlying skin was healthy. A neck and abdomen ultrasound were performed. It was showed the presence of bilateral cervical lymphadenopathy with inflammatory aspect and no significant alterations in the abdomen. Blood exams were normal. There were no fever or others signs and symptoms as night sweats, unexplained weight loss, sore throat or difficulty in swallowing or breathing. Based on the clinical situation and according to parents a surgical excision of cervical lymph node was performed. The histopathology revealed necrotizing granulomatous lymphadenitis (Fig. 1). The cultural examination identified the presence of Mycobacterium’s specie. The growth and biochemical characteristics were most closely compatible with Mycobacterium triplex (M. triplex). The susceptibility to isoniazid and ethambutol was decreased by in vitro testing. This suspect was confirmed with molecular identification by PCR amplification and gene sequencing study of the 16S rRNA. Indeed, it showed that our isolate exhibited 100 % homology with the reference of M. triplex. The patient was discharged one day after surgery without any antimicrobial therapy. The clinical complete improvement was within 7 days. To date the patient is well without any health problems.
Fig. 1.
Histopathologic features
The incidence of infections caused by non tuberculous Mycobacterium (NTM) has increased in recent years [1]. They are ubiquitous organisms, commonly isolated from environmental and animal sources, whose pathogenicity may vary according to the host’s immune status. Although exposure to NTM frequently causes no symptoms [2]. M. triplex represents a unique species of Mycobacterium firstly described in 1996 by Floyd et al. [3]. The evidence is based on sequencing the 16S rRNA hypervariable region [4]. Currently the clinical relevance has not been systematically studied for M. triplex. A literature search was made on the PubMed database and it showed that it is more commonly associated with infections in immunocompromised adults patients. M. triplex has been rarely isolated from immunocompetent host and in children. Table 1 summarized the previous published cases of infection by M. Triplex with the main clinical features [5–12]. Our review of the Literature confirmed that the descriptions regarding lymphadenitis caused by this type of Mycobacterium in healthy children is very poor. To our knowledge, our patient is the second documented report of cervical lymphadenitis in a healthy child and the first in Italy. Also reviewing our series [13, 14], on 261 cases of cervical adenopathies we found no cases by M. triplex. The clinical picture of our child strongly suggested the diagnosis of nontubercolosus mycobacterial lymphadenitis and the clinical features were similar to another reported case [11]. According to the Literature we believe that the surgical excision represents the gold standard therapy in nontubercolous Mycobacterial lymphadenitis. Our documented case may suggest to clinicians and medical microbiologists to keep in mind the M. triplex in the etiological differential diagnosis of cervical lymphadenitis in otherwise healthy children. In any case, the differential diagnosis should be always considered thought this diagnostic improvement has only a taxonomic and epidemiological value: the clinical features and the treatment are the same as all other adenopathies caused by nontuberculous Mycobacteria. We also believe that important questions regarding epidemiology and pathogenesis of the disease caused by these organism remain today unanswered. It is possible that the few reported cases can be due to a failure diagnosis for difficult isolation and specificity in cultural examination.
Table 1.
Published cases of human infection with M. triplex and related site involved
| Site | Patient (age and sex) and references | Patient’s immunocompetency | Clinical/instrumental manifestations | Diagnostic methods | Treatment | Outcome |
|---|---|---|---|---|---|---|
| Lung | 54 year-old female (10) | Yes | Cought, fatigue CT: lung nodule, cavitations and bronchiectases | Culture of broncoalveolar lavage and bronchial aspirate genetic diagnosis | Chemotherapy RMP-EMB-INH; EMB-CLA; EMB-CLA-LVX; | A&W |
| 54 year-old female (9) | Yes | Hemoptysis, cought, fever, fatigue CT: lung infiltrate and nodule (0.3 cm) | Culture of broncoalveolar lavage genetic-diagnosis | Chemotherapy RMP-CLA-INH | A&W | |
| 67 year-old male (8) | Yes | Hemoptysis CT: bronchiectases, alveolar opacities, and micronodules. | Culture of bronchial aspirate and sputum genetic-diagnosis | Chemotherapy RMP-CLA-CIP-EMB | A&W | |
| Neck | 4 year-old female (11) | Yes | Preauricolar mass, submandibular adenopathy | Culture of lymph-node biopsy speciment genetic-diagnosis | Chemotherapy RMP-EMB-CLA; RFB-EMB-CLA; Surgical incision. | A&W |
| 4 year-old male (our case) | Yes | Submandibular lymph-node (3 × 2 cm) feel rubbery, movable and painful. no alterations of overlying skin. afebrile. | Culture of two pem lymph-node genetic-diagnosis | Surgical excision | A&W | |
| Brain | 41 year-old male (7) | Hiv-infection | Fever, cachexia, several edema of the lower limbs, diarrhea, ascite, cought. | Culture of sputum, ascitic fluid genetic diagnosis | Chemotherapy RMP-EMB-INH-CLA-PZA + Antiretroviral therapy (RITONAVIR, INDINAVIR ZIDOVUDINE AND LAMIVUDINE) | Death |
| Other | ||||||
| Disseminated disease | 40 year-old male (5) | Hiv-infection | Fever, night sweats, chills, weight loss and articular pain. MRI/CT: colliquative abscess of the left knee; spleen abscess, multiple lymphadenopathy. | Culture of join fluid, bone and blood genetic diagnosis | Chemotherapy CLA-ETIO Antiretroviral therapy was modified to include quadruple combination of 2 new nucleoside analogues (didanosine and stavudine) and 2 protease inhibitors (ritonavir and saquinavir hard-gel capsules). Surgical drainage | Condition is slowly worsening |
| Pericardial and peritoneal fluid | 13 year-old female (6) | Drug-inducted immunodepression | Ascitis, pericarditis | Culture of pericardial and peritoneal fluid GENETIC DIAGNOSIS | Drainage procedure (paracentesis and pericardiocentesis) | A&W |
| Lymph-nodes | 47 year-old male (12) | Hiv-infection | Axillary adenopathy | Culture of lymph-node biopsy speciment | Chemotherapy INH-RFP-ETB (3mo) and INH-RFP (2mo) | Death |
RMP rifampicin, CLA clarithromycin, CIP ciprofloxacin, EMB ethambutol, INH isoniazid, LVX levofloxacin, ETIO ethionamide, PZA pyrazinamide, RFB rifambutin
Contributor Information
R. Angotti, Phone: +39 0577 586502, FAX: +39 0577 586174, Email: rossellaangotti@libero.it
F. Molinaro, Email: fmolidoc@me.com
E. Benicchi, Email: dr.gcaruso2002@virgilio.it
E. Cerchia, Email: elicerc@virgilio.it
M. Messina, Email: mario.messina@unisi.it
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