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Volume 31, Number 3—March 2025
Research

A 28-Year Multicenter Cohort Study of Nontuberculous Mycobacterial Lymphadenitis in Children, Spain

Aina Martínez-Planas, Fernando Baquero-Artigao, Ana Méndez-Echevarría, Teresa Del Rosal, Paula Rodríguez-Molino, Carlos Toro-Rueda, Matilde Bustillo-Alonso, Miguel Lafuente, Anna Canet, Ángela Manzanares, Alfredo Tagarro, Francisco José Sanz-Santaeufemia, Sara Guillén-Martín, María José Cilleruelo, Lola Falcón-Neyra, Begoña Santiago, Elena Rincón, Miguel Lillo, Antoni Soriano-Arandes, Luigi Sedda, Clàudia Fortuny, Manuel Monsonís, Julián González-Martín, Marc Tebruegge1, Antoni Noguera-Julian1Comments to Author , and Spanish Pediatric Tb Research Network and The European Nontuberculous Mycobacterial Lymphadenitis In Children Study2
Author affiliation: Institut de Recerca Pediàtrica Sant Joan de Déu, Barcelona, Spain (A. Martínez-Planas, C. Fortuny, M. Monsonís, A. Noguera-Julian); La Paz Research Institute, Madrid, Spain (F. Baquero-Artigao, A. Méndez-Echevarría, T. Del Rosal, P. Rodríguez-Molino); Universidad Autónoma de Madrid, Madrid (F. Baquero-Artigao, A. Méndez-Echevarría, T. Del Rosal, P. Rodríguez-Molino); Hospital La Paz, Madrid (F. Baquero-Artigao, A. Méndez-Echevarría, T. Del Rosal, P. Rodríguez-Molino, C. Toro-Rueda); Centro de Investigación Biomédica en Red en Enfermedades Infecciosas, Madrid (F. Baquero-Artigao, A. Méndez-Echevarría, P. Rodríguez-Molino, B. Santiago, J. González-Martín); Centro de Investigación Biomédica en Red en Enfermedades Raras, Madrid (T. Del Rosal); Hospital Universitario Miguel Servet, Zaragoza, Spain (M. Bustillo-Alonso, M. Lafuente); Hospital Universitario 12 de Octubre, Madrid, Spain (A. Canet, A. Manzanares); Instituto de Investigación 12 de Octubre, Madrid (A. Tagarro); Hospital Universitario Infanta Sofía, Madrid (A. Tagarro); Universidad Europea de Madrid, Madrid (A. Tagarro); Hospital Universitario Niño Jesús, Madrid (F.J. Sanz-Santaeufemia); Hospital Universitario de Getafe, Getafe, Spain (S. Guillén-Martín); Hospital Universitario Puerta de Hierro, Majadahonda, Spain (M.J. Cilleruelo); Hospital Virgen del Rocío, Seville, Spain (L. Falcón-Neyra); Instituto de Investigación Sanitaria Gregorio Marañón, Madrid (B. Santiago); Hospital Gregorio Marañón, Madrid (B. Santiago, E. Rincón); Hospital General Universitario de Albacete, Albacete, Spain (M. Lillo); Hospital Vall d'Hebron, Barcelona, Spain (A. Soriano-Arandes); Lancaster University, Lancaster, UK (L. Sedda); Centro de Investigación Biomédica en Red en Epidemiología y Salud Pública, Madrid (C. Fortuny, A. Noguera-Julian); Universitat de Barcelona, Barcelona, Spain (C. Fortuny, A. Noguera-Julian); Hospital Clínic, Barcelona (J. González-Martín); Austrian Reference Centre for Childhood Tuberculosis, Vienna, Austria (M. Tebruegge); Vienna Healthcare Group, Vienna (M. Tebruegge); Royal Children’s Hospital Melbourne, University of Melbourne, Melbourne, Victoria, Australia (M. Tebruegge); UCL Great Ormond Street Institute of Child Health, London, UK (M. Tebruegge).

Main Article

Table 1

Baseline characteristics, clinical manifestations, treatment, outcomes, and comparisons of MAC and M. lentiflavum lymphadenitis cases from a 28-year multicenter cohort study of NTM lymphadenitis in children, Spain*

Characteristics All, n = 311 MAC, n = 134 M. lentiflavum, n = 124 p value†
Sex
F 167 (53.7) 77 (57.5) 67 (54.0) 0.579
M
 144 (46.3)
 57 (42.5)
 57 (46.0)
Age, y (IQR)
 2.4 (1.7–3.2)
 2.7 (2.0–3.8)
 1.9 (1.6–2.6)
 <0.001
Patients <5 y of age at diagnosis
 26 (8.4)
 14 (10.4)
 4 (3.2)
 0.042
Prospective phase
 196 (63.0)
 78 (58.2)
 88 (71.0)
 0.041
Season at symptom onset‡ 0.338
Spring 102 (38.1) 39 (33.6) 42 (38.5)
Summer 58 (21.6) 22 (19.0) 28 (25.7)
Autumn 46 (17.2) 23 (19.8) 17 (15.6)
Winter
 62 (23.1)
 32 (27.6)
 22 (20.2)
Reported in central Spanish regions
 189 (60.8)
 52 (38.8)
 107 (86.3)
 <0.001
TB infection risk factors 11 (3.5) 4 (3.0) 2 (1.6) 0.685
Positive TST 168/278 (60.4) 76/118 (64.4) 62/111 (55.9) 0.186
TST induration, mm (IQR)
 7.0 (0–10)
 7.5 (4–11)
 7.0 (0–10)
 0.055
NTM lymphadenitis disease characteristics
Unilateral disease 282 (90.7) 124 (92.5) 111 (89.5) 0.395
Single site disease 242 (77.8) 106 (79.1) 90 (72.6) 0.220
Symptom duration, wks (IQR) 4.0 (2.0–6.0) 4.0 (2.0–7.3) 3.0 (2.0–4.0) 0.001
Maximum lymph node diameter, cm (IQR)
 3.0 (2.1–4.0)
 3.0 (2.0–4.0)
 3.0 (2.1–4.0)
 0.558
Clinical stage§ 0.926
Stage I 148 (48.6) 62 (47.3) 62 (50.8)
Stage II 27 (8.8) 13 (9.9) 13 (10.7)
Stage III 105 (34.4) 44 (33.6) 37 (30.3)
Stage IV
 25 (8.2)
 12 (9.2)
 10 (8.2)
Affected site
Submandibular 197 (63.3) 74 (55.2) 91 (73.4) 0.002
Superficial/deep cervical 81 (26.0) 39 (29.1) 28 (22.6) 0.247
Preauricular 44 (14.1) 13 (9.7) 25 (20.2) 0.019
Parotid 28 (9.0) 10 (7.5) 12 (9.7) 0.523
Jugulodigastric 22 (7.1) 15 (11.2) 6 (4.8) 0.062
Other¶
 22 (7.1)
 15 (11.2)
 3 (2.4)
 0.119
Treatment and outcomes
Initial treatment strategy 0.006
Observation alone 25 (8.0) 15 (11.2) 5 (4.0)
Antimicrobial drugs alone 76 (24.5) 22 (16.4) 35 (28.2)
Drainage alone 6 (1.9) 2 (1.5) 2 (1.6)
Drainage + antimicrobial drugs 29 (9.3) 14 (10.5) 10 (8.1)
Complete resection alone 80 (25.7) 46 (34.3) 25 (20.2)
Complete resection + antimicrobial drugs 88 (28.3) 32 (23.9) 43 (34.7)
Not reported 7 (2.3) 3 (2.2) 4 (3.2)
Lost to follow-up 23 (7.4) 12 (9.0) 9 (7.3) 0.618
Recurrent NTM lymphadenitis 16 (5.6) 10 (8.2) 6 (5.2) 0.257
New fistulization#
 36 (12.9)
 20 (18.2)
 9 (8.6)
 0.042
Sequelae
None 188 (65.3) 78 (63.4) 76 (66.7) 0.600
Hypertrophic scar 52 (18.1) 25 (20.5) 15 (13.0) 0.126
Skin discoloration 27 (9.4) 13 (10.7) 12 (10.4) 0.956
Transient facial palsy 23 (8.0) 11 (9.0) 10 (8.7) 0.931
Permanent facial palsy 7 (2.4) 1 (0.8) 5 (4.3) 0.093
Frey syndrome 1 (0.3) 0 (0) 1 (0.9) 0.986

*Vaues are no. (5) except as indicated. Groups were compared with Student t tests or Mann-Whitney U tests for continuous variables and χ2 tests for categorical variables. MAC, Mycobacterium avium complex; NTM, nontuberculous mycobacteria; TB, tuberculosis; TST, tuberculin skin test. †Comparison between MAC and M. lentiflavum cases. ‡Available in 268 cases in the whole cohort (116 MAC and 109 M. lentiflavum cases). §Available in 305 cases in the whole cohort (131 MAC and 122 M. lentiflavum cases). ¶Includes posterior cervical (n = 6), occipital (n = 5), postauricular (n = 4), inguinal (n = 3), axillar (n = 2), and supraclavicular (n = 2). #Excludes patients with clinical stage IV (fistulization) at initial examination.

Main Article

1These senior authors contributed equally to this article.

2Members of the European Nontuberculous Mycobacterial Lymphadenitis in Children Study are listed at the end of this article.

Page created: February 04, 2025
Page updated: February 21, 2025
Page reviewed: February 21, 2025
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