Abstract
Tracheobronchial mucoepidermoid carcinomas (MEC) are rare in the pediatric population with literature limited primarily to case reports. Here we present our institutional experience treating MEC in three patients and review the literature of 142 pediatric cases previously published from 1968 to 2013. Although rare, tracheobronchial MEC should be included in the differential diagnosis in a child with recurrent respiratory symptoms. Conservative surgical management is often sufficient to achieve complete resection and good outcomes.
References
Dishop MK, Kuruvilla S (2008) Primary and metastatic lung tumors in the pediatric population: a review and 25-year experience at a large children’s hospital. Arch Pathol Lab Med 132:1079–1103
Stevenson GF, Hazard JB (1953) Mucoepidermoid carcinoma of salivary gland origin. Cleve Clin Q 20:445–456
Smetana HF, Iverson L, Swan LL (1952) Bronchogenic carcinoma; an analysis of 100 autopsy cases. Mil Surg 111:335–351
Liebow AA (1952) Tumors of the lower respiratory tract, in Atlas of Tumor Pathology, A.F.I.o. Pathology, Editor. Armed Forces Institute of Pathology: Washington, D.C. 26-53
Welsh JH, Maxson T, Jaksic T, Shahab I, Hicks J (1998) Tracheobronchial mucoepidermoid carcinoma in childhood and adolescence: case report and review of the literature. Int J Pediatr Otorhinolaryngol 45:265–273
Leschke H (1956) Regionally malignant and cancerously degenerated bronchial adenomas or carcinoids. Virchows Arch 328:635–657
Nunez VA, Spjut HJ, Rosenberg HS (1965) Bronchial adenomas in children. Tex Med 61:683–686
DeParedes CG, Pierce WS, Groff DB, Waldhausen JA (1970) Bronchogenic tumors in children. Arch Surg 100:574–576
DeBuse PJ, Lewis MJ, Shepherd JJ (1971) Muco-epidermoid tumour of the bronchus in a child. Br J Dis Chest 65:130–133
Miller RE, Hopeman AR (1975) Bronchial adenoma. Ann Thorac Surg 19:378–385
Wellons HA Jr, Eggleston P, Golden GT, Allen MS (1976) Bronchial adenoma in childhood. Two case reports and review of literature. Am J Dis Child 130:301–304
Bharani SN, Arbeit JM, Hyde JS, Dainauskas JR, Wilson RR (1976) Mucoepidermoid tumor of trachea. Chest 70:782–784
Conlan AA, Payne WS, Woolner LB, Sanderson DR (1978) Adenoid cystic carcinoma (cylindroma) and mucoepidermoid carcinoma of the bronchus. Factors affecting survival. J Thorac Cardiovasc Surg 76:369–377
Chavrier Y, Gounot J, Gounot R, Laydevant JP, Freycon F (1978) Muco epidermoid tumor of the bronchus in a 8 year old girl. Chir Pediatr 19:275–278
Leonardi HK, Jung-Legg Y, Legg MA, Neptune WB (1978) Tracheobronchial mucoepidermoid carcinoma. Clinicopathological features and results of treatment. J Thorac Cardiovasc Surg 76:431–438
Jaubert de Beaujeu M, Chavrier Y, Allantaz F (1979) Primary bronchopulmonary tumors in children. Apropos of 25 cases. Pediatrie 34:419–431
Mullins JD, Barnes RP (1979) Childhood bronchial mucoepidermoid tumors: a case report and review of the literature. Cancer 44:315–322
Nakagawara A, Ikeda K, Ohgami H (1979) Mucoepidermoid tumor of the bronchus in an infant. J Pediatr Surg 14:608–609
Breyer RH, Dainauskas JR, Jensik RJ, Faber LP (1980) Mucoepidermoid carcinoma of the trachea and bronchus: the case for conservative resection. Ann Thorac Surg 29:197–204
Hetta L, Olin C, Rutfors PE (1980) Tracheal adenoma in a 13-year-old boy with suspected asthma. Lakartidningen 77:3532–3534
Eskenasy A, Mangiulea V (1980) Ultrastructure of a bronchial mucoepidermoid tumour of a 7-year-old boy. Morphol Embryol 26:335–340
McDougall JC, Gorenstein A, Unni K, O’Connell EJ (1980) Carcinoid and mucoepidermoid carcinoma of bronchus in children. Ann Otol Rhinol Laryngol 89:425–427
Regadera JF, Rodríguez JI, Perera A, Mestre MJ (1982) Mucoepidermoid tumour in an 8-year-old boy. An Esp Pediatr 16:249–253
Lifschultz BD, Vanecko R, Hidvegi DF (1982) Three case reports. Mucoepidermoid tumors of the bronchus. IMJ Ill Med J 161:335–339
Binda R, Spreafico G, Sartori F, Calabrò F, Rampazzo F (1982) Mucoepidermoid carcinoma of the lung. Minerva Chir 37:517–521
Lack EE, Harris GB, Eraklis AJ, Vawter GF (1983) Primary bronchial tumors in childhood. A clinicopathologic study of six cases. Cancer 51:492–497
Seo IS, Warren J, Mirkin LD, Weisman SJ, Grosfeld JL (1984) Mucoepidermoid carcinoma of the bronchus in a 4-year-old child. A high-grade variant with lymph node metastasis. Cancer 53:1600–1604
Helin I, Tedgård U, Dejmek A, Lindgren S (1984) Muco-epidermoid tumour of the bronchus. Int J Pediatr Otorhinolaryngol 7:289–295
Leiberman A, Bar-Ziv J, Zirkin HJ (1986) Low grade mucoepidermoid tumour of the bronchus in childhood: a therapeutic dilemma. Eur J Pediatr 145:130–132
Archer RL, Grogg SE, Sanders SP (1987) Mucoepidermoid bronchial adenoma in a 6-year-old girl: a case report and review of the literature. J Thorac Cardiovasc Surg 94:452–454
Augustin N, Hofmann-v Kap-herr S, Wurnig P (1987) Endotracheal and endobronchial tumours in childhood. Prog Pediatr Surg 21:136–144
Yousem SA, Hochholzer L (1987) Mucoepidermoid tumors of the lung. Cancer 60:1346–1352
Torres AM, Ryckman FC (1988) Childhood tracheobronchial mucoepidermoid carcinoma: a case report and review of the literature. J Pediatr Surg 23:367–370
Ramírez-Mendoza P, Angeles-Angeles A, Grados-García C, Meneses-García A, Toledo-García A (1989) Bronchial mucoepidermoid carcinoma in a child. Bol Med Hosp Infant Mex 46:728–731
Heitmiller RF, Mathisen DJ, Ferry JA, Mark EJ, Grillo HC (1989) Mucoepidermoid lung tumors. Ann Thorac Surg 47:394–399
Allen ED, McCoy KS (1990) Presentation of bronchial mucoepidermoid carcinoma as unilateral hyperlucent lung. Pediatr Pulmonol 8:294–297
Watterson KG, Wisheart JD (1990) Tracheobronchial mucoepidermoid carcinoma in childhood with a 10 year follow-up. Eur J Cardiothorac Surg 4:112–113
Hofmann WJ, Hinz P, Roth H, Daum R (1990) Mucoepidermoid tumor of the lung in children. A case report and review of the literature. Pathologe 11:35–40
Su WJ, Lee YC, Perng RP (1990) Bronchial adenoma—a heterogenous group of tumors: report of 38 patients. Zhonghua Yi Xue Za Zhi (Taipei) 45:174–180
Inoue T, Maeda H, Ikeda M, Tani Y, Imachi T, Maeda M (1991) A case of bronchial mucoepidermoid carcinoma in a 8-year-old child treated with right upper sleeve lobectomy. Nihon Kyobu Geka Gakkai Zasshi 39:979–982
Hause DW, Harvey JC (1991) Endobronchial carcinoid and mucoepidermoid carcinoma in children. J Surg Oncol 46:270–272
Cohen MC, Kaschula RO (1992) Primary pulmonary tumors in childhood: a review of 31 years’ experience and the literature. Pediatr Pulmonol 14:222–232
Bellah RD, Mahboubi S, Berdon WE (1992) Malignant endobronchial lesions of adolescence. Pediatr Radiol 22:563–567
Hancock BJ, Di Lorenzo M, Youssef S, Yazbeck S, Marcotte JE, Collin PP (1993) Childhood primary pulmonary neoplasms. J Pediatr Surg 28:1133–1136
Gaissert HA, Mathisen DJ, Grillo HC, Vacanti JP, Wain JC (1994) Tracheobronchial sleeve resection in children and adolescents. J Pediatr Surg 29:192–197
Sakurada T, Kusajima K, Watanabe A, Inaoka M, Komatsu S (1995) A case of bronchial mucoepidermoid carcinoma in a 7-year-old girl. Kyobu Geka 48:585–588
Patel RG, Norman JR (1995) Unilateral hyperlucency with left lower lobe mass in a patient with bronchial asthma. Chest 107:569–570
Matsuzaki Y, Shibata K, Yoshioka M, Inoue M, Sekiya R, Onitsuka T, Koga Y, Tsuneyoshi A, Sumiyoshi A (1996) Successful treatment of bronchial mucoepidermoid carcinoma in an 11-year-old boy by bronchoplasty: report of a case. Surg Today 26:64–67
Deschildre A, Sardet A, Brouard J, Delaisi B, Boussard L, Boccon-Gibod L, Gosselin B, Tournier G, Leclerc F (1996) Bronchial mucoepidermoid carcinoma: apropos of three cases. Arch Pediatr 3:689–693
Tsuchiya H, Nagashima K, Ohashi S, Takase Y (1997) Childhood bronchial mucoepidermoid tumors. J Pediatr Surg 32:106–109
Granata C, Battistini E, Toma P, Balducci T, Mattioli G, Fregonese B, Gambini C, Rossi GA (1997) Mucoepidermoid carcinoma of the bronchus: a case report and review of the literature. Pediatr Pulmonol 23:226–232
Kim J, Park C, Kim K, Shim YM, Yang MK, Han J, Lee SI (1998) Surgical resection of mucoepidermoid carcinoma at the carina in a 9-year-old boy. J Pediatr Surg 33:1561–1562
Noda S, Sundaresan S, Mendeloff EN (1998) Tracheal mucoepidermoid carcinoma in a 7-year-old child. Ann Thorac Surg 66:928–929
Stenman G, Petursdottir V, Mellgren G, Mark J (1998) A child with a t(11;19)(q14-21;p12) in a pulmonary mucoepidermoid carcinoma. Virchows Arch 433:579–581
Takeda S, Hashimoto T, Kusu T, Kawamura T, Nojiri T, Funakoshi Y, Kadota Y, Maeda H (2007) Management and surgical resection for tracheobronchial tumors institutional experience with 12 patients. Interact Cardiovasc Thorac Surg 6:484–489
Desai DP, Mahoney EM, Miller RP, Holinger LD (1998) Mucoepidermoid carcinoma of the trachea in a child. Int J Pediatr Otorhinolaryngol 45:259–263
Anton-Pacheco J, Jimenez MA, Rodriguez-Peralto JL, Cuadros J, Berchi FJ (1998) Bronchial mucoepidermoid tumor in a 3-year-old child. Pediatr Surg Int 13:524–525
Bregante JI, Rituerto B, Font de Mora F, Alonso F, Andreu MJ, Figuerola J, Mulet JF (1998) Bronchial mucoepidermoid carcinoma. Cir Pediatr 11:129–131
Yildizeli B, Yüksel M, Ercan S, Karakoç F, Dagh E (1998) Childhood bronchial mucoepidermoid tumors. J Pediatr Surg 33:1586–1587
Kim TS, Lee KS, Han J, Im JG, Seo JB, Kim JS, Kim HY, Han SW (1999) Mucoepidermoid carcinoma of the tracheobronchial tree: radiographic and CT findings in 12 patients. Radiology 212:643–648
Segletes LA, Steffee CH, Geisinger KR (1999) Cytology of primary pulmonary mucoepidermoid and adenoid cystic carcinoma. A report of four cases. Acta Cytol 43:1091–1097
Dinopoulos A, Lagona E, Stinios I, Konstadinidou A, Kattamis C (2000) Mucoepidermoid carcinoma of the bronchus. Pediatr Hematol Oncol 17:401–408
Smiddy PF, Abdul Manaf ZM, Roberts IF, Gordon I (2000) Endobronchial mucoepidermoid carcinoma in childhood: case report and literature review. Clin Radiol 55:647–649
Andronikou S, Kader E (2001) Bronchial mucoepidermoid tumour in a child presenting with organomegaly due to secondary amyloidosis: case report and review of the literature. Pediatr Radiol 31:348–350
Scott KJ, Greinwald JH Jr, Darrow D, Smith RJ (2001) Endobronchial tumors in children: an uncommon clinical entity. Ann Otol Rhinol Laryngol 110:63–69
Niggemann B, Gerstner B, Guschmann M, Paul K, Wit J, Mau H, Wahn U (2002) An 11-year-old male with pneumonia and persistent airway obstruction. Eur Respir J 19:582–584
Kantar M, Cetingül N, Veral A, Kansoy S, Ozcan C, Alper H (2002) Rare tumors of the lung in children. Pediatr Hematol Oncol 19:421–428
Santambrogio L, Cioffi U, De Simone M, Rosso L, Ferrero S, Giunta A (2002) Video-assisted sleeve lobectomy for mucoepidermoid carcinoma of the left lower lobar bronchus: a case report. Chest 121:635–636
Barrett W, Heaps LS, Diaz S, Sharma P, Arbuckle S, Smith A (2002) Mucoepidermoid carcinoma of the bronchus in a 15-year-old girl with complex cytogenetic rearrangement involving 11q and over-expression of cyclin D1. Med Pediatr Oncol 39:49–51
Bashar MA, Sadeque AS, Bhuiyan MA, Uddin MN, Hossain MI (2003) Mucoepidermoid carcinoma of the lung–a case report. Bangladesh Med Res Counc Bull 29:125–129
Menon P, Singh M, Rao KL, Gulati M, Saxena AK, Gupta K, Kakkar N, Vasishta RK (2003) Mucoepidermoid carcinoma of the bronchus: is conservative surgery always justified? Indian J Cancer 40:34–36
Tomiyama K, Ishida H, Miyake M, Taki T, Koh S, Okuno T (2003) Surgically treated mucoepidermoid carcinoma in a 6-year-old boy; report of a case. Kyobu Geka 56:505–508
Al-Qahtani AR, Di Lorenzo M, Yazbeck S (2003) Endobronchial tumors in children: institutional experience and literature review. J Pediatr Surg 38:733–736
Giusti RJ, Flores RM (2004) Mucoepidermoid carcinoma of the bronchus presenting with a negative chest X-ray and normal pulmonary function in two teenagers: two case reports and review of the literature. Pediatr Pulmonol 37:81–84
Vaos G, Zavras N, Priftis K, Micahil-Strantzia C, Antypas G (2004) Bronchotomy in the treatment of a low-grade bronchial mucoepidermoid carcinoma in a child. J Thorac Cardiovasc Surg 128:782–783
Li CH, Huang SF, Li HY (2004) Bronchoscopic Nd-YAG laser surgery for tracheobronchial mucoepidermoid carcinoma—a report of two cases. Int J Clin Pract 58:979–982
Vogelberg C, Mohr B, Fitze G, Friedrich K, Hahn G, Roesner D, Leupold W, Suttorp M (2005) Mucoepidermoid carcinoma as an unusual cause for recurrent respiratory infections in a child. J Pediatr Hematol Oncol 27:162–165
Kinoshita H, Shimotake T, Furukawa T, Deguchi E, Iwai N (2005) Mucoepidermal carcinoma of the lung detected by positron emission tomography in a 5-year-old girl. J Pediatr Surg 40:E1–E3
Shilo K, Foss RD, Franks TJ, DePeralta-Venturina M, Travis WD (2005) Pulmonary mucoepidermoid carcinoma with prominent tumor-associated lymphoid proliferation. Am J Surg Pathol 29:407–411
Kut A, Karadag B, Karakoc F, Ersu R, Yildizeli B, Kotiloglu E, Yuksel M, Dagli E (2005) Mucoepidermoid carcinoma of the bronchus: a rare entity in childhood. Pediatr Int 47:203–205
Fauroux B, Aynie V, Larroquet M, Boccon-Gibod L, Ducou le Pointe H, Tamalet A, Clément A (2005) Carcinoid and mucoepidermoid bronchial tumours in children. Eur J Pediatr 164:748–752
Lal DR, Clark I, Shalkow J, Downey RJ, Shorter NA, Klimstra DS, La Quaglia MP (2005) Primary epithelial lung malignancies in the pediatric population. Pediatr Blood Cancer 45:683–686
Chan EY, MacCormick JA, Rubin S, Nizalik E (2005) Mucoepidermoid carcinoma of the trachea in a 4-year-old boy. J Otolaryngol 34:235–238
Lee EY, Vargas SO, Sawicki GS, Boyer D, Grant FD, Voss SD (2007) Mucoepidermoid carcinoma of bronchus in a pediatric patient: (18)F-FDG PET findings. Pediatr Radiol 37:1278–1282
Minami H, Sano I, Matsuo S, Oikawa M, Takagi K, Iwasaki K (2007) Mucoepidermoid carcinoma in a young man treated by bronchoplasty. Kyobu Geka 60:127–130
Ghraïri H, Kartas S, Ammar J, Abid H, Ayadi A, Kilani T, Hamzaoui A (2007) Prognosis of mucoepidermoid carcinoma of the bronchi. Rev Pneumol Clin 63:29–34
Sogut A, Yilmaz O, Yuksel H (2008) A rare cause of persistent atelectasis in childhood: mucoepidermoid carcinoma. Tuberk Toraks 56:325–328
Fujita K (2009) Mucoepidermoid carcinoma in a 13-year-old girl; report of a case. Kyobu Geka 62:423–426
Romão RL, de Barros F, Maksoud Filho JG, Gonçalves ME, Cardoso S, Tannuri AC, Tannuri U (2009) Malignant tumor of the trachea in children: diagnostic pitfalls and surgical management. J Pediatr Surg 44:e1–e4
Lei J (2010) Successful treatment of bronchial mucoepidermoid carcinoma in a longstanding collapsed lung. Ann Thorac Surg 90:655–657
Roby BB, Drehner D, Sidman JD (2011) Pediatric tracheal and endobronchial tumors: an institutional experience. Arch Otolaryngol Head Neck Surg 137:925–929
Wu M, Wang Q, Xu XF, Xiang JJ (2011) Bronchial mucoepidermoid carcinoma in children. Thorac Cardiovasc Surg 59:443–445
Yu Y, Song Z, Chen Z, Jian H, Lu S (2011) Chinese pediatric and adolescent primary tracheobronchial tumors: a hospital-based study. Pediatr Surg Int 27:721–726
Brassesco MS, Valera ET, Lira RC, Torres LA, Scrideli CA, Elias J Jr, Teixeira SR, Tone LG (2011) Mucoepidermoid carcinoma of the lung arising at the primary site of a bronchogenic cyst: clinical, cytogenetic, and molecular findings. Pediatr Blood Cancer 56:311–313
Firinci F, Ates O, Karaman O, Tekin A, Ozer E, Cakmakci H, Olgun N, Uzuner N (2011) A 7-year-old girl with cough, fever, pneumonia. Diagnosis: mucoepidermoid carcinoma (MEC). Pediatr Ann 40:124–127
Wildbrett P, Horras N, Lode H, Warzok R, Heidecke CD, Barthlen W (2012) Mucoepidermoid carcinoma of the lung in a 6-year-old boy. Afr J Paediatr Surg 9:159–162
Kanemoto A, Oshiro Y, Sugahara S, Kamagata S, Hirobe S, Toma M, Okumura T, Sakurai H (2012) Proton beam therapy for inoperable recurrence of bronchial high-grade mucoepidermoid carcinoma. Jpn J Clin Oncol 42:552–555
Seok Y, Lee E, Cho S (2012) Sleeve lobectomy of mucoepidermoid carcinoma in a 5-year-old girl. Ann Thorac Cardiovasc Surg 18:236–238
Papiashvilli M, Ater D, Mandelberg A, Sasson L (2012) Primary mucoepidermoid carcinoma of the trachea in a child. Interact Cardiovasc Thorac Surg 15:311–312
Wang H, Zhang N, Tao M, Li D, Zhou Y, Zou H, Liang S, Li J (2012) Application of interventional bronchoscopic therapy in eight pediatric patients with malignant airway tumors. Tumori 98:581–587
Sharma SV, Bell DW, Settleman J, Haber DA (2007) Epidermal growth factor receptor mutations in lung cancer. Nat Rev Cancer 7:169–181
Han SW, Kim HP, Jeon YK, Oh DY, Lee SH, Kim DW, Im SA, Chung DH, Heo DS, Bang YJ, Kim TY (2008) Mucoepidermoid carcinoma of lung: potential target of EGFR-directed treatment. Lung Cancer 61:30–34
Rossi G, Sartori G, Cavazza A, Tamberi S (2009) Mucoepidermoid carcinoma of the lung, response to EGFR inhibitors, EGFR and K-RAS mutations, and differential diagnosis. Lung Cancer 63:159–160
O’Neill ID (2009) Gefitinib as targeted therapy for mucoepidermoid carcinoma of the lung: possible significance of CRTC1-MAML2 oncogene. Lung Cancer 64:129–130
Yu Y, Song Z, Gao H, Zhu L, Lu S, Zhang J, Luo Q (2012) EGFR L861Q mutation is a frequent feature of pulmonary mucoepidermoid carcinoma. J Cancer Res Clin Oncol 138:1421–1425
Klacsmann PG, Olson JL, Eggleston JC (1979) Mucoepidermoid carcinoma of the bronchus: an electron microscopic study of the low grade and the high grade variants. Cancer 43:1720–1733
Chin CH, Huang CC, Lin MC, Chao TY, Liu SF (2008) Prognostic factors of tracheobronchial mucoepidermoid carcinoma—15 years experience. Respirology 13:275–280
Neville HL, Hogan AR, Zhuge Y, Perez EA, Cheung MC, Koniaris LG, Thompson WR, Sola JE (2009) Incidence and outcomes of malignant pediatric lung neoplasms. J Surg Res 156:224–230
Behboudi A, Enlund F, Winnes M, Andrén Y, Nordkvist A, Leivo I, Flaberg E, Szekely L, Mäkitie A, Grenman R, Mark J, Stenman G (2006) Molecular classification of mucoepidermoid carcinomas-prognostic significance of the MECT1-MAML2 fusion oncogene. Genes Chromosomes Cancer 45:470–481
Okabe M, Miyabe S, Nagatsuka H, Terada A, Hanai N, Yokoi M, Shimozato K, Eimoto T, Nakamura S, Nagai N, Hasegawa Y, Inagaki H (2006) MECT1-MAML2 fusion transcript defines a favorable subset of mucoepidermoid carcinoma. Clin Cancer Res 12:3902–3907
Serra A, Schackert HK, Mohr B, Weise A, Liehr T, Fitze G (2007) t(11;19)(q21;p12 ~ p13.11) and MECT1-MAML2 fusion transcript expression as a prognostic marker in infantile lung mucoepidermoid carcinoma. J Pediatr Surg 42:E23–E29
Seethala RR, Dacic S, Cieply K, Kelly LM, Nikiforova MN (2010) A reappraisal of the MECT1/MAML2 translocation in salivary mucoepidermoid carcinomas. Am J Surg Pathol 34:1106–1121
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Jaramillo, S., Rojas, Y., Slater, B.J. et al. Childhood and adolescent tracheobronchial mucoepidermoid carcinoma (MEC): a case-series and review of the literature. Pediatr Surg Int 32, 417–424 (2016). https://doi.org/10.1007/s00383-015-3849-y
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00383-015-3849-y