Background: Mucoepidermoid carcinoma (MEC) is the most common malignant salivary gland tumour in paediatric population, accounting for 16% of all cases. Patients affected by a previous solid or leukaemic neoplasm during their childhood may develop a second different tumour during the follow-up. In this setting, salivary gland MEC is relatively frequent, accounting for 6% of the second neoplasms in paediatric patients. Consequently, the occurrence of salivary gland nodules in paediatric patients with a previous neoplasm should be considered an event with a high risk of malignancy that poses peculiar diagnostic challenges. Summary: This study was designed to define clinical and instrumental findings and morphological features of MEC on fine-needle aspiration cytology (FNAC) samples in paediatric patients with and without a previous neoplasm. Five patients under 19 years are included in this series. FNAC was performed in all patients on a parotid nodule. We have identified 2 groups of patients: (a) 2 cases with previous history of malignancy (acute lymphoblastic leukaemia and Hodgkin lymphoma) and (b) 3 cases without previous malignant neoplasms. In all cases, a final diagnosis of MEC was rendered. Key Messages: MEC may occur as a second malignancy in paediatric patients. FNAC is certainly a valid and accurate diagnostic tool for this type of neoplasm, even in the paediatric age, allowing the correct management of the patients.

1.
Védrine
PO
,
Coffinet
L
,
Temam
S
,
Montagne
K
,
Lapeyre
M
,
Oberlin
O
, et al
Mucoepidermoid carcinoma of salivary glands in the pediatric age group: 18 clinical cases, including 11 second malignant neoplasms
.
Head Neck
.
2006 Sep
;
28
(
9
):
827
33
.
2.
Ronchi
A
,
Montella
M
,
Zito Marino
F
,
Panarese
I
,
Pagliuca
F
,
Colella
G
, et al
Diagnostic accuracy of FNA cytology for diagnosis of salivary gland tumors in pediatric patients
.
Cancer Cytopathol
.
2019 Aug
;
127
(
8
):
529
38
.
3.
Da Cruz Perez
DE
,
Pires
FR
,
Alves
FA
,
Almeida
OP
,
Kowalski
LP
.
Salivary gland tumors in children and adolescents: a clinicopathologic and immunohistochemical study of fifty-three cases
.
Int J Pediatr Otorhinolaryngol
.
2004
;
68
(
7
):
895
902
.
4.
Muenscher
A
,
Diegel
T
,
Jaehne
M
,
Ussmüller
J
,
Koops
S
,
Sanchez-Hanke
M
.
Benign and malignant salivary gland diseases in children A retrospective study of 549 cases from the Salivary Gland Registry, Hamburg
.
Auris Nasus Larynx
.
2009
;
36
(
3
):
326
31
.
5.
Guzzo
M
,
Ferrari
A
,
Marcon
I
,
Collini
P
,
Gandola
L
,
Pizzi
N
, et al
Salivary gland neoplasms in children: the experience of the Istituto Nazionale Tumori of Milan
.
Pediatr Blood Cancer
.
2006 Nov
;
47
(
6
):
806
10
.
6.
Ethunandan
M
,
Ethunandan
A
,
Macpherson
D
,
Conroy
B
,
Pratt
C
.
Parotid neoplasms in children: experience of diagnosis and management in a district general hospital
.
Int J Oral Maxillofac Surg
.
2003
;
32
(
4
):
373
7
.
7.
Mamlouk
MD
,
Rosbe
KW
,
Glastonbury
CM
.
Paediatric parotid neoplasms: a 10 year retrospective imaging and pathology review of these rare tumours
.
Clin Radiol
.
2015 Mar
;
70
(
3
):
270
7
.
8.
Gontarz
M
,
Wyszyńska-Pawelec
G
,
Zapała
J
.
Primary epithelial salivary gland tumours in children and adolescents
.
Int J Oral Maxillofac Surg
.
2018 Jan
;
47
(
1
):
11
5
.
9.
Fang
QG
,
Shi
S
,
Li
ZN
,
Zhang
X
,
Liu
FY
,
Sun
CF
.
Epithelial salivary gland tumors in children: a twenty-five-year experience of 122 patients
.
Int J Pediatr Otorhinolaryngol
.
2013 Aug
;
77
(
8
):
1252
4
.
10.
Allan
BJ
,
Tashiro
J
,
Diaz
S
,
Edens
J
,
Younis
R
,
Thaller
SR
.
Malignant tumors of the parotid gland in children: incidence and outcomes
.
J Craniofac Surg
.
2013 Sep
;
24
(
5
):
1660
4
.
11.
Aro
K
,
Leivo
I
,
Grénman
R
,
Mäkitie
AA
.
Paediatric salivary gland cancer in Finland
.
Int J Pediatr Otorhinolaryngol
.
2012 Sep
;
76
(
9
):
1304
7
.
12.
Sultan
I
,
Rodriguez-Galindo
C
,
Al-Sharabati
S
,
Guzzo
M
,
Casanova
M
,
Ferrari
A
.
Salivary gland carcinomas in children and adolescents: a population-based study, with comparison to adult cases
.
Head Neck
.
2011 Oct
;
33
(
10
):
1476
81
.
13.
Laikui
L
,
Hongwei
L
,
Hongbing
J
,
Zhixiu
H
.
Epithelial salivary gland tumors of children and adolescents in west China population: a clinicopathologic study of 79 cases
.
J Oral Pathol Med
.
2008 Apr
;
37
(
4
):
201
5
.
14.
Ellies
M
,
Schaffranietz
F
,
Arglebe
C
,
Laskawi
R
.
Tumors of the salivary glands in childhood and adolescence
.
J Oral Maxillofac Surg
.
2006 Jul
;
64
(
7
):
1049
58
.
15.
Yu
GY
,
Li
ZL
,
Ma
DQ
,
Zhang
Y
.
Diagnosis and treatment of epithelial salivary gland tumours in children and adolescents
.
Br J Oral Maxillofac Surg
.
2002 Oct
;
40
(
5
):
389
92
.
16.
Ribeiro
KdC
,
Kowalski
LP
,
Saba
LM
,
de Camargo
B
.
Epithelial salivary glands neoplasms in children and adolescents: a forty-four-year experience
.
Med Pediatr Oncol
.
2002 Dec
;
39
(
6
):
594
600
.
17.
Bentz
BG
,
Hughes
CA
,
Lüdemann
JP
,
Maddalozzo
J
.
Masses of the salivary gland region in children
.
Arch Otolaryngol Head Neck Surg
.
2000 Dec
;
126
(
12
):
1435
9
.
18.
Hicks
J
,
Flaitz
C
.
Mucoepidermoid carcinoma of salivary glands in children and adolescents: assessment of proliferation markers
.
Oral Oncol
.
2000 Sep
;
36
(
5
):
454
60
.
19.
Kaste
SC
,
Hedlund
G
,
Pratt
CB
.
Malignant parotid tumors in patients previously treated for childhood cancer: clinical and imaging findings in eight cases
.
AJR Am J Roentgenol
.
1994
;
162
(
3
):
655
9
.
20.
Levitt
SH
,
McHugh
RB
,
Gómez-Marin
O
,
Hyams
VJ
,
Soule
EH
,
Strong
EW
, et al
Clinical staging system for cancer of the salivary gland: a retrospective study
.
Cancer
.
1981 Jun 1
;
47
(
11
):
2712
24
.
21.
Techavichit
P
,
Hicks
MJ
,
López-Terrada
DH
,
Quintanilla
NM
,
Guillerman
RP
,
Sarabia
SF
, et al
Mucoepidermoid carcinoma in children: a single institutional experience
.
Pediatr Blood Cancer
.
2016 Jan
;
63
(
1
):
27
31
.
22.
Vitagliano
G
,
Santoro
G
,
Landolfi
L
,
Cozzolino
I
,
Peluso
AL
,
Ieni
A
, et al
Fine-needle cytology of intraglandular parotid lymph node: a useful procedure in the management of salivary gland nodules
.
Diagn Cytopathol
.
2019 Jul
;
47
(
7
):
695
700
.
23.
Stewart
CJ
,
MacKenzie
K
,
McGarry
GW
,
Mowat
A
.
Fine-needle aspiration cytology of salivary gland: a review of 341 cases
.
Diagn Cytopathol
.
2000 Mar
;
22
(
3
):
139
46
.
24.
Rammeh
S
,
Romdhane
E
,
Sassi
A
,
Belhajkacem
L
,
Blel
A
,
Ksentini
M
, et al
Accuracy of fine-needle aspiration cytology of head and neck masses
.
Diagn Cytopathol
.
2019 May
;
47
(
5
):
394
9
.
25.
Zajicek
J
,
Eneroth
CM
,
Jakobsson
P
.
Aspiration biopsy of salivary gland tumors. VI. Morphologic studies on smears and histologic sections from mucoepidermoid carcinoma
.
Acta Cytol
.
1976
;
20
(
1
):
35
41
.
26.
Klijanienko
J
,
Vielh
P
.
Fine-needle sampling of salivary gland lesions. IV. Review of 50 cases of mucoepidermoid carcinoma with histologic correlation
.
Diagn Cytopathol
.
1997 Aug
;
17
(
2
):
92
8
.
27.
Dombrowski
ND
,
Wolter
NE
,
Irace
AL
,
Cunningham
MJ
,
Mack
JW
,
Marcus
KJ
, et al
Mucoepidermoid carcinoma of the head and neck in children
.
Int J Pediatr Otorhinolaryngol
.
2019 May
;
120
:
93
9
.
28.
Mavec
P
,
Eneroth
CM
,
Franzen
S
,
Moberger
G
,
Zajicek
J
.
Aspiration biopsy of salivary gland tumours. I. Correlation of cytologic reports from 652 aspiration biopsies with clinical and histologic findings
.
Acta Otolaryngol
.
1964
;
58
:
471
84
.
29.
Persson
PS
,
Zettergren
L
.
Cytologic diagnosis of salivary gland tumors by aspiration biopsy
.
Acta Cytol
.
1973
;
17
(
4
):
351
4
.
30.
Cohen
MB
,
Fisher
PE
,
Holly
EA
,
Ljung
BM
,
Löwhagen
T
,
Bottles
K
.
Fine needle aspiration biopsy diagnosis of mucoepidermoid carcinoma. Statistical analysis
.
Acta Cytol
.
1990
;
34
(
1
):
43
9
.
31.
Faquin
W
,
Rossi
E
,
Baloch
Z
.
The milan system for reporting salivary gland cytopathology
.
Cham, Switzerland
:
Springer International Publishing
;
2018
.
32.
Boukheris
H
,
Curtis
RE
,
Land
CE
,
Dores
GM
.
Incidence of carcinoma of the major salivary glands according to the WHO classification, 1992 to 2006: a population-based study in the United States
.
Cancer Epidemiol Biomarkers Prev
.
2009 Nov
;
18
(
11
):
2899
906
.
33.
Auclair
PL
,
Goode
RK
,
Ellis
GL
.
Mucoepidermoid carcinoma of intraoral salivary glands. Evaluation and application of grading criteria in 143 cases
.
Cancer
.
1991
;
69
(
8
):
2021
30
.
34.
Spiro
RH
,
Huvos
AG
,
Berk
R
,
Strong
EW
.
Mucoepidermoid carcinoma of salivary gland origin. A clinicopathologic study of 367 cases
.
Am J Surg
.
1978
;
136
(
4
):
461
8
.
35.
Evenson
JW
,
Cawson
RA
.
Salivary gland tumours: a review of 2410 cases with particular reference to histological types, site, age and sex distribution
.
J Pathol
.
1985
;
146
:
51
8
.
36.
Jakobsson
PA
,
Blanck
C
,
Eneroth
CM
.
Mucoepidermoid carcinoma of the parotid gland
.
Cancer
.
1968
;
22
(
1
):
111
24
.
37.
Micheau
C
,
Lacour
J
,
Génin
J
,
Brugère
J
.
Tumeurs mucoepidermoides de la parotide et de la cavité buccale. A propos de 33 observations. Etude anatomo-clinique et histochimique
.
Ann Anat Pathol
.
1972
;
17
:
59
71
.
38.
Sood
S
,
McGurk
M
,
Vaz
F
.
Management of salivary gland tumours: United Kingdom National Multidisciplinary Guidelines
.
J Laryngol Otol
.
2016 May
;
130
(
S2
):
S142
S49
.
39.
Garden
AS
,
Weber
RS
,
Morrison
WH
,
Ang
KK
,
Peters
LJ
.
The influence of positive margins and nerve invasion in adenoid cystic carcinoma of the head and neck treated with surgery and radiation
.
Int J Radiat Oncol Biol Phys
.
1995
;
32
(
3
):
619
26
.
40.
Rauso
R
,
Colella
G
,
Franco
R
,
Ronchi
A
,
Chirico
F
.
Ossified Carcinoma Ex Pleomorphic Adenoma in accessory lobe of parotid gland: complexity in clinical, imaging and histologic diagnosis and minimally invasive surgery
.
Oral Oncol
.
2019 Mar 7
;
92
:
95
8
.
41.
Lindberg
LG
,
Åkerman
M
.
Aspiration cytology of salivary gland tumors: diagnostic experience from six years of routine laboratory work
.
Laryngoscope
.
1976
;
86
(
4
):
584
94
.
42.
Bhanuprasad
V
,
Mallick
S
,
Bhasker
S
,
Mohanti
BK
.
Pediatric head and neck squamous cell carcinoma: Report of 12 cases and illustrated review of literature
.
Int J Pediatr Otorhinolaryngol
.
2015 Aug
;
79
(
8
):
1279
82
.
43.
Atahan
IL
,
Ayhan
A
,
Ozyar
E
,
Ertoy
D
,
Gürkaynak
M
.
A case of mucoepidermoid carcinoma of the parotid gland developing in a child after the treatment of acute lymphoblastic leukemia
.
Pediatr Hematol Oncol
.
1995
;
12
(
4
):
403
5
.
44.
Boccon-Gibod
L
,
Boman
F
,
Josset
P
,
Landman-Parker
J
.
Mucoepidermoid carcinoma of the parotid gland in a child previously treated for acute lymphoblastic leukemia
.
Pediatr Blood Cancer
.
2005 Jun 15
;
44
(
7
):
673
5
.
45.
Prasannan
L
,
Pu
A
,
Hoff
P
,
Weatherly
R
,
Castle
V
.
Parotid carcinoma as a second malignancy after treatment of childhood acute lymphoblastic leukemia
.
J Pediatr Hematol Oncol
.
1999
;
21
(
6
):
535
8
.
46.
Savelli
SL
,
Klopfenstein
KJ
,
Termuhlen
AM
.
Mucoepidermoid carcinoma of the parotid gland as a second malignant neoplasm
.
Pediatr Blood Cancer
.
2005Dec
;
45
(
7
):
997
1000
.
47.
Berridge
DC
,
Hitchcock
A
,
Bradley
PJ
,
Kapila
L
.
Second primary tumour following retinoblastoma
.
J Laryngol Otol
.
1989
;
103
(
7
):
690
1
.
48.
Zappia
JJ
,
McClatchey
KD
.
Mucoepidermoid carcinoma associated with acute lymphoblastic leukemia
.
Int J Pediatr Otorhinolaryngol
.
1992
;
23
(
1
):
85
9
.
49.
Tikku
G
,
Jain
D
,
Kumari
A
,
Grover
R
.
Mucoepidermoid carcinoma of parotid as a second malignancy in acute lymphoblastic leukemia
.
Indian Pediatr
.
2015Nov
;
52
(
11
):
979
80
.
50.
Loy
TS
,
McLaughlin
R
,
Odom
LF
,
Dehner
LP
.
Mucoepidermoid carcinoma of the parotid as a second malignant neoplasm in children
.
Cancer
.
1989
;
64
(
10
):
2174
7
.
51.
Rodríguez-Cuevas
S
,
Ocampo
LB
.
A case report of mucoepidermoid carcinoma of the parotid gland developing after radioiodine therapy for thyroid carcinoma
.
Eur J Surg Oncol
.
1995
;
21
(
6
):
692
.
52.
Sandoval
C
,
Jayabose
S
.
Parotid mucoepidermoid carcinoma following chemotherapy for childhood acute lymphoblastic leukemia
.
Pediatr Hematol Oncol
.
2001
;
18
(
3
):
217
20
.
53.
Walker
MJ
,
Chaudhuri
PK
,
Wood
DC
,
Das Gupta
TK
.
Radiation-induced parotid cancer
.
Arch Surg
.
1981
;
116
(
3
):
329
31
.
54.
Leung
W
,
Ribeiro
RC
,
Hudson
M
,
Tong
X
,
Srivastava
DK
,
Rubnitz
JE
, et al
Second malignancy after treatment of childhood acute myeloid leukemia
.
Leukemia
.
2001 Jan
;
15
(
1
):
41
5
.
55.
Zidar
N
,
Gale
N
,
Zupevc
A
,
Zargi
M
.
Salivary gland tumours as second neoplasms: two cases and literature review
.
J Laryngol Otol
.
2010May
;
124
(
5
):
577
80
.
56.
Myer
CM
 III
.
Second primary malignancies of the head and neck in children
.
Am J Otolaryngol
.
1995
;
16
(
6
):
415
7
.
57.
Piccinelli
KJ
,
Taj
M
,
Lucraft
HH
,
Skinner
R
.
Secondary parotid mucoepidermoid carcinoma after TBI and chemotherapy in childhood AML
.
Pediatr Blood Cancer
.
2006Sep
;
47
(
3
):
345
6
.
58.
Blatt
J
,
Zdanski
C
,
Scanga
L
,
Rao
KW
,
Morris
DE
,
Shockley
WW
.
Mucoepidermoid carcinoma of the parotid as a secondary malignancy after chemotherapy in a child with neuroblastoma
.
J Pediatr Hematol Oncol
.
2013 Jul
;
35
(
5
):
399
401
.
59.
Cheuk
DK
,
Shek
TW
,
Chan
GC
,
Lau
YL
,
Ha
SY
,
Chiang
AK
.
Parotid acinar cell carcinoma in a long-term survivor of childhood acute lymphoblastic leukemia
.
Pediatr Blood Cancer
.
2008 Mar
;
50
(
3
):
636
9
.
60.
Tugcu
D
,
Akici
F
,
Aydogan
G
,
Salcioglu
Z
,
Akcay
A
,
Sen
H
, et al
Mucoepidermoid carcinoma of the parotid gland in childhood survivor of acute lymphoblastic leukemia with need of radiotherapy for treatment and review of the literature
.
Pediatr Hematol Oncol
.
2012 May
;
29
(
4
):
380
5
.
61.
Sukumaran Nair
RK
,
Rajeswari
B
,
Thankamony
P
,
Parukuttyamma
K
.
Mucoepidermoid carcinoma of parotid gland as a subsequent neoplasm in children treated for acute lymphoblastic leukemia
.
J Cancer Res Ther
.
2015 Jul-Sep
;
11
(
3
):
655
.
62.
León
X
,
Ferlito
A
,
Myer
CM
 3rd
,
Saffiotti
U
,
Shaha
AR
,
Bradley
PJ
, et al
Second primary tumors in head and neck cancer patients
.
Acta Otolaryngol
.
2002 Oct
;
122
(
7
):
765
78
.
63.
Lim
YC
,
Lee
SY
,
Kim
K
,
Lee
JS
,
Koo
BS
,
Shin
HA
, et al
Conservative parotidectomy for the treatment of parotid cancers
.
Oral Oncol
.
2005
;
41
(
10
):
1021
7
.
64.
Venkatesh
S
,
Srinivas
T
,
Hariprasad
S
.
Parotid gland tumors: 2-year prospective clinicopathological study
.
Ann Maxillofac Surg
.
2019 Jan-Jun
;
9
(
1
):
103
9
.
65.
Harish
K
.
Management of primary malignant epithelial parotid tumors
.
Surg Oncol
.
2004
;
13
(
1
):
7
16
.
66.
Jensen
AD
,
Nikoghosyan
A
,
Windemuth-Kieselbach
C
,
Debus
J
,
Münter
MW
.
Combined treatment of malignant salivary gland tumours with intensity-modulated radiation therapy (IMRT) and carbon ions: COSMIC
.
BMC Cancer
.
2010
;
10
:
546
.
67.
Ferlito
A
,
Rinaldo
A
,
Silver
CE
,
Gourin
CG
,
Shah
JP
,
Clayman
GL
, et al
Elective and therapeutic selective neck dissection
.
Oral Oncol
.
2006
;
42
(
1
):
14
25
.
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