Oral Oncology 50 (2014) 84–89

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Oral Oncology
journal homepage: www.elsevier.com/locate/oraloncology

Review

Oral squamous cell carcinoma in patients twenty years of age or

younger – Review and analysis of 186 reported cases
Lipa Bodner a,⇑, Esther Manor b, Michael D. Friger c, Isaac van der Waal d
a
Department of Oral and Maxillofacial Surgery, Soroka Medical Center, Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel
b
Institute of Human Genetics, Soroka Medical Center, Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel
c
Department of Public Health, Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel
d
Department of Oral and Maxillofacial Surgery/Oral Pathology, VU University Medical Center/ACTA, Amsterdam, The Netherlands

a r t i c l e i n f o s u m m a r y

Article history: To review the literature on reported cases of squamous cell carcinoma (SCC) of the oral cavity in patients
Received 22 August 2013 twenty-years-of-age or younger.
Received in revised form 7 November 2013 All well-documented cases of oral SCC in patients twenty-years-of-age or less, published between 1936
Accepted 11 November 2013
and 2012, were collected and the clinicopathologic features were evaluated. Primary cases of oral SCC
Available online 2 December 2013
were selected. Age, gender, intra-oral subsite were recorded.
A total of 88 articles describing 186 cases were included. The group of otherwise healthy patients had a
Keywords:
mean age of 14.08 years (range newborn–20 years), the m:f ratio was 1.36; the oral subsites were the
Squamous cell carcinoma
Oral
tongue, gingiva, and lower lip. A second group of patients who have disorders that predispose to cancer
Children development, such as xeroderma pigmentosum, Fanconi’s anemia, and a history of bone marrow trans-
Young adults plant, had a mean age of 13.17 years (range 5–20 years); the m:f ratio was 1.23; the oral subsite was
mainly the tongue. There was a slight difference between otherwise healthy patients and patients with
predisposing systemic factors, but this difference was not statistically significant.
It contrast to adults, there is only a weak predilection for males (m:f ratio of 1.23–1.36). In the young
population SCC occurs most frequently in the tongue, followed by gingiva and lips. Unlike in adults, SCC is
very uncommon in the floor of mouth.
Oral SCC may, indeed, occur in patients younger than 20 years and clinicians should take cognizance of
this. Periodical examination of the oral cavity of young patients is recommended in cases of systemic
diseases that predispose to cancer development such as xeroderma pigmentosum, Fanconi’s anemia,
and a history of bone marrow transplant.
Ó 2013 Published by Elsevier Ltd.

Introduction
Oral squamous cell carcinoma (SCC) typically occurs in the
elderly during the fifth through the eighth decades of life and is
associated with a high incidence of tobacco and alcohol abuse.
Patients younger than 40-years-of-age account for less than 4% of
all oral malignancies. In the pediatric and adolescent age subgroup
it is considered to be very rare. The tongue and lip are the main
subsites in the young population, whilst in adults the tongue and
floor of mouth are the regions most affected. Metastases are very
rare in the young population [1–3].
Oral SCC in young patients is believed to be etiologically distinct
from oral SCC in older adults, due to less significant exposure to the
⇑ Corresponding author. Address: Department of Oral and Maxillofacial Surgery,
Soroka Medical Center, Ben-Gurion University of the Negev, P.O. Box 151, Beer-
Sheva 84101, Israel. Tel.: +972 8 6400505; fax: +972 8 6483366.
E-mail address: lbodner@bgu.ac.il (L. Bodner).
common risk factors such as tobacco and alcohol. On the other
hand, the outcome of oral SCC in older patients with or without
known risk factors was found to be similar, suggesting that the
pathogenesis involves factors other than smoking and alcohol [4].
Although oral SCC in the young population seems to represent a
distinct biological entity that is different from its adult counter-
part, the underlying genetic causes remain unknown [5,6].
Some studies report that oral SCC follows a more aggressive
course in young patients [7,8], while other studies suggest that
age is not a predictor of outcome [9–11].
Recently, it has been reported that there is an increasing
incidence of oral SCC in younger individuals [12–15].
The aim of the present study was to review the literature on oral
SCC in pediatric and adolescent patients younger than 20 years,
compare the data with the literature on an adult oral SCC popula-
tion, and: (a) elucidate whether there is a gender-based or an oral
site-based predilection among these young individuals, (b)
evaluate the possible higher risk among patients with systemic

1368-8375/$ - see front matter Ó 2013 Published by Elsevier Ltd.

http://dx.doi.org/10.1016/j.oraloncology.2013.11.001
 L. Bodner et al. / Oral Oncology 50 (2014) 84–89
cancer predisposing factors, and (c) look for a possible risk factor
that might be more specific for SCC in children and adolescents.
Materials and methods
The review is based on articles published in the literature
between 1936 and 2012. Data were collected regarding age,
gender, oral sub-site and histologic differentiation. Student–
Newman–Keuls test was used for statistical analysis with p = 0.05
for significant differences.
Results
A list of on 156 reported cases of oral SCC in patients 20-years-
of-age or less, who are generally healthy, is shown in Table 1
(group I). A list of reports on 30 cases of oral SCC in patients 20-
years-of-age or less who have an associated systemic condition
that predisposes to cancer development is shown in Table 2 (group
II).
Age and gender
Age ranged from newborn to 20-years-of-age. The mean age in
group I was 14.08 years; the mean age in group II was 13.17 years.
The difference between the groups was not statistically significant
(p = 0.913). Gender was documented in 133 cases, 76 were male
and 57 were female, with a general m:f ratio of 1.33. In group I
the m:f was 1.36; in group II it was 1.23. The difference between
the groups was not significant (p > 0.05).
Oral subsite was documented in 144 cases. In Group I, the most
common subsite was the tongue (69.4%) followed by the gingiva
(20%) and the lip (5%) The floor of the mouth was affected only
in 2 (2%) cases. In group II 2 the most common subsite was the
tongue (73%), followed by the gingiva (13%) and buccal mucosa
(6.7%). The differences in oral subsites between the groups was
not statistically significant (p > 0.05).
The histologic differentiation was documented in only 34 cases;
therefore, this parameter has not been further evaluated in this
study.
Discussion
Oral subsite
A striking difference in oral subsites was found between our
young patients and adults. Among adults, the classic oral subsites
are: tongue (38%), floor of mouth (26%), buccal mucosa (22%),
and gingiva (13%) [96,97]. In the present review among healthy pa-
tients under the age of 20 years, the most common oral subsites
were: tongue (69%), gingiva (20%), and lower lip (5%). Only 2 out
of 144 documented cases (1.4%) of floor of mouth involvement
were found.
It is hypothesized that the stem cell activity in the tooth-bear-
ing area is the possible reason for the high incidence of gingival
SCC found in children [98].
While in adults there is a strong male predominance, with a
reported m:f ratio of 2.3–1.27 [50,97,99–103], the gender predilec-
tion found in children and adolescents was weak, with a m:f of
1.36–1.23. The possible reason is the etiopathogenesis. In adults
the classic carcinogenic risk factors, such as smoking and alcohol
abuse, may be more common among males, causing the male pre-
dilection. Among children, where the exposure to these risk factors
is very limited, some other factors are probably more active,
resulting in a neglectable sex predilection.
85
Etiopathology
The pathogenesis of oral SCC affecting young patients has long
been debated. The classic risk factors most strongly implicated in
oral SCC such as tobacco smoking, alcohol consumption, and betel
quid use [104], cannot be considered major risk factors, due to the
limited duration of the exposure of young patients to these risk
factors [101].
It has been postulated that tumorigenesis of oral SCC is not
dependent only on the type, duration and level of exposure to
a specific carcinogen, but rather on the genetic sensitivity of
the individual [105]. The clonal and non-clonal chromosomal
aberrations in the upper aerodigestive tract mucosa are age
dependent. The sum of genetic changes in a given individual is
determined by two major parameters, the level of exposure to
mutagenic factors and the inherent ability to efficiently repair
DNA damage.
Obviously, the overall exposure to mutagens on average is
greater in older individuals [106]. Overexpression of P53 tumor
suppressor gene was found in young patients 18–39-years-of-age
who had tongue SCC, with no known risk factors, similar or higher
to the expression found among the ‘‘classical’’ SCC [107].
Chronic inflammation as a risk factor for cancer in general was
observed in the association of various chronic inflammatory dis-
eases, including irritable bowel syndrome, atrophic gastritis,
chronic cholecystitis, and reflux esophagitis with the development
of cancer [108]. The possible role of inflammation in oral carcino-
genesis has been also discussed [109,110].
Oral SCC among the patients in group II, who have systemic dis-
eases or syndromes that predispose to cancer development, is
somewhat different. The development of oral cancer among them
is likely to be related to genetic instability. Tumor suppressor
genes are now thought of as either gatekeepers or caretakers.
The gatekeepers control cell growth by inhibiting cell proliferation
or promoting apoptosis. The caretakers maintain the integrity of
the genome by DNA repair mechanisms [111]. Inactivation of a
caretaker gene is thought to promote neoplasia indirectly by facil-
itating genetic instability, which in turn leads to increased muta-
tions of all genes, including gatekeepers. Patients with
syndromes or predisposing factors for cancer inherit a single
mutant allele of a caretaker gene from an effected parent and only
develop cancer after 3–4 somatic mutations.
Generally, abnormalities of gatekeeper genes do not predispose
to oral cancer, with the exception of Li Fraumeni syndrome, which
promotes second primary malignancies, including oral cancer. The
syndromes that are associated with caretaker genes are:
xeroderma pigmentosum, ataxia telangiectasia, Bloom syndrome,
colorectal cancer, Werner syndrome, and Fanconi’s anemia. Genet-
ic instability may, indeed, be of fundamental importance in the
pathogenesis of oral cancer [112]. Patients with Fanconi’s anemia
have an increased risk of developing non-hematological second
primary malignancies, especially head and neck carcinoma. In
our study on 21 cases of FA, 11 cases (52%) were on the tongue
and 4 cases (19%) were found on the gingiva. This is compatible
with the findings in adults, where the oral SCC is mainly found
on the tongue and gingiva [113].
Management and prognosis
The standard course of treatment for pediatric oral SCC has to
be specifically tailored to each patient after appropriate consulta-
tion with an interdisciplinary team. Generally, it will be a wide
surgical excision with tumor free margins, while trying to avoid
the sequelae of radiation therapy [67]. The prognosis is similar to
that of adults [9–11].
86 L. Bodner et al. / Oral Oncology 50 (2014) 84–89

Table 1
Reports on oral SCC in otherwise healthy paediatric and adolescent patients less than twenty years of age (n = 156).

Author Year Age (years) Gender Oral subsite

Frank et al. [16] 1936 Newborn M Tongue
Saleeby [17] 1940 15 F Tongue
New and Hertz [18] 1940 13 M Tongue
DeNicola [19] 1946 12 F Lower lip
Kapiloff [20] 1952 0.5 M Lower lip
Merrifield et al. [21] 1955 5 F Floor of mouth
Moore [22] 1958 13 M Tongue
Frazelle and Lucas [23] 1962 19 F Tongue
Venables [24] 1966 17 F Tongue
Elfenbaum [25] 1967 11 M Gingiva
Venables and Craft [26] 1967 17 F Tongue
Lancester and Fournet [27] 1969 6 M Tongue
Jones [28] 1970 10 M Floor of mouth
15 M Gingiva-mandible
Goyanes and Frazell [29] 1971 16–19 Tongue (n = 4)
Pichler et al. [30] 1972 19 M Tongue
Turner and Snitzer [31] 1974 12 M Tongue
Byers [32] 1975 17 Tongue
19 Tongue
19 Tongue
19 Tongue
Patel and Dave [33] 1976 16 M Tongue
17 M Tongue
16 M Tongue
11 M Tongue
11 M Tongue
Krolls and Hoffman [1] 1976 14 M Tongue
14 F Tongue
14 M Lower lip
Soni and Chatterji [34] 1982 11 M Tongue
McGregor et al. [35] 1983 18 F Tongue
Newman et al. [36] 1983 14–18 Tongue (n = 4)
Son and Kapp [37] 1985 10 M Gingiva-maxilla
17 F Buccal mucosa
18 M Tongue
19 M Tongue
Sacks et al. [38] 1985 13 M Gingiva- maxillary
Usenius et al. [39] 1987 14 M Tongue
Karja et al. [40] 1988 14 M Tongue
7 F Tongue
10 F Tongue
Earle et al. [41] 1988 7 M Gingiva-maxillary
Amichetti [42] 1989 14 F Tongue
Lund and Howard [43] 1990 20 Tongue
Tsukuda et al. [44] 1993 14–19 2-M Not specified (n = 4)
2-F
Atula et al. [45] 1996 19 M Tongue
de Carvalho et al. [46] 1998 6 F Gingiva-maxilla
14 M Gingivo-buccal-maxillary
15 F Soft palate
Thompson et al. [2] 1999 20-February 10-M Tongue (n = 9)
10-F Lip (n = 6)
Not specified (n = 5)
Jin et al. [5] 1999 6.5 F Gingiva
19 M Lower lip
Torossian et al. [47] 2000 13 M Tongue
Oliver et al. [48] 2000 20 F Tongue
Myers et al. [49] 2000 19 Tongue
Pitman et al. [50] 2000 16 Tounge
Soni et al. [51] 2001 8 F Tongue
Bill et al. [52] 2001 14 M Gingiva-mandible
Wang et al. [53] 2001 20 M Tongue
Annertz et al. [54] 2002 20 Tongue
Veness et al. [9] 2003 19 Tongue
Hyam et al. [55] 2003 19 Tongue
Lamaroon et al. [56] 2004 17 Not specified (n = 1)
Sturgis et al. [57] 2005 18 M Tongue
16 F Tongue
Sasaki et al. [58] 2005 19 F Not specified (n = 1)
O’Regan et al. [59] 2006 16–20 M Not specified (n = 1)
Ajayi et al. [60] 2007 M Gingiva mandible
Binahmed et al. [61] 2007 10 F Gingiva-maxilla
Effion et al. [62] 2008 1st decade-3 M-15 Not specified (n = 19)
2nd decade-16 F-4
 L. Bodner et al. / Oral Oncology 50 (2014) 84–89 87

Table 1 (continued)

Author Year Age (years) Gender Oral subsite

Seyedmajidi and Faizabadi [63] 2008 13 M Tongue
Randhawa et al. [64] 2008 18 F Tongue
Stolk-Liefferink et al. [65] 2008 11 M Gingiva-maxilla
Mehanna et al. [66] 2009 10 M Gingiva-maxilla
Woo et al. [3] 2009 18 F Gingiva-maxilla
17 M Gingiva-maxilla
11 F Gingiva-maxilla
11 F Gingiva-maxilla
Sidell et al. [67] 2009 6 M Gingiva-maxilla
Marochino et al. [13] 2010 1st decade-1 Not specified (n = 11)
2nd decade-10
Fadoo et al. [68] 2010 11 F Tongue
Nagy et al. [69] 2010 15 F Tongue
Morris et al. [70] 2010 15–20 2-M Tongue (No = 10)
8-F
Bachar et al. [4] 2010 15 Tongue
Hutton et al. [71] 2010 7 F Gingiva- maxilla
Moubayed et al. [72] 2011 Neonate F Lowe lip
Riberio et al. [73] 2011 7 M Gingiva-maxilla
Solanki et al. [74] 2012 10 M Gingiva-maxilla
Abdel-Aziz [75] 2012 15 F Tongue
Harirchi et al. [76] 2012 15 F Tongue

Table 2
Reports on oral SCC in paediatric and adolescent patients less than twenty years of age who have systemic disease (n = 30).

Author Year Age Sex Oral subsite Associated conditions

Vaitiekaitis and Grau [77] 1980 19 F Gingiva-mandible FA
Harper and Copeman [78] 1981 18 M Tongue XP
Yagi et al. [79] 1981 10 F Tongue XP
Kennedy and Hart [80] 1982 20 F Tongue FA
Kaplan et al. [81] 1985 13 F Tongue FA
Kozhevnikov and Khodorenko [82] 1986 14 M Buccal mucosa FA
Keukens et al. [83] 1989 9 M Tongue XP
Murayama et al. [84] 1990 11 M Tongue FA
Socie et al. [85] 1991 12 M Tongue FA
Huet-Lamy et al. [86] 1992 11 M Tongue XP
Morehead et al. [87] 1993 12 M Tongue Acute leukemia
Anil et al. [88] 1994 17 F Tongue DCG
Somers et al. [89] 1995 Tongue FA
Millen et al. [90] 1997 18 F Buccal mucosa FA
Abdelsayed et al. [91] 2002 14 M Tongue BMT, GVHD
Salum et al. [92] 2006 12 F Tongue FA
Reinhart et al. [93] 2007 13 F Tongue FA
Chow et al. [14] 2007 15 M Tongue FA, BMT, RT
14 M Tongue FA, BMT, RT
16 F Tongue Dermatomyosis
11 F Cheek/Palate CT/RT Ewing’s
Masserot et al. [94] 2008 12 M Tongue FA, BMT
11 M Tongue FA, BMT
9 F Tongue FA, BMT
5 M Tongue FA, BMT
10 M Palate FA, BMT
19 M Gingiva FA, BMT
14 F Gingiva FA, BMT
7 F Tongue FA, BMT
Erdem et al. [95] 2011 16 M Gingiva-mandible FA, BMT

FA – Fanconi’s anemia, XP – xeroderma pigmentosum, DCG – dyskeratosis congenita, BMT – bone marrow transplant, GVHD – graft versus host disease, CT – chemotherapy,
RT – radiotherapy.

Conclusion oral subsites in children and young adults. Treatment involves

resection with tumor free margins, without radiation, if feasible.
In view of the possible occurrence of oral SCC in children and Further studies are necessary to identify more specific etiopatho-
adolescents in general and in patients with predisposing factors logic risk factors associated with pediatric and adolescent oral SCC.
to cancer development in particular, clinicians should be alert to
the fact that young patients can develop oral cancer. The best out-
Conflict of interest
come is likely to be achieved by early diagnosis. The tongue and
the gingiva, mainly the maxillary gingiva, are the most common
None.
88 L. Bodner et al. / Oral Oncology 50 (2014) 84–89

References [35] McGregor GI, Davis N, Robins RE. Squamous cell carcinoma of the tongue and
lower oral cavity in patients under 40 years of age. Am J Surg
1983;146:88–92.
[1] Krolls SO, Hoffman S. Squamous cell carcinoma of the oral soft tissues: a
[36] Newman AN, Rice DH, Ossoff RH, Sisson GA. Carcinoma of the tongue in
statistical analysis of 14,253 cases by age, sex and race of patients. J Am Dent
persons younger than 30 years of age. Arch Otolaryngol 1983;109:302–4.
Assoc 1976;92:571–4.
[37] Son YH, Kapp DS. Oral cavity and oropharyngeal cancer in a younger
[2] Thompson L, Castle J, Heffner DK. Oral squamous cell carcinoma in pediatric
population. Review of literature and experience at Yale. Cancer
patients: a clinicopathologic study of 20 cases [abstract]. Oral Surg Oral Med
1985;15:441–4.
Oral Pathol 1999;88:204.
[38] Sacks HG, Holly R, Blum B, Joy ED. Case 58: maxillary alveolar mass in a 13-
[3] Woo VL, Kelsch RD, Su L, Kim T, Zegarelli DJ. Gingival squamous cell
year-old boy. J Oral Maxillofac Surg 1985;43:958–63.
carcinoma in adolescence. Oral Surg Oral Med Oral Pathol Oral Radiol Endod
[39] Usenius T, Karja J, Collan Y. Squamous cell carcinoma in children. Cancer
2009;107:92–9.
1987;60:236–9.
[4] Bachar G, Hod R, Goldstein DP, Irish JC, Gullane PJ, Brown D, et al. Outcome of
[40] Karja J, Syrjanen S, Usenius T, Vornanen M, Collan Y. Oral cancer in children
oral tongue squamous cell carcinoma in patients with and without known
under 15 years of age. A clinicopathological and virological study. Acta
risk factors. Oral Oncol 2011;47:45–50.
Otolaryngol Suppl 1988;449:145–9.
[5] Jin YT, Myers J, Tsai ST, Goepfert H, Bastakis JG, el-Naggar AK. Genetic
[41] Earle AS, Park CH, Vlastou C. Oral squamous cell carcinoma in children. Ann
alterations in oral squamous cell carcinoma of young adults. Oral Oncol
Plast Surg 1988;20:148–52.
1999;35:251–6.
[42] Amichetti M. Squamous cell carcinoma of the oral tongue in patients less than
[6] Koch WM, Lango M, Sewell D, Zahurak M, Sidransky D. Head and neck cancer
fifteen years of age. J Cranio-Max Fac Surg 1989;17:75–7.
in nonsmokers: a distinct clinical and molecular entity. Laryngoscope
[43] Lund VJ, Howard DJ. Head and neck cancer in the young: a prognostic
1999;109:1544–51.
conudrum? J Laryngol Otol 1990;104:544–8.
[7] Sarkaria JN, Harai PM. Oral tongue cancer in young adults less than 40 years of
[44] Tsukuda M, Ooishi K, Mochimatsu I, Sato H. Head and neck carcinoma in
age: rationale for aggressive therapy. Head Neck 1994;16:107–11.
patients under the age of forty years. Jpn J Cancer Res 1993;84:748–52.
[8] Goldstein DP, Irish JC. Head and neck squamous cell carcinoma in young
[45] Atula S, Grenman R, Laippala P, Syriaenen S. Cancer of the tongue in patients
patient. Curr Opin Otolaryngol Head Neck Surg 2005;13:207–11.
younger than 40 years. A distinct entity? Arch Otolaryngol Head Neck Surg
[9] Veness MJ, Morgan GJ, Sathiyaseelan Y, Gebsky V. Anterior tongue cancer: age
1996;122:1313–9.
is not a predictor of outcome and should not alter treatment. ANZ J Surg
[46] de Carvalho MB, de Sobrinho I de A, Rapoport A, Fava AS, Mendes AF, Kanda
2003;73:899–904.
JL, et al. Head and neck squamous cell carcinoma in childhood. Med Pediatr
[10] Popovtzer A, Shpitzer T, Bahar G, Marshak G, Ulanovski D, Feinmesser R.
Oncol 1998;31:96–9.
Squamous cell carcinoma of the oral tongue in young patients. Laryngoscope
[47] Torossian JM, Beziat JL, Bejuri FT. Squamous cell carcinoma of the tongue in a
2004;114:915–7.
13-year-old boy. J Oral Maxillofac Surg 2000;58:1407–10.
[11] Morris LG, Patel SG, Shah JP, Ganly I. Squamous cell carcinoma of the oral
[48] Oliver RJ, Dearing J, Hindle I. Oral cancer in young adults: report of three cases
tongue in the pediatric age group: a matched-pair analysis of survival. Arch
and review of the literature. Br Dent J 2000;188:362–5.
Otolaryngol Head Neck Surg 2010;136:697–701.
[49] Myers JN, Elkins T, Roberts D, Byers RM. Squamous cell carcinoma of the
[12] Patel SC, Carpenter WR, Tyree S, Couch ME, Weissler M, Hackman T, et al.
tongue in young adults: increasing incidence and factors that predict
Increasing incidence of oral tongue squamous cell carcinoma in young white
treatment outcome. Otolaryngol Head Neck Surg 2000;122:44–51.
women age 18 to 44 years. J Clin Oncol 2011;29:1488–94.
[50] Pitman KT, Johnson JT, Wagner RL, Myers EM. Cancer of the tongue in patients
[13] Marocchio LS, Lima J, Sperandio FF, Correa L, de Sousa SO. Oral squamous cell
less than forty. Head Neck 2000;22:297–302.
carcinoma: an analysis of 1,564 cases showing advances in early detection. J
[51] Soni S, Radel E, Smith RV, Edelman M, Sattenberg R, Walder S, et al. Stage 4
Oral Sci 2010;52:267–73.
squamous cell carcinoma of the tongue in a child: complete response to
[14] Chow CW, Tabrizi SN, Tiedemann K, Waters KD. Squamous cell carcinomas in
chemotherapy. J Pediatr Hematol Oncol 2001;23:612–5.
children and young adults: a new wave of very rare tumor? J Pediatr Surg
[52] Bill TJ, Reddy VR, Ries KL, Gampper TJ, Hoard MA. Adolescent gingival
2007;42:2035–9.
squamous cell carcinoma: report of a case and review of the literature. Oral
[15] Mackenzie J, Ah-See K, Thakker N, Sloan P, Maran AG, Birch J, et al. Increasing
Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:682–5.
incidence of oral cancer amongst young persons: what is the aetiology? Oral
[53] Wang Y, Irish J, MacMillan C, Brown D, Xuan Y, Boyington C, et al. High
Oncol 2000;36:387–9.
frequency of microsatelite instability in young patients with head and neck
[16] Frank LW, Enfield CD, Miller AJ. Carcinoma of the tongue in a newborn child:
squamous cell carcinoma: lack of involvent of the mismatch repair genes
report of a case. Am J Cancer 1936;26:775–7.
Hmlh1 and Hmsh2. Int J Cancer 2001;93:353–60.
[17] Saleeby GW. Cancer of the tongue in young subjects with report of a case. Am
[54] Annertz K, Anderson H, Biorklund A, Moller T, Kantolla S, Mork J, et al.
J Cancer 1940;38:257–62.
Incidence and survival of squamous cell carcinoma of the tongue in
[18] New GB, Hertz CS. Malignant disease of the face, mouth, pharynx and larynx
Scandinavia, with special reference to young adults. Int J Cancer
in the first three decades of life. Surg Gynecol Obstet 1940;70:163.
2002;101:95–9.
[19] DeNicola RR. Cancer of the lip in a twelve year old girl. West J Surg Obstet
[55] Hyam DM, Conway RC, Sathiyaseelan Y, Gebsky V, Morgan GJ, Walker DM,
Gynecol 1946;54:300–2.
et al. Tongue cancer: do patients younger than 40 do worst? Aust Dent J
[20] Kapillof B. Squamous cell carcinoma in children. Plast Reconstr Surg
2003;48:50–4.
1952;10:50–4.
[56] Lamaroon A, Pattanaporn K, Pongsiriwet S, Wanachantararak S, Propayasatok
[21] Merrifield FW, Dalitsch WW, Steiner MM. Epidermoid carcinoma of the
S, Jittidecharacs I. Analysis of 587 cases of oral squamous cell carcinoma in
tongue and floor of mouth in infancy and childhood: report of a case and
northern Thailand with focus on young people. Int J Oral Maxillofac Surg
review of the literature. Am J Dis Child 1955;89:281.
2004;33:84–8.
[22] Moore C. Visceral squamous cell cancer in children. Pediatrics
[57] Sturgis EM, Moore BA, Glisson BS, Kies MS, Shin DM, Byers RM. Neoadjuvant
1958;21:573–81.
chemotherapy for squamous cell carcinoma of the oral tongue in young
[23] Frazelle EL, Lucas Jr JC. Cancer of the tongue. Report of the management of
adults: a case series. Head Neck 2005;27:748–56.
1,554 patients. Cancer 1962;15:1085–9.
[58] Sasaki T, Moles DR, Imai Y, Speight PM. Clinico-pathological features of
[24] Venables CW. Carcinoma of the tongue in a 17-year-old girl. Proc R Soc Med
squamous cell carcinoma of the oral cavity in patients <40 years of age. J Oral
1966;59:58–9.
Pathol Med 2005;34:129–33.
[25] Elfenbaum A. Cancer of the gingiva in a child. Dent Dig 1967;73:124–5.
[59] O’Regan EM, Timon C, Sheils O, Codd M, O’Leary JJ, Toner M. Squamous cell
[26] Venables CW, Craft IL. Carcinoma of the tongue in early adult life. Br J Cancer
carcinoma of the head and neck in young Irish adults. Br J Oral Maxillofac
1967;21:645–50.
Surg 2006;44:203–6.
[27] Lancaster Jr L, Fournet LF. Carcinoma of the tongue in a child: report of a case.
[60] Ajayi OF, Adeyemo WL, Ladeinde AL, Ogunlewe MO, Omitola OG, Effion, et al.
J Oral Surg 1969;27:269–70.
Malignant orofacial neoplasms in children and adolescents: a
[28] Jones BJ. Oral carcinoma in the young patient. With a report of two cases. Br J
clinicopathologic review of cases in a Nigerian tertiary hospital. Int J
Oral Surg 1970;8:159–64.
Pediatr Otorhinolaryngol 2007;71:959–63.
[29] Goyanes AD, Frazell EL. Cancer of the tongue in young persons. J Surg Oncol
[61] Bimhamed A, Charles M, Campisi P, Forte V, Carmichael RP, Sandor GKB.
1971;3:421–9.
Primary squamous cell carcinoma of the maxillary alveolus in a 10-year-old
[30] Pichler AG, Williams JR, Moore JA. Carcinoma of the tongue in childhood and
girl. JCDA 2007;73:715–7.
adolescence. Report of a case and review of the literature. Arch Otolaryngol
[62] Effion OA, Adeyemo WL, Omitola OG, Ajayi OF, Emmanuel MM, Gbotolorum
1972;95:178–9.
OM. Oral squamous cell carcinoma: a clinicopathologic review of 233 cases in
[31] Turner H, Snitzer J. Carcinoma of the tonguein a child. Report of a case and
Lagos, Nigeria. J Oral Maxillofac Surg 2008;66:1595–9.
review of the literature. Oral Surg Oral Med Oral Pathol 1974;37:663–7.
[63] Seyedmajidi M, Faiabadi M. Squamous cell carcinoma of the tongue in a 13-
[32] Byers RM. Squamous cell carcinoma of the oral tongue in patients less than
year-old boy. Arch Iran Med 2008;11:341–3.
thirty years of age. Am J Surg 1975;130:475–8.
[64] Randhawa T, Shameena P, Sudha S, Nair R. Squamous cell carcinoma of
[33] Patel DD, Dave RI. Carcinoma of the anterior tongue in adolescence. Cancer
tongue in a 19-year-old female. Indian J Cancer 2008;45:128–30.
1976;37:917–21.
[65] Stolk-Liefferink SA, Dumans AG, van der Meij EH, Knegt PP, van der Wal KG.
[34] Soni NK, Chatterji P. Carcinoma of the tongue in a young child of eleven years.
Oral squamous cell carcinoma in children: review of an unusual entity. Int J
Indian Pediatr 1982;19:187–9.
Pediatr Otorhinolaryngol 2008;72:127–31.
 L. Bodner et al. / Oral Oncology 50 (2014) 84–89
[66] Mehana P, Patel J, Bailey BM. Mandibular SCC in a 10 year old child – a clinical
rarity. Br J Oral Maxillofac Surg 2009;47:148–50.
[67] Sidell D, Nabili V, Lai C, Cheung G, Kirsch C, Abemayor E. Pediatric squamous
cell carcinoma: case report and literature review. Laryngoscope
2009;119:1538–41.
[68] Fadoo Z, Naz F, Husen Y, Pervez S, Hasan NU. Squamous cell carcinoma of
tongue in an 11-year-old girl. J Pediatr Hematol Oncol 2010;32:e199–201.
[69] Nagy A, Barabas J, Vannai A, Nemeth Z, Bogdan S. Report of a rare case of
tongue cancer in childhood. Orv Hetil 2010;151:462–4.
[70] Morris LG, Patel SG, Shah JP, Ganly I. Squamous cell carcinoma of the oral
tongue in pediatric age group: a matched-pair analysis of survival. Arch
Otolaryngol Head Neck Surg 2010;136:697–701.
[71] Hutton A, McKaig S, Bardsley P, Monaghan A, Parmar S. Oral carcinoma
cuniculatum in a young child. J Clin Pediatr Dent 2010;35:89–94.
[72] Moubayed SP, Chami R, Woods O, Oliver MJ, Dal Sogilo DB, Lapointe A.
Neonatal squamous cell carcinoma of the lip: case report and review of the
literature. J Clin Oncol 2011;29:e549–552.
[73] Riberio CM, Gueiros LA, Leon JE, do Carmo Abreu e Lima M, de Almeida OP,
Leao JC. Oral squamous cell carcinoma in a 7-year-old Brazilian boy. Int J Oral
Maxillofac Surg 2011;40:994–7.
[74] Solanki MC, Gandhi S, Koshy G, Mathew GC. Squamous cell carcinoma of the
maxilla in 10 year old boy: a rare case report. Int J Pediatr Otorhinolaryngol
Extra 2012;7:33–5.
[75] Abdel-Aziz M. Mucoepidermoid carcinoma of the tongue in a child. Int J
Pediatr Otorhinolaryngol Extra 2012;7:6–8.
[76] Harirchi I, Hakimian S, Kiamoosavi S, Mahmoodzadeh H. Childhood tongue
squamous cell carcinoma. J Res Med Sci 2012;17:495–7.
[77] Vaitiekaitis AS, Grau WH. Squamous cell carcinoma of the mandible in
Fanconi anemia: report of case. J Oral Surg 1980;38:372–3.
[78] Harper JI, Copeman PW. Carcinoma of the tongue in a boy with xeroderma
pigmentosum. Clin Exp Dermatol 1981;6:601–4.
[79] Yagi K, Ali A el-G, Abbas K el-D, Prabhu SR. Carcinoma of the tongue in patient
with xeroderma pigmentosum. Int J Oral Surg 1981;10:73–6.
[80] Kennedy AW, Hart WR. Multiple squamous cell carcinomas in Fanconi’s
anemia. Cancer 1982;50:811–4.
[81] Kaplan MJ, Sabio H, Wanebo HJ, Centrell RW. Squamous cell carcinoma in the
immunosuppressed patient: Fanconi’s anemia. Laryngoscope 1985;95(7 Pt
1):771–5.
[82] Kozhevnikov VA, Khodorenko SA. Cancer of the mucous membrane of the left
side of the mouth associated with congenital hypoplastic Fanconi’s anemia in
a 14-year-old boy. Vestn Khir II Grek 1986;136:105–6.
[83] Keukens F, van Voorst Vader PC, Panders AK, Vinks S, Oosterhuis JW, Kleijer
WJ. Xeroderma pigmentosum: squamous cell carcinoma of the tongue. Acta
Derm Venerol 1989;69:530–1.
[84] Murayama S, Manzo RP, Kirkpatrick DV, Robinson AE. Squamous cell
carcinoma of the tongue associated with Fanconi’s anemia: MR
characteristics. Pediatr Radiol 1990;20:347.
[85] Socie G, Henry-Amar M, Cosset JM, Devergie A, Girinskiy T, Gluckman E.
Increased incidence of solid malignant tumors after bone marrow
transplantation for severe aplastic anemia. Blood 1991;78:277–9.
[86] Huet-Lamy P, Dereure O, Degavre B, Atlan PY, Guillot B, Guilhou JJ. Carcinoma
of the tongue in xeroderma pigmentosum. Ann Dermatol Venereol
1992;119:980–2.
[87] Morehead JM, Parsons DS, McMahon DP. Squamous cell carcinoma of the
tongue occurring as subsequent malignancy in a 12-year-old acute leukemia
survivor. Int J Pediatr Otorhinolaryngol 1993;26:89–94.
[88] Anil S, Beena VT, Raji MA, Ankathil R, Vijayakumar T. Oral squamous cell
carcinoma of dyskeratosis congenita. Ann Dent 1994;53:15–8.
[89] Somers GR, Tabrizi SN, Tiedeman K, Chow CW, Garland SM, Venter DJ.
Squamous cell carcinoma of the tongue in a child with Fanconi anemia: a case
report and review of the literature. Pediatr Pathol Lab Med 1995;15:597–607.
[90] Millen FJ, Rainey MG, Hows JM, Burton PA, Irvine GH, Swirsky D. Oral
squamous cell carcinoma after allogeneic bone marrow transplantation for
Fanconi anemia. Br J Haematol 1997;99:410–4.
[91] Abdelsayed RA, Summner T, Allen CM, Treadway A, Ness GM, Penza SL. Oral
precancerous and malignant lesions associated with graft-versus-host
89
disease: report of two cases. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 2002;93:75–80.
[92] Salum FG, Martins BG, de Figueiredo MAZ, Cherubini K, Yurgel LS, Torres-
Pereira C. Squamous cell carcinoma of the tongue after bone marrow
transplantation in a patient with Fanconi anemia. Braz Dent J 2006;17:161–5.
[93] Reinhard H, Peters I, Gottschling S, Ebell W, Graf N. Squamous cell carcinoma
of the tongue in a 13-year-old girl with Fanconi anemia. J Pediatr Hematol
Oncol 2007;29:488–91.
[94] Masserot C, de Latur RP, Rocha V, Leblanc T, Rigolet A, Pascal F, et al. Head and
neck squamous cell carcinoma in 13 patients with Fanconi anemia after
hematopoietic stem cell transplantation. Cancer 2008;113:3315–22.
[95] Martin-Granizo R, Rodriguez-Campo F, Naval L, Diaz Gonzalez FJ. Squamous
cell carcinoma of the oral cavity in patients younger than 40 years.
Otolaryngol Head Neck Surg 1997;117(3 Pt 1):268–75.
[96] Martin-Granizo R, Rodriguez-Campo F, Naval L, Diaz Gonzalez FJ. Squamous
cell carcinoma of the oral cavity in patients younger than 40 years.
Otolaryngol Head Neck Surg 1997;117(3Pt1):268–75.
[97] Tremblay S, Pintor Dos Reis P, Bradley G, Galloni NN, Perez-Ordonez B,
Freeman J, et al. Young patients with oral squamous cell carcinoma: study of
the involvement of GSTP1 and deregulation of the Fanconi anemia genes.
Arch Otolaryngol Head Neck Surg 2006;132:958–66.
[98] Gronthos S, Brahim J, Li W, Fisher LW, Cherman N, Boyde A, et al. Stem cell
properties of human dental pulp stem cells. J Dent Res 2002;81:531–5.
[99] Iype EM, Pandey M, Mathew A, Thomas G, Sebastian P, Nair MK. Oral cancer
among patients under the age of 35 years. J Postgrad Med 2001;47:171–6.
[100] Llewellyn CD, Linklater K, Bell J, Johnson NW, Warnakulasuriya KAAS.
Squamous cell carcinoma of the oral cavity in patients aged 45 years and
under: a descriptive analysis of 116 cases diagnosed in the South East of
England from 1990 to 1997. Oral Oncol 2003;39:106–14.
[101] Llewellyn CD, Linklater K, Bell J, Johnson NW, Warnakulasuriya S. An analysis
of risk factors for oral cancer in young people: a case – control study. Oral
Oncol 2004;40:304–13.
[102] Rojas-Alcayaga G, Brignardello R, Espinoza I, Franco ME, Lobos N, Ortega-
Pinto AV. Oral malignant tumors: a report on 728 cases in Chile. Acta Odontl
Latinoam 2010;23:136–42.
[103] Barnes L, Eveson JW, Reichart P, Sidransky D. Pathology and genetics head
and neck tumors. Lyon, France: IARC Press; 2005. p. 168–75.
[104] Schantz SP, Hsu TC, Ainslie N, Moser RP. Young adults with head and neck
cancer express increased susceptibility to mutagen-induced chromosome
damage. JAMA 1989;262:331–5.
[105] Schantz SP, Hsu TC, Ainslie N, Moser RP. Young adults with head and neck
cancer express increased susceptability to mutagen-induced chromosome
damage. JAMA 1989;15(262):3313–5.
[106] Jin C, Jin Y, Wennerberg J, Akervall J, Grenthe B, Mandahl N, et al. Clonal
chromosome aberrations accumulate with age in upper aerodigestive tract
mucosa. Mutat Res 1997;374:63–72.
[107] Lingen MW, Chang K-W, McMurray SJ, Solt DB, Kies MS, Mittal BB, et al.
Overexpession of p53 in squamous cell carcinoma of the tongue in young
patients with no known risk factors is not associated with mutations in exons
5–9. Head Neck 2000;22:328–35.
[108] Weitzman SA, Gordon LI. Inflammation and cancer: role of phagocyte-
generated oxidants in carcinogenesis. Blood 1990;76:655–63.
[109] Piva MR, De Souza LB, Martins-Filho PR, Soares RC, De Santana Santos T, De
Souza Andrade ES. Role of inflammation in oral carcinogenesis (Part I):
histological grading of malignancy using a binary system. Oncol Lett
2011;2:1225–31.
[110] Meurman JH, Bascones-Martinez A. Are oral and dental diseases linked to
cancer? Oral Dis 2011;17:779–84.
[111] Kinzler KW, Vogelsein B. Cancer-susceptibility genes. Gatekeepers and
caretakers. Nature 1997;386:761–3.
[112] Prime SS, Thakker NS, Pring M, Guest PG, Paterson IC. A review of inherited
cancer syndromes and their relevance to oral squamous cell carcinoma. Oral
Oncol 2001;37:1–16.
[113] Lustig JP, Lugassy G, Neder A, Sigler E. Head and neck carcinoma in Fanconi’s
anaemia – report of a case and review of the literature. Eur J Cancer B Oral
Oncol 1995;31B:68–72.