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A 40-year-old male patient presented with diffuse radiolucent lesions on the upper maxilla at the apex of the incisors, canines, premolars, and molars. Surgical excision of the lesions was performed, and the biopsy specimens were submitted for histological analysis: the lesions of the first quadrant were found to be a squamous odontogenic tumor, a rare benign neoplasm of odontogenic origin. The purpose of this article is to describe the clinical, radiographic and histological aspects of odontogenic squamous tumor.
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en
Antonio  D’Addona
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Annali di stomatologia
Maxilla
Squamos Odontogenic Tumor: A case report
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Original Article
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Antonio  D’Addona
2023/03/09
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Department of Head and Neck, Oral Surgery and Implantology Unit, Institute of Clinical Dentistry, Catholic University of Sacred Hearth, Fondazione Policlinico Universitario Gemelli, 00168 Rome, Italy.
1971-1441
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Maxilla
en
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Squamos Odontogenic Tumor: A case report
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                                                                                                        Original Article


Squamos Odontogenic Tumor:
A case report



Paolo De Angelis1                                                The process of tissue formation and differentiation in
Camilla Cavalcanti1                                              squamous odontogenic tumor is multifactorial; Malassez
Giuseppe De Rosa1                                                remnants may underlie epithelial proliferation in lesions
Noemi Cesaroni1                                                  associated with the alveolar process while the dental
Teresa Musarra2                                                  lamina may be the cause of the occurrence of lesions
Antonio D’Addona1                                                associated with unerupted or impacted teeth (4,5).
                                                                 Although SOT is more frequently solitary intraosseous
1
    Department of Head and Neck, Oral Surgery and Im-            lesion (central SOT), peripheral SOT and SOT-like pro-
    plantology Unit, Institute of Clinical Dentistry, Catholic   liferation arising in odontogenic cyst variants have also
    University of Sacred Hearth, Fondazione Policlinico          been described.
    Universitario Gemelli, 00168 Rome, Italy.                    Peripheral SOT is rare and affects soft tissues producing
2
    Division of Anatomic Pathology and Histology, Fon-           ‘saucerization’ of underlying bone. (6)
    dazione Policlinico Universitario A. Gemelli-IRCCS,          Histologically they are typically described as mural out-
    Largo Agostino Gemelli, 00168 Rome, Italy.                   growths inside the walls of odontogenic cysts. In fact,
                                                                 SOT from a microscopic point of view shows large islands
                                                                 of benign squamous epithelium separated by a stroma
Corresponding author:                                            of collagenous material with an outer layer of squamous
Antonio D’Addona                                                 cells. Some cases show calcifications both within the stro-
E-Mail: antonio.daddona@gmail.com                                ma and within the squamous islands. These islands may
                                                                 undergo cystic degeneration over time. (7, 8, 9)
                                                                 The usual treatment is enucleation of the lesion with ap-
Abstract                                                         propriate curettage of the affected site. Only one case
A 40-year-old male patient presented with diffuse ra-            of malignant transformation of intraosseous SOT at the
diolucent lesions on the upper maxilla at the apex of            mandibular level has been described in the literature,
the incisors, canines, premolars, and molars. Surgi-             and cases of recurrence are also quite rare.
cal excision of the lesions was performed, and the               Clinical signs typically reported are asymptomatic swell-
biopsy specimens were submitted for histological                 ing associated with tooth mobility and bone resorption.
analysis: the lesions of the first quadrant were found           Diagnosis is made primarily during routine radiographic
to be a squamous odontogenic tumor, a rare benign                examinations in which predominantly unilocular radiolu-
neoplasm of odontogenic origin. The purpose of this              cency is evident although cases of SOT with a multilocu-
article is to describe the clinical, radiographic and his-       lar appearance have been described in the literature.
tological aspects of odontogenic squamous tumor.                 Radiographically appearing with a triangular shape be-
                                                                 tween the roots of the teeth, the treatment of election is
                                                                 surgical excision with curettage of the surgical site.
Keywords: Squamos Odontogenic Tumor, Radiolu-                    The aim of this case report is to describe the diagnostic
cent lesions, Maxilla.                                           course and surgical and squamous cell tumor treatment
                                                                 in a patient treated at our department.
Introduction
                                                                 Case report
Squamous odontogenic tumor (SOT) is a rare benign
neoplasm that originates from the odontogenic epitheli-          A 40-year-old male patient came on clinical examina-
um. It was first described by Pullon in 1975 and was rec-        tion with multiple cavities and root remnants of lateral
ognized by World Health Organization (WHO) in 2005               incisors, canine, premolar and second molar in the first
and, more recently, in 2017 (1,2)                                quadrant; canine, first premolar, first and second molar
The recent WHO classification of odontogenic and max-            in the second one.
illofacial bone tumors defines SOT as “a benign epithe-          Palpation of the affected area did not result in discharge
lial odontogenic tumor in which the tumor cells show             of purulent material, and the area appeared slightly com-
terminal squamous differentiation.” SOT can manifest in          pressible. Radiographic examinations (Orthopanoramic
different age ranges; however, it is mainly diagnosed in         and Cone Beam) showed well-circumscribed radiotrans-
the fourth decade of life. The sites mainly involved are         lucent lesions of spherical shape nearby all root remnants.
the anterior part of the upper jaw and the posterior part        In the premolar region of the first quadrant such lesions
of the mandible (3).                                             appeared overlapped. (Fig. 1-2; 3-4)



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    Squamos Odontogenic Tumor: A case report




    Figure 1. Ortopantomography.




      Figure 2. Lateral Telecranium.




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P. De Angelis et al.




Figure 3. Element 1.2 CBCT frames




Figure 4. Element 1.6 CBCT frames.




Treatment                                                        Moreover, severely compromised elements 22 and 24
                                                                 and root residues 23 24 and 26 were extracted.
Treatment included extraction of root remnants and               In order to get primary intention healing, all the sharp
compromised dental elements, removal of lesions for              corners of the crest were smoothed with a diamond bur
histologic analysis, and adequate curettage of the post-         and silk 4/0 sutures were used.
extraction cavity and washings with physiological saline.        Then, the same crestal full thickness-flap was subse-
The patient was treated under general anesthesia and             quently made in the first quadrant from the incisal area
since the lesion corresponding to elements 22 23 24              to the last molar. The dental elements and root residues
was the most extensive the surgery started from the              were all extracted except for 11.
second quadrant: after plexus anesthesia with adrena-            In this area there were multiple lesions of a cystic na-
line 1:100.000, a full thickness crestal flap with distal dis-   ture, the major one involving the lateral incisor and they
charge was made from the second molar area up to the             appeared very similar to that of the second quadrant
central incisor. A mucoperiosteal flap was detached until        that we had previously enucleated. Once all the lesions
a good view of the areas affected by the cystic lesions          had been excised, an accurate curettage was per-
was obtained.                                                    formed with abundant physiological washes. The se-
An osteotomy was then performed until the right cleav-           verely compromised buccal cortex was smoothed and
age was obtained to mobilize the cystic epithelium until         made as regular as possible before the suture. It was
it was completely “shelled out”. The lesion excised from         not possible to completely close the flap in correspon-
the surrounding bone consisted of fibrous and compress-          dence of the cavity at the level of the buccal molar area
ible tissue with a citrine yellow liquid content. Curettage      where a dehiscence remained. All the lesions were col-
of the cavity and abundant washing was then carried out.         lected for histological analysis.



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    Squamos Odontogenic Tumor: A case report

    After the surgery, the patient was given zitromax (500 mg)    Diagnosis
    for three days, ibuprofen (600 mg) for four days and mouth-
    rinses with clorexidine 0,12 % , twice a day, for a week.     Histologic analysis of the lesion from the second quadrant
    Sutures were removed after one week and a dehiscence          revealed a cystic formation with multi-layered non-kera-
    was noticed on the first quadrant with bone exposure: a       topoietic hyperplastic epithelial lining due to a chronic in-
    revision and curettage of the zone was performed and it       flammatory process: it was compatible with radicular cyst.
    was closed by primary intention.                              As regards the first quadrant, three samples were ana-
    One week later, the area completely healed and the next       lysed and they revealed a multi-layered epithelial lining
    step will be the rehabilitation of the upper jaw with a re-   associated with surrounding nests of variably shaped
    movable prothesis and a follow-up at 6 months.                and sized epithelial cells without atypia: the samples
    As regards the lower jaw, also multiple cavities, root rem-   were compatible with the diagnosis of squamous odon-
    nants and radiolucent lesions are detectable: elements        togenic tumor (SOT). (Fig. 6-7)
    48,47,43,35,36,38 will be extracted and all the related
    lesions will be enucleated and analyzed (Fig. 5).




    Figure 5. Enucleation of the lesion.




                                                                            Figure 6. SOT 1x section stained with Hematox-
                                                                            ylin-Eosin.



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P. De Angelis et al.




                 Figure 7. SOT 10x section stained with Hematoxylin-Eosin.




Discussion                                                      Also Barbeiro et al. (2021) described a case of maxil-
                                                                lary dentigerous cyst involving tooth 18 which showed
Squamous Odontogenic Tumor (SOT) is a benign lesion             squamous odontogenic tumor like proliferation: their
with slow proliferation rate that is thought to origin from     group managed to treat it by enucleation of the lesion
dental lamina remnants. However, it is locally infiltrative     and involved tooth and obtained complete healing and
and it may cause tooth displacement or un-eruption and          no recurrence after 3 year follow-up. Considering all clin-
root divergency, irregular swelling and bony expansion          ical differences between the two kind of lesions it is not
due to the perforation of the cortical bone; other cases of     possible to assert that SOT is the natural progression of
mild discomfort, soreness or a feeling of pressure when         SOT-LPOC. (12)
the nerves are involved have been reported; sometimes,          The election treatment modality is conservative enucle-
it is totally asymptomatic and discovered with routine ex-      ation with curettage; other cases reported in literature
ams. In our case the patient did not have any kind of           received a more aggressive treatment consisting on en
symptom related to the growth of the lesions and was re-        bloc resection, hemimaxillectomy or radical alveolecto-
ferred to the oral surgery department to treat the multiple     my. Moreover, it is suggested to treat multilocular, multi-
cavities and dental remnants. As reported in literature,        focal and/or recurrent lesions involving maxilla, which is
the most common location of the SOT is the premolar-            known to be more porous than mandible, with an exten-
canine in maxilla or the molar region of the mandible.          sive intervention. (13)
Our patient showed multiple radiolucent lesions in all          Cases of recurrence of SOT are reported in literature es-
quadrants and we attempted to analyze the ones affect-          pecially when enucleation was performed but were not
ing the first and the second ones. (10)                         influenced by the age of the patient, location of the le-
The histological analysis revealed the presence of both         sion, expansion and perforation of cortical bone and loc-
SOT and a radicular cyst, which come from proliferation         ularity in radiological exams. Since there are no guide-
of Malassez remnants after inflammatory stimulus. Also          lines about the gold stardard of treatment of SOT, it is
SOT is frequently associated with necrotic or unerupted         suggested that the surgeon would evaluate the suitable
teeth which may have cause the proliferation of dental          surgical treatment for the case.
lamina, thus suggesting a mild correlation between the          In our case, the enucleation was completed with accu-
tumor and the cyst.                                             rate curettage and physiological washes due to the wide
A review by Chrnovic and Gomez (2018) analyzed cases            extension of the lesions: we preferred not to extend the
of proliferation of SOT and squamous odontogenic tu-            resection beyond the cavities and preserve the residual
mor-like proliferation in odontogenic cysts (SOT-LPOC)          bone crest to a future rehabilitation.
thus concluding that they are different clinicopathological
conditions since SOT showed a more aggressive growth
pattern: the former had unilocular or multilocular aspect
                                                                Conclusion
and was associated with cortical bone perforation in 62         Our study is an additional case of intraosseos multifo-
% of central lesions and tooth displacement while the           cal SOT, which has been described in literature as a
latter showed no signs of bone erosion or root resorption       rare but locally aggressive odontogenic tumor with low
and were most associated with radicular and dentiger-           recurrence rate: our approach was conservative since
ous cysts, as it appeared most of the times as an uni-          the patient had multiple lesions and we attempted to
locular lesion. (11)                                            preserve the bone volume. The limitation of the study


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    Squamos Odontogenic Tumor: A case report

    is the short follow-up, which will be updated, and the                      port and Review of the Literature. Head Neck Pathol. 2017
    absence of a surgical protocol treatment, since in litera-                  Jun;11(2):168-174.
    ture there are still not data and guidelines to treat SOT             7.    Hopper TL, Sadeghi EM, Pricco DF. Squamous odontogenic
    with predictable results.                                                   tumor. Report of a case with multiple lesions. Oral Surg Oral
                                                                                Med Oral Pathol. 1980 Nov;50(5):404-10.
                                                                          8.    Norris LH, Baghaei-Rad M, Maloney PL, Simpson G, Guinta
                                                                                J. Bilateral maxillary squamous odontogenic tumors and the
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