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Parapharyngeal space tumors account for about 0.5% of all head and neck cancers; the majority of these rare tumors (about 80%) are benign (1). The most frequent parapharyngeal space tumor originates in salivary glands and the pleomorphic adenoma represents the most common histological type. Generally, most tumors originating from the retro-styloid space are neurogenic, while tumors coming from salivary glands usually fill the pre-styloid space. We present a case report of combined cervical trans-parotid and intraoral access in patient with double neoplasms, starting from superficial and deep parotid spreading in the parapharyngeal space. After superficial parotid neoformation removal through a pre-tragal preauricular approach, we performed an intraoral incision laterally to the left tonsil pillar. We detached the neoformation with its capsule, showing no neurovascular structure placed anteriorly to the neoplasm. Through intraoral approach, thus, it is possible to access to MPS in safety, avoiding excessively invasive incisions and approaches, in selected cases of well-defined neoformations, regardless of dimensions.
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Annali di stomatologia
oro-pharyngeal cancer
A rare case of double whartin parotid tumor. Parapharyngeal intraoral surgical approach
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Chief of Maxillo-Facial Surgery, San Filippo Neri, Rome Italy, Vice-president Operation Smile Italy 
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A rare case of double whartin parotid tumor. Parapharyngeal intraoral surgical approach
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                                                                                                   Original article


A rare case of double whartin parotid tumor.
Parapharyngeal intraoral surgical approach.


Paolo Arangio1                                              Introduction
Edoardo Cerbelli2
Alessandro Agrillo3                                         Parapharyngeal space tumors account for about 0.5%
Domenico Scopelliti4                                        of all head and neck cancers; the majority of these rare
                                                            tumors are benign (about 80%) (1).
1
    San Filippo Neri Hospital, Rome                         The most frequent parapharyngeal space tumor origi-
    Arapaolo@gmail.com                                      nates in the salivary glands and pleomorphic adenoma
2
    San Filippo Neri Hospital, Rome                         represents the most common histological type. General-
    Edoardo.cerbelli91@gmail.com                            ly, most tumors originating from the retro-styloid space
3
    San Filippo Neri Hospital, Rome                         are neurogenic, while tumors coming from salivary
    Alessandro.agrillo@me.com                               glands usually fill the pre-styloid space (2).
4
    Chief of Maxillo-Facial Surgery, San Filippo Neri,      The parapharyngeal space, also called maxillary or lat-
    Rome Italy, Vice-president Operation Smile Italy        eral pharyngeal space, is a challenging area for cancer
    Scopelliti61@gmail.com                                  treatment. It is an inverted cone-shaped space extending
                                                            from the hyoid bone to the basicranium, bounded medi-
                                                            ally by the pre-tracheal fascia and laterally by pterygoid
Corresponding author:                                       muscles and the mandible. It is frontally bounded by the
Domenico Scopelliti                                         sub-mandibular space and posteriorly by the pre-man-
Chief of Maxillo-Facial Surgery, San Filippo Neri, Rome     dibular space (3).
Italy, Vice-president Operation Smile Italy.                In most cases patients are asymptomatic, at least the
Scopelliti61@gmail.com                                      mass reaches 3 cm size, and tumors are discovered as
                                                            an incidental finding examining patients for an unrelated
                                                            problem.
Abstract
                                                            The most frequent symptoms reported were pain, dys-
Parapharyngeal space tumors account for about               phagia, odynophagia, a sense of hearing loss and mid-
0.5% of all head and neck cancers; the majority of          dle ear effusion due to Eustachian tubes obstruction
these rare tumors (about 80%) are benign (1).               and, in case of an enlarged tumor, clinical examination
The most frequent parapharyngeal space tumor                may also reveal intraoral asymmetry and protrusion of
originates in salivary glands and the pleomorphic           the anterior tonsillar pillar and/or palate (4-5).
adenoma represents the most common histological             Surgery is the main approach for the management of
type. Generally, most tumors originating from the           parapharyngeal masses except for lymphoproliferative
retro-styloid space are neurogenic, while tumors            diseases (6).
coming from salivary glands usually fill the pre-sty-       Several surgical strategies have been described for the
loid space.                                                 management of parapharyngeal space masses, and an
We present a case report of combined cervical               accurate surgical planning is mandatory, considering pa-
trans-parotid and intraoral access in patient with          tients’ nature, and neoplasm position (7).
double neoplasms, starting from superficial and             We present a case report of combined cervical trans-pa-
deep parotid spreading in the parapharyngeal space.         rotid and intraoral access in patient with double ne-
After superficial parotid neoformation removal              oplasms, starting from superficial and deep parotid
through a pre-tragal preauricular approach, we per-         spreading in the parapharyngeal space.
formed an intraoral incision laterally to the left tonsil
pillar. We detached the neoformation with its capsule,      Clinical case and surgical approach
showing no neurovascular structure placed anterior-
                                                            A 72-year-old male showed to our hospital with a
ly to the neoplasm.
                                                            10-months history of a left-sided parotid mass associat-
Through intraoral approach, thus, it is possible to
                                                            ed with an intraoral foreign body sensation.
access to MPS in safety, avoiding excessively inva-
                                                            He also reported temporomandibular joint pain, spread-
sive incisions and approaches, in selected cases of
                                                            ing in the cervical area, recurrent headaches and difficul-
well-defined neoformations, regardless of dimen-
                                                            ties in swallowing.
sions.
                                                            During clinical examination we appreciated a palpable
                                                            swelling in the left latero-cervical region, not sore nor
Keywords: multidisciplinary team care, oral medi-           painful during palpation; the same clinical finding was
cineand maxillofacial surgery, oral potentially malig-      present in the oral cavity.
nant disorders, oro-pharyngeal cancer.                      MRI showed two neoformations, the first one arising in
Annali di Stomatologia 2018; IX (4): 142-145                                                                              142
      A rare case of double whartin parotid tumor. Parapharyngeal intraoral surgical approach




      Figure 1. Axial CT                            Figure 2. Sagittal CT                    Figure 3. Coronal CT


      the left parotid area and the remaining spreading from                tached the neoformation with its capsule, showing no
      the deep lobe of the homolateral parotid gland, facing                neurovascular structure placed anteriorly to the neo-
      the oral cavity and causing a compression of the homo-                plasm, as expected by MRI images (Fig. 4).
      lateral airways. (Figg.1-3)                                           Thus, we cleaved the whole neoformation, safely, from
      In particular, this last lesion occupied the middle par-              the surrounding planes, exposing the neurovascular
      apharyngeal space (MPS) and the pre-styloid space,                    structures placed posteriorly.
      causing a neurovascular structure (internal jugular vein              The two neoformations, one deriving from the superficial
      and internal carotid artery) posterior displacement; the              lobe of the parotid gland and the other spreading from
      features made the intraoral access the most appropri-                 the deep pole, measure respectively 2.3 and 3 cm in di-
      ate approach, able to minimize the risk of aesthetic and              ameter (Fig. 5).
      functional damage.                                                    The patient was released in good conditions and the
      After superficial parotid neoformation removal, through               convalescence was carried out regularly. Neither com-
      a pre-tragal preauricular approach, we performed an                   plications nor sensitive defects were reported in the
      intraoral incision laterally to the left tonsil pillar. We de-        post-operative period.




      Figure 4. Intraoperative view of the lesion




      Figure 5. Intraoperative view after lesion removal

143                                                                                          Annali di Stomatologia 2018; IX (4): 142-145
P. Arangio et al.




         Figure 6. Patient two weeks after surgery




Headaches and TMJ disorders disappeared after sur-            opsy. The surgical removal of these tumors is the best
gery, with a total recovery within 1 month (Fig. 6).          treatment; several surgical approaches, classified as
                                                              transoral, transcervical, transparotid–transcervical, tran-
Discussion                                                    scervical–transmandibular or infratemporal, have been
                                                              described in the literature according to the position and
The para-pharyngeal space represents a challenging            type of neoformation.
area to access, for the maxillofacial surgeons, due to the    As described by Prasad et al., parapharyngeal space
depth of the location, the complex anatomy and the pres-      can be divided into three portions:
ence of adjoining vital structures.                           - Upper parapharyngeal space (UPS), from the crani-
The fascia, arising from the styloid process to the tensor        al base to the axial plane passing through the lower
veli palatine muscle, splits the parapharyngeal fossa into        edge of the lateral pterygoid muscle
two compartments: the pre-styloid and the retro-styloid       - Middle parapharyngeal space (MPS), from the axial
sections (8).                                                     plane bounded above by the UPS and below by the
This space is bounded antero-superiorly by the maxil-             insertion of the medial pterygoid muscle
lary sinus, continuing supero-laterally with the infratem-    - Lower parapharyngeal space (LPS), bounded at the
poral fossa, which is separated from the medial ptery-            top by the MPS and below by the hyoid bone (16).
goid fascia (9).                                              The goal of surgery is to ensure a complete enucleation
The retro-styloid PPS contains: IX-XII cranial nerves,        minimizing aesthetic and functional damage.
internal carotid artery (ICA), internal jugular vein (IJV),   In our patient the intraoral portion of the neoplasm oc-
paraganglia, ectopic salivary glands and lymph nodes.         cupies the pre-styloid space and the MPS, displacing
Due to the deep location, parapharyngeal space tumors         posteriorly the neurovascular structures represented by
are usually asymptomatic until the lesion reaches a di-       IJV and ICA.
mension greater than 2,5-3 cm.                                Among the intraoral approaches, we distinguish:
For this reason, in most cases, these are diagnosed in-       - Transantral sublabial
cidentally, with an imaging exam, performed for other         - Transoral transvestibular
reasons (10).                                                 - Transoral transpharyngeal (our approach)
The vast majority of PPS lesions are benign salivary          - Transmandibular transoral
gland tumors of which pleomorphic adenoma is the most         As described by Ferrari et al., one of the conservative
common (11).                                                  techniques, able to adequately expose the neoplasms
Warthin tumor extending from the deep lobe to the par-        placed on the MPS, is represented by the transoral
apharyngeal space represents a very rare occurrence,          trans-pharyngeal approach: which consists in an incision
accountant for about 1% of all histotypes involving this      placed on the front tonsil pillar (17).
region (12).                                                  Although it is discredited by some authors (18) for the
Preoperative CT, MRI and angiography are the most             greater risk of capsule rupture, dissemination in case of
common tools used to diagnose PPS tumors, investigat-         malignant tumors and infection due to contamination by
ing the relationship with major neurovascular structures,     the germs present in the oral cavity, intraoral approach is
glandular tissue, and craniofacial skeleton.                  a valid alternative, from a functional and aesthetic point
CT and MRI are both valid options, although the latter        of view.
gives more information about the lesion’s feature (13).       This was described by Goodwin and Chandler, who first
Angiography is recommended if imaging shows a wid-            presented a series of transoral parapharyngeal tumors
ening of the carotid bifurcation (14), however, PET CT is     removal (19).
recommended too when the presence of metastases is            As described by Betka et al., in case of selected tumors,
suspected (15).                                               an intraoral approach allows the access to PPS minimiz-
If malignancy is suspected, histological diagnosis should     ing side effects and ensuring a good ability to eradicate
be obtained using fine needle aspiration cytology or bi-      the disease (20).

Annali di Stomatologia 2018; IX (4): 142-145                                                                                144
      A rare case of double whartin parotid tumor. Parapharyngeal intraoral surgical approach

      Conclusion                                                           9.    Maheshwar AA, Kim EY, Pensak ML, et al. Roof of the para-
                                                                                 pharyngeal space: defining its boundaries and clinical impli-
      Through intraoral approach, thus, it is possible to access                 cations. Ann Otol Rhinol Laryngol. 2004;113:283 was 288.
      to MPS in safety, avoiding excessively invasive incisions                  doi: 10.1177/000348940411300405.
                                                                           10.   Dimitrijevic MV, Jesic SD, Mikic AA, Arsovic NA, Tomanovic
      and approaches, in selected cases of well-defined ne-                      NR. Para- pharyngeal space tumors: 61 case reviews. Int J
      oformations, regardless of dimensions.                                     Oral Maxillofac Surg 2010;39:983 was 989.
      An accurate imaging is required for a correct surgical ap-           11.   Parapharyngeal Space Pleomorphic Adenoma: A 30-Year
                                                                                 Review Abie H. Mendelsohn, MD; Sunita Bhuta, MD; Thom-
      proach to show possible displacement of neurovascular
                                                                                 as C. Calcaterra, MD; Hubert B. Shih, BS; Elliot Abemayor,
      structures.                                                                MD, Phd; Maie A. St. John, MD, PhD.
      Postoperative course is free of significant complications,           12.   Shaw CK, Sood S, Bradley PJ, Krishnan S. Unusual mass in
      with no discomfort reported by the patient; headaches                      the parapharyngeal space: a Warthin’s tumour. ANZ J Surg.
                                                                                 2006 Mar;76(3):193-4. PubMed PMID: 16626365. Takashi-
      and TMJ pain disappeared after surgery.                                    ma S, Sone S, Honjho Y, Horii A, Yoshida J. Warthin’s tumor
                                                                                 of the parotid gland with extension into the parapharyngeal
                                                                                 space. Eur J Radiol. 1997 May;24(3):227-9. PubMed PMID:
      References                                                                 9232394.
      1.   Dallan I, Seccia V, Muscatello L, et al. Transoral endoscopic   13.   Dimitrijevic MV, Jesic SD, Mikic AA, Arsovic NA, Tomanovic
           anatomy of the parapharyngeal space: a step-by-step logi-             NR. Para- pharyngeal space tumors: 61 case reviews. Int J
           cal approach with surgical considerations. Head Neck 2011;            Oral Maxillofac Surg 2010;39:983 was 989.
           33:557-561.                                                     14.   (Surgical managment of parapharingeal space tumours:
      2.   Excision of tumors in the parapharyngeal space using an               results of 10-year follow up 14 F Bozza, MG Vigili,1 P Rusci-
           endoscopically assisted transoral approach: a case series             to,2 A. Marzetti,1 and F Marzetti2. Surgical management of
           and literature review Zhe Chen1, Ya-Lian Chen1, Qi Yu1,               parapharyngeal space tumours: results of 10-year follow-up.
           Shui-Hong Zhou1, Yang-Yang Bao1, De-sheng Shang2 and            15.   Parapharyngeal Space Tumor: Submandibular Approach
           Ling-Xiang Ruan2.                                                     Without Mandibulotomy Kuauhyama Luna Ortiz1 • Oscar
      3.   Fakhry, Nicolas (25 July 2016). “A proposal for a level               Villa-Zepeda1 • Jose F. Carrillo1 • Ernesto Molina-Frias1 •
           for parapharyngeal extension of parotid gland”. Europe-               Antonio Go ́mez-Pedraza1.
           an Archives of Oto-Rhino-Laryngology. 273 (10):3455.            16.   Prasad SC, Piccirillo E, Chovanec M, La Melia C, De Dona-
           doi:10.1007/s00405-016-4226-8. PMID 27455864.                         to G, Sanna M. Lateral skull base approaches in the man-
      4.   Jansen JC, et al. Estimation of growth rate in patients with          agement of benign parapharyngeal space tumors. Auris
           head and neck paragangliomas influences the treatment                 Nasus Larynx. 2015;42(3):189-198.
           proposal. Cancer. 2000;88(12):2811.                             17.   Surgical anatomy of the parapharyngeal space: A multiper-
      5.   Surgical Approaches to Benign Parapharyngeal Space                    spective, quantification-based study Marco Ferrari MD1 | Al-
           Tumors-5-Year Experience Keyvan Aghazadeh1, MD; Mo-                   berto Schreiber MD, Phd1 | Davide Mattavelli MD1 | Davide
           hammadtaghi Khorsandi1,MD; Mohamamadjavad Rikhte-                     Lombardi MD1 | Vittorio Rampinelli MD1 | Francesco Dogli-
           gar1,MD; *Amirsina Sharifi1,MD; Arsalan Hashemiagh-                   etto MD, Phd2 | Luigi Fabrizio Rodella MD, Msc3 | Piero
           dam2,MD; Arezou Hashem Zade2,MD.                                      Nicolai MD1.
      6.   Case report: extranodal non-Hodgkin’s lymphoma of the           18.   Parapharyngeal Space Tumor: Submandibular Approach
           parapharyngeal space Fatma Tulin Kayhan, Naif Ozkul, De-              Without Mandibulotomy Kuauhyama Luna-Ortiz1 • Oscar
           partment of Otolaryngology, Head and Neck Surgery, Vakif              Villa-Zepeda1 • Jose F. Carrillo1 • Ernesto Molina-Frias1 •
           Gueha Hospital, Istanbul, Turkey.                                     Antonio Go ́mez-Pedraza1.
      7.   Parapharyngeal Space Pleomorphic Adenoma: A 30-Year             19.   Goodwin WJ Jr, Chandler JR (1988) Transoral excision of
           Review Abie H. Mendelsohn, MD; Sunita Bhuta, MD; Thom-                lateral parapharyngeal space tumors presenting intraorally.
           as C. Calcaterra, MD; Hubert B. Shih, BS; Elliot Abemayor,            Laryngo- scope 98:266=269.
           MD, PhD; Maie A. St. John, MD, PhD.                             20.   Eur Arch Otorhinolaryngol. 2010 May;267(5):765-72. doi:
      8.   Maheshwar AA, Kim EY, Pensak ML, et al. Roof of the para-             10.1007/s00405-009-1071-z. Epub 2009 Aug 28. Transoral
           pharyngeal space: defining its boundaries and clinical impli-         and combined transoral-transcervical approach in the sur-
           cations. Ann Otol Rhinol Laryngol. 2004;113:283 was 288.              gery of parapharyngeal tumors. Betka J1, Chovanec M, Klo-
           doi: 10.1177/000348940411300405.                                      zar J, Taudy M, Plzák J, Kodetová D, Lisý J.




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