10.14639/0392-100X-suppl.1-43-2023-10
Benign laterocervical pathologies are not without pitfalls. Many may relapse after many years and, sometimes, they cannot be predicted. The purpose of this review is to describe the surgical measures necessary for the treatment of relapses of the most common benign laterocervical masses. We searched PubMed, Embase and Cochrane Central Register of Controlled Trials databases for articles describing the treatment of the most common benign cervical disease recurrences, and summarised available evidence in this narrative review. We overviewed observations about recurrent benign mixed tumour (pleomorphic adenoma), parapharyngeal space tumours and carotid body paragangliomas, thyroglossal duct anomalies and branchial cleft anomalies. Proper surgical technique is crucial for safely and effectively managing the relapses of benign latero-cervical diseases. Radiotherapy is indicated in several cases of recurrence such as pleomorphic adenoma and unresectable paraganglioma. Long-term follow-up is of utmost importance to promptly recognise and treat recurrencies.
text/xml
1827-675X
2612
en
Marco de Vincentiis
http://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
branchial cleft
Treatment of relapses of benign latero-cervical pathology: a narrative literature review
Text.Serial.Journal
https://www.actaitalica.it/
43
Annual congress report
2023-04-21
2023-04-21
2023-04-28
2023-04-28
S76-S83
1
Marco de Vincentiis
2023/04/21
10.14639/0392-100X-suppl.1-43-2023-10
S76
Department of Sense Organs, Sapienza University of Rome, Rome, Italy
1827-675X
branchial cleft
S83
Treatment of relapses of benign latero-cervical pathology: a narrative literature review
https://www.actaitalica.it/article/download/2612/947
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ACTA OTORHINOLARYNGOLOGICA ITALICA 2023;43(SUPPL.1):S76-S83; doi: 10.14639/0392-100X-suppl.1-43-2023-10




Head and Neck section


Treatment of relapses of benign latero-cervical
pathology: a narrative literature review
Trattamento delle recidive della patologia benigna latero-cervicale:
revisione narrativa della letteratura
Piero Giuseppe Meliante, Andrea Colizza, Massimo Ralli, Antonio Minni, Diletta Angeletti, Francesca Cambria,
Marco de Vincentiis
Department of Sense Organs, Sapienza University of Rome, Rome, Italy


SUMMARY
Benign laterocervical pathologies are not without pitfalls. Many may relapse after many
years and, sometimes, they cannot be predicted. The purpose of this review is to describe
the surgical measures necessary for the treatment of relapses of the most common benign
laterocervical masses. We searched PubMed, Embase and Cochrane Central Register of
Controlled Trials databases for articles describing the treatment of the most common
benign cervical disease recurrences, and summarised available evidence in this narrative
review. We overviewed observations about recurrent benign mixed tumour (pleomorphic
adenoma), parapharyngeal space tumours and carotid body paragangliomas, thyroglos-
sal duct anomalies and branchial cleft anomalies. Proper surgical technique is crucial
for safely and effectively managing the relapses of benign latero-cervical diseases. Ra-
diotherapy is indicated in several cases of recurrence such as pleomorphic adenoma and
unresectable paraganglioma. Long-term follow-up is of utmost importance to promptly
recognise and treat recurrencies.
KEY WORDS: benign pathologies, laterocervical relapse, parotid gland tumor,
paragangliomas, thyroglossal duct, branchial cleft

RIASSUNTO
                                                                                                  Received: December 12, 2022
Le patologie benigne laterocervicali non sono prive di insidie. Molte di esse possono
                                                                                                  Accepted: January 8, 2023
andare incontro a recidiva a distanza di molti anni e, talvolta, non è possibile prevedere
se questo accadrà. Lo scopo di questa review è di descrivere gli accorgimenti chirurgici
                                                                                                  Correspondence
necessari al trattamento delle recidive delle più comuni masse benigne laterocervicali.           Andrea Colizza
Sono stati analizzati gli articoli scientifici inerenti al trattamento delle recidive delle più   Department of Sense Organs, Sapienza University
comuni patologie laterocervicali nei database internazionali PubMed, Embase e Cochra-             of Rome, Rome, Italy
ne Central Register of Controlled Trial, e sono state sintetizzate le evidenze nel presente       E-mail: andrea.colizza@uniroma1.it
manoscritto. In questa revisione narrativa sono state descritte le osservazioni riguar-
danti il trattamento delle recidive delle patologie benigne laterocervicali. In particolare,
                                                                                                    How to cite this article: Meliante PG, Colizza
sono stati inclusi il tumore misto benigno (adenoma pleomorfo), i tumori dello spazio               A, Ralli M, et al. Treatment of relapses of be-
para-faringeo e i paragangliomi carotidei, le anomalie del dotto tireoglosso e quelle               nign latero-cervical pathology: a narrative
degli archi branchiali. Una tecnica chirurgica appropriata è essenziale per gestire in              literature review. Acta Otorhinolaryngol Ital
sicurezza e con efficacia le recidive delle patologie benigne laterocervicali. La radiote-          2023;43(SUPPL.1):S76-S83.            https://doi.
rapia è indicata in alcuni casi di recidive come l’adenoma pleomorfo e il paraganglioma             org/10.14639/0392-100X-suppl.1-43-2023-10
non resecabile. Il follow-up a lungo termine è fondamentale per una precoce diagnosi e
il trattamento delle recidive.                                                                    © Società Italiana di Otorinolaringoiatria
                                                                                                  e Chirurgia Cervico-Facciale


                                                                                                                 OPEN ACCESS
PAROLE CHIAVE: patologie benigne, recidive laterocervicali, tumori della ghiandola
parotide, paragangliomi, dotto tireoglosso, archi branchiali                                      This is an open access article distributed in accordance with
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                                                                                                  Commercial-NoDerivatives 4.0 International) license. The
                                                                                                  article can be used by giving appropriate credit and mentio-
Introduction                                                                                      ning the license, but only for non-commercial purposes and
                                                                                                  only in the original version. For further information: https://
Patients presenting with laterocervical masses are usually relieved when diag-                    creativecommons.org/licenses/by-nc-nd/4.0/deed.en




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                                                                        Surgical treatment of benign latero-cervical diseases recurrencies




nosed with a benign disease. However, although they rarely     niques to be adopted in the treatment of relapses of later-
metastasise, these conditions are not without pitfalls such    ocervical benign diseases.
as recurrencies. Communication with the patient is essen-      All authors discussed evidence and the conflicts were re-
tial for a successful diagnostic-therapeutic process, since    solved by our senior author MdV. We summarise our con-
compliance during follow-up is essential to guarantee the      clusions in the present manuscript.
correct treatment and to identify relapses of the disease.
The most common benign laterocervical pathologies in-          Results
clude pleomorphic adenoma (PA), also called benign
mixed tumour, tumours of the parapharyngeal space in-          From the initial search, after removal of duplicates, 977 ar-
cluding paragangliomas of the carotid body, thyroglossal       ticles were identified for recurrent benign mixed tumour,
duct cysts and brachial cleft anomalies. Parotid gland tu-     317 for recurrent parapharyngeal space tumour, 281 for ca-
mours are 3% of all head and neck tumors and PA is the         rotid body paraganglioma, 302 for thyroglossal duct anom-
most common 1. Parapharyngeal space tumours account for        alies and 238 for branchial cleft anomalies. We screened
only 0.5% of head and neck cancers and 80% are benign 2,3.     the articles by title and abstract, and then read the full text
Salivary gland neoplasms are the most common followed          and identified 48 papers as relevant to our review.
by neurogenic ones 4. The most frequent congenital anom-
aly of the neck is the thyroglossal duct cyst, which affects   Recurrent benign mixed tumour (Pleomorphic Adenoma)
7% of the population 4. Branchial cleft anomalies are 30%      of the parotid gland
of congenital neck masses and can present as cysts, sinuses    PA accounts for 65% of benign parotid tumours 6. Ultrasound
or fistulas, and usually become symptomatic in the first few   is the first-level examination in the diagnosis of parotid tu-
years of life 4,5.                                             mours. It allows to determine the characteristics, anatomical
The purpose of this review is to analyse the surgical tech-    relationships, dimensions and acts as a guide for the execu-
niques for treatment of relapses of the most common be-        tion of a needle aspiration 7. One of the best indicators of tu-
nign laterocervical conditions.                                mour proximity to the facial nerve can be detected with MRI
                                                               or ultrasound using the retromandibular vein as a reference 7.
                                                               MRI is the technique of choice in recurrent forms of PA and
Materials and methods                                          can sometimes be useful in primary forms 8.
                                                               Psychogios et al. indicated radiological/ultrasound criteria
Search strategy                                                for choosing the type of intervention. They defined the in-
In this narrative review of the literature, we searched Pub-   dication to perform extracapsular resection of the tumour
Med, Embase and Cochrane Central Register of Controlled        for masses ≤ 2 cm in size as “ideal”, especially if they are
Trials for articles about the treatment of the benign cervi-   palpable, mobile and not in contact with the facial nerve,
cal disease recurrence. The diseases we included in our re-    except for tumours near the Stenos duct. In all other cases,
search were: pleomorphic adenoma, thyroglossal duct cyst,      superficial or partial parotidectomy is recommended. Total
branchial cleft anomalies and carotid body paraganglioma.      parotidectomy is reserved for malignant disease and some
We also hand searched through the bibliographies of arti-      benign tumours of the deep lobe 7.
cles to find relevant papers.                                  Local recurrence of benign mixed tumour of the parotid
                                                               gland may occur in an unifocal or multifocal fashion. The
Eligibility and inclusion criteria                             latter is more common in patients who have undergone su-
Prospective and retrospective studies, both randomised         perficial parotidectomy; multifocal recurrence is the most
and non-randomised, as well as literature reviews and case     common and has usually nodules involving the overlying
series, were included. Case reports and rare diseases were     skin. The histopathological features associated with an in-
excluded. We analysed articles with no time limit and ex-      creased risk of recurrence are a predominant myxoid compo-
cluded non-English papers. At least two authors for each       sition and extracapsular extension 9,10. Rupture of the capsule
topic independently searched the evidence in the literature    during surgical excision, the presence of satellite lesions or
for each disease.                                              pseudopodia, and positive resection margins are risk factors
                                                               for the development of recurrence. 11 The relapse rate with a
Data extraction                                                non-enucleation procedure is between 1% and 4%, and are
We screened the selected papers by title and abstract, and     typically observed after 7–10 years from surgery 12,13.
then considered the entire text of the selected articles to    The multinodular recurrent PA has usually microscopical
search for evidence and descriptions of the surgical tech-     seedings even if the number of nodules is limited. They



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usually involve the facial nerve, especially if it was dis-                            Rates of facial nerve dysfunction increase with each revi-
sected during primary surgery. Given the complexity of                                 sion procedure due to previous dissection making it diffi-
execution, the increased risk of facial nerve damage, the                              cult to distinguish the facial nerve from scar tissue.
need for extensive resections and, sometimes, reconstruc-                              Some techniques are reported in literature to avoid fa-
tive surgery, some authors suggest observation instead of                              cial nerve lesions during retrograde facial nerve dissec-
surgery in elderly patients 1,8.                                                       tion 8. The dissection starts from the temporal facial nerve
                                                                                       branch, which is generally the more undisturbed branch in
Treatment                                                                              the first surgery. In parotid surgery, intra-operative facial
Preservation of the facial nerve is much more complex in                               nerve monitoring (IFNM) is of great value in the treatment
surgery of recurrence of PA. This is due to the formation                              of recurrences due to the greater difficulty encountered in
of scar tissue which makes it more difficult to isolate the                            locating the nerve in scar tissue. Thanks to the use of neu-
nerve. In addition, the loss of normal planes further com-                             romonitoring, it is possible not to expose the branches of
plicates the ability to locate the nerve. For these reasons,                           the nerve, decreasing the incidence rate of permanent facial
some authors suggest identifying the nerve in the mastoid                              paralysis and shortening the recovery time of post-opera-
segment or with retrograde technique when performing re-                               tive facial nerve function. Removal of a recurrent PA can
vision surgery. It is also recommended to remove the scar                              be accomplished by reducing mechanical manipulation and
of the previous surgery “en bloc” with the neoformation 1.                             surgical trauma to the nerve 8. Some authors have observed

Table I. Characteristics of the main laterocevical benign pathologies.
    Disease           Symptoms          Relapse risk factors                             Treatment                                 Surgical pitfalls             Ref
 Pa                     Mass in the        Surgical risk factors:        Radiation therapy. Superficial or total      Complex preservation of facial            6, 9, 10,

                       parotid region      histopathological risk parotidectomy with retrograde nerve identification,            nerve                           15, 16

                                           factors: predominant    removal of the overlying skin and previous scar.
                                          myxoid composition and         Flap reconstruction may be required
                                          extracapsular extension
 MPA                       Mass in              Not known          Surgical removal of masses, total parotidectomy,         Poor prognosis                       19-22

                           cervical                                       neck dissection. Radiation therapy
                        lymph nodes,
                          metastasis
 Parapharyngeal           Cervical or       Incomplete excision      Surgical excision of the mass and eventual lymph                                         3, 25, 26, 34,

 space tumours         intraoral mass                                 nodal metastasis. TORS. Radiotherapy in case of                                          36-39, 42

                                                                     non-resectable lesion, involvement of the internal
                                                                     carotid artery and negative balloon occlusion test,
                                                                      patients with contralateral vagus or hypoglossal
                                                                                      nerve deficiency
 Carotid body          Cervical mass        Incomplete excision      Surgical excision with pre-operative embolisation                                             33

 paraganglioma                                                               and eventual adrenergic blockade
 Thyroglossal          Cervical mass        Incomplete excision                      Sistrunk procedure                     Extensive resection of the duct     4, 40-42

 duct cyst             in the midline,        usually with plan
                         infection of             removal
                          the mass
 First branchial         Swelling in        Incomplete excision                       Surgical excision.                      Excise skin and cartilage of     43, 46, 47

 cleft anomalies        pre-, infra- or                                                                                        the external auditory canal.
                       post-auricular                                                                                           Dissection from the facial
                            region                                                                                                         nerve
 Second                    Swelling        Increasing their size                      Surgical excision                       Cannulation of the tract with   4, 5, 42, 43

 branchial cleft        between the       after upper respiratory                                                             a 2-0 ore 3-0 monofilament
 anomalies                SCM and          infections that leads                                                              suture probe or the injection
                           pharynx        torticollis or dysphagia                                                                 with methylene blue
 Third and fourth        Hypoglossal        Incomplete excision                       Surgical excision                       Fourth arch anomalies often     4, 42, 43, 50

 cleft anomalies         nerve palsy                                                                                         require hemithyroidectomy to
                          in case of                                                                                          completely excise the mass
                         infection or                                                                                        and, sometimes, resection of
                         lower neck                                                                                         thyroid cartilage to expose the
                         swallowing                                                                                                   pyriform sinus
PA: pleomorphic adenoma or benign mixed tumour; MPA: malignant pleomorphic adenoma; SCM: sternocleidomastoid muscle; TORS: transoral robotic surgery.




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                                                                            Surgical treatment of benign latero-cervical diseases recurrencies




that its use reduces the operating time, the recovery time in      ed pyramid, the tip is located at the greater horn of the
case of temporary dysfunction of the cranial nerve septum          hyoid bone and the floor at the skull base (petrous por-
and the incidence of permanent dysfunction 14.                     tion of the temporal bone and sphenoid bone). Medially
In case of cutaneous invasion, it is necessary to remove, to-      there is the buccopharyngeal fascia, laterally the medial
gether with the nodules and any parotid tissue still present,      pterygoid muscle, the mandible, the posterior belly of the
the involved preauricular and cervical skin. In such cases,        digastric muscle and the retromandibular portion of the
flap reconstruction is required 15 (Tab. I).                       parotid gland; anteriorly the pterygomandibular raphe;
Surgical treatment for PA recurrence can be performed              posteriorly the carotid sheath 25. Parapharyngeal space
with conventional excision or parotidectomy, depending             tumours constitute the 0.5% of all head and neck neo-
on the specific case. Usually, this surgery requires a high        plasms 2. Eighty percent are benign and surgical excision
degree of expertise and more extended surgical approach;           is the standard treatment in most cases 3,25,26. The most
superficial or total parotidectomy are the techniques of           common are salivary gland tumours, accounting for 40-
choice in most cases 8,16. Reoperation complicates the             50% of cases 26.
preservation of the facial nerve, which must always be             Neurogenic tumours represent about 40% of parapharyn-
sought by the surgeon. In case of nerve damage during              geal space neoplasms and 95% are benign. There are three
the excision of the recurrent PA it is advisable to perform        main subtypes, paragangliomas, schwannomas and neu-
an immediate repair using a nerve graft 17. Adjuvant radio-        rofibromas. The most common presenting symptom is the
therapy may reduce the recurrence rate in recurrent PA 18.         growth of a cervical or intraoral mass (50% and 47%, re-
Witt et al. observed that radiation therapy has better out-        spectively) 3. During treatment of parapharyngeal space tu-
comes in controlling recurrent pleomorphic adenoma com-            mours, it must always be considered that 40% of sporadic
pared to surgical excision; however, treatment must be             and 80% of familial cases have multiple tumours, and that
individualised depending on the patient’s conditions and           10% of sporadic and 30% of familial neurogenic tumours
disease 8.                                                         are bilateral. Therefore, a careful analysis of MRI and CT
                                                                   images is also necessary in the contralateral area to that in
Malignant pleomorphic adenoma
                                                                   which the symptoms occur to identify small, unrecognised
PA can rarely metastasise even without malignant histologi-
                                                                   neoformations 3,25.
cal degeneration; it is called malignant pleomorphic adenoma
(MPA) and parotid gland is the most affected site. According       Treatment
to the 2005 classification of the WHO, there are three distinct    The main treatment of the primary tumour or relapses is
clinicopathologic types for malignant pleomorphic adenoma:         surgical excision. Surgical management should also con-
carcinoma ex pleomorphic adenoma (CXPA), carcinosarco-             sider the presence of lymph node metastasis. It is recom-
ma, and metastasising mixed tumour. MPA is a condition with        mended to perform a neck dissection involving at least
histological characteristics of PA with the capacity to generate   levels II to IV during resection of these tumours 3,25,26.
local recurrencies and distant metastases (both to local lymph     Relapse risk is mostly related to incomplete excision or
nodes and other organs) 19.Most occur after one or more surgi-     tumour dissemination during surgery, and accurate fol-
cal procedures and histological examination is not predictive      low-up is of utmost importance for early diagnosis and
regarding the ability to metastasise. The hypothesised aetiol-     treatment of relapses 8.
ogy is the cell spreading during surgery 19,20.                    Paragangliomas can also have malignant forms with
Nearly 30% of MPA cases are localised in the lymph nodes,          lymph node metastases, and, as with pleomorphic adeno-
and for this reason it is recommended to also perform neck         ma, their malignant behaviour is not histologically pre-
dissection. MPA relapse treatment should involve total pa-         dictable 27.
rotidectomy with conservation of facial nerve when it is not       Along with traditional CT and MRI, it may be useful in
infiltrated 19-22.                                                 cases suspect of malignancy, once a lesion of vascular ori-
Radiotherapy is considered as a complementary treatment            gin has been excluded, to perform a fine needle aspiration.
in recurrent disease 19,22,23 (Tab. I).                            The approach can be both pre-oral and transcervical. The
Despite the histological features of benignity, it has poor        result is useful to characterise the lesion in the treatment
prognosis (50% 5-year survival rate); therefore, it is classi-     of both primary neoplasms and relapses 28,29.
fied as a malignant tumour by the WHO 24.                          To avoid recurrence in case of incomplete resection, adju-
                                                                   vant therapy can be considered 25.
Parapharyngeal space tumours                                       In case of uncomplete paraganglioma resection, radiation
The parapharyngeal space shape resembles as invert-                therapy is a valid option. Low radiation doses are suf-



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P.G. Meliante et al.




ficient to stop tumour growth with a low incidence and           crine neoplasms, they have receptors for somatostatin and
severity of adverse events. However, in most cases low           have intense indium-111 octreotide uptake 33.
radiation doses do not eradicate the mass 8,30-32.               The treatment for carotid body tumour paragangliomas
Less than 10% of paragangliomas are malignant; how-              is surgical resection, and it is prudent to perform a pre-
ever, as in the case of pleomorphic adenomas, their ma-          operative embolisation and consider adrenergic blockade.
lignancy cannot be predicted on the basis of histological        After complete surgical resection, recurrence and metas-
features 24,33. It is necessary to carry out accurate and pro-   tasis may occur years later. Surgery remains the treatment
longed follow up.                                                of choice in most cases since chemo- and radiotherapy do
In case of recurrent disease for which neck dissection was       not have significant benefits 33 (Tab. I).
not performed in the previous surgical treatment, it is rec-
ommended that the patient undergo the procedure.                 Thyroglossal duct anomalies
The criteria for ineligibility for surgery are the same as for   Thyroglossal duct cyst is the most common congenital anom-
primary forms such as unresectable lesion, involvement of        aly of the neck, and occurs in 7% of the population 4. Its origin
the internal carotid artery and negative balloon occlusion       is from epithelial remnants of the thyroglossal duct, a divertic-
test, or contralateral vague or hypoglossal nerve deficiency.    ulum located at the base of the tongue that displaces caudally
In these cases, the therapeutic choice must lean towards ra-     to the lower neck. This duct usually obliterates at the eighth
diotherapy 30.                                                   week of gestation, and its failure results in thyroglossal duct
The selection of the appropriate surgical approach is simi-      cyst 40. The most common clinical presentation is a neck mass
lar to the one used for primary lesions. The most widely         in the midline which sometimes gets infected and treatment is
used approach is the trans-cervical one. However, cervi-         surgical excision using the Sistrunk procedure 41,42.
cal-parotid, mandibular split, trans-cervical trans-mastoid      Once clinical suspicion has been placed, in the primary di-
and infratemporal approaches can also be used depending          agnosis it is recommended, together with physical exami-
on tumour localisation and size 34. These techniques can         nation, to perform ultrasound and evaluation of the thyroid
also be combined. It is sometimes necessary to perform           stimulating hormone (TSH). If median ectopic thyroid is
a mandibulotomy for better exposure 35. Intra-operative          suspected, usually in the case of a solid mass or hypothy-
monitoring of the facial nerve can also be useful for pre-       roidism, it is advisable to perform a thyroid scan before
serving facial motility 36.                                      removal 43. Although some authors assert that thyroid scin-
TORS can be used in patients with good oropharyngeal             tigraphy should be performed in all patients due to the risk
exposure and neoplasm with a defined cleavage plane              of ectopic thyroid 42,43, MRI or CT are recommended only
from neurovascular bundle. This technique is associated          in complicated cases and relapsing forms.
with increase in capsule disruption percentage, but no in-       A relapse can be suspected, especially in patients who un-
crease in the recurrence rate 37-39. TORS has an advantage       derwent plain excision instead of a Sistrunk procedure,
in aesthetic terms. The blemish produced by a cervicoto-         which has a significantly lower recurrence rate (5.3% vs
my is often poorly accepted by patients, especially if they      55.6%). Post-operative infection is also associated with re-
are young. Being able to propose a safe treatment, which         currence 40. Relapses can be removed surgically with ex-
does not increase the risk of relapse and does not under-        tensive application of the Sistrunk technique. On occasion,
mine the patient’s quality of life and aesthetics, represents    trans-oral removal of the cyst is also useful (e.g., tongue) 44.
                                                                 Thyroglossal duct cyst relapses can occur in the area be-
a great advantage in terms of compliance and acceptance
                                                                 tween the blind foramen of the tongue and the thyroid
of the intervention.
                                                                 gland. The most effective technique to avoid thyroglos-
Surgery is not the treatment of choice in case of patients
                                                                 sal duct cyst recurrence is to choose the correct surgical
that fail balloon occlusion, the elderly, or unresectable tu-
                                                                 technique, the Sistrunk procedure. Head and neck surgeons
mors that require sacrifice of cranial nerves. In such cases,
                                                                 should avoid plain excision because of the high risk for re-
the available options include watchful waiting and radia-
                                                                 lapses. Sistrunk technique, with hyoid bone body excision,
tion therapy 25 (Tab. I).
                                                                 is the key to avoid relapses and treat them 40. An accurate
Carotid body paragangliomas                                      intra-operative exploration of the thyroglossal duct cyst
Carotid body tumour paragangliomas are vascular lesions          tract reduces the risk of leaving residues of the duct.
that splay apart internal and external carotid arteries with-    OK-432 (Picibanil) injection after cyst fluid aspiration
out narrowing them. They usually absorb a large amounts          showed encouraging results in a paediatric population with
of contrast medium and have a “salt and pepper” appear-          recurrent thyroglossal duct cyst with no need of surgery or
ance on T2 weighted images. Since they are neuroendo-            anaesthesia (either general or local) 45 (Tab. I).



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                                                                          Surgical treatment of benign latero-cervical diseases recurrencies




Branchial cleft anomalies                                        ber of procedures necessary to achieve complete resection
Branchial cleft anomalies represent 30% of congenital neck       are 2.4 per patient 47. In case of recurrence, it is therefore
masses 4. By the end of the fourth gestation week there are      necessary to carefully explore the patient’s ear both clini-
four pairs of arches and two rudimental arches separated by      cally and radiologically.
clefts externally and pouches internally. These latter gradu-    In particular, it is important to subject the patient to MRI,
ally obliterate, and their incomplete obliteration leads to      CT, and otomicroscopy before surgery 48.
branchial anomalies 43. They can present as cysts, sinuses,      In case of recurrent disease, it is fundamental to perform
or fistulae. Cysts have no external opening, sinuses have        a radicalisation surgery by extending the resection to the
an external opening, and fistulae involve a communication        external and middle ear in case of extension to those areas
with the pharynx resulting in a pharyngocutaneous fistula 4.     (Tab. I). Each repeated surgery increases the risk of facial
Diagnosis of branchial arch anomalies includes history           nerve injury due to fibrosis 5,47.
and physical examination accompanied by endoscopy to             Second cleft anomalies
rule out pharyngeal communication. For the same reason,          Second cleft anomalies represent 95% of all branchial cleft
the piriform sinus and tonsillar cough should be carefully       anomalies 4. The second arch evolves in hyoid bone and
examined during this procedure. Once the lesion has been         structures of the adjacent area. The anomalies are classified
identified, fine needle aspiration is recommended in adults      in four categories. Type I are anterior to the sternocleid-
to exclude laterocervical metastases of cervix-cephalic          omastoid muscle (SCM) and not contacting the carotid
neoplasms, while it is not recommended to perform this           sheath. Type II lesions are deep to the SCM and anterior or
procedure in children.                                           posterior to the carotid sheath, and are the most common
The radiological investigation of choice is CT, but ultra-       second cleft anomalies. Type III lesions are adjacent to the
sound and MRI can also be helpful in some cases 43.              pharynx and pass between the external and internal carotid
First arch anomalies                                             artery. Type IV lesions are between the carotid sheath and
First arch anomalies are only 1% of all branchial cleft          the pharynx, close to the tonsillar fossa.
anomalies 5. The first brachial arch is also called the man-     Second cleft anomalies are diagnosed after increasing their
dibular arch and forms the maxillary process of the upper        size after upper respiratory infections that leads to torticol-
jaw and part of the inner ear. The first cleft and pouch form    lis or dysphagia 4,42,43.
the external auditory canal, eustachian tube and middle ear,     In case of recurrence, it is essential to explore the fistula
and mastoid air cells. Consequently, first cleft anomalies       tract and excise it. Several techniques have been proposed
involve these structures. Consequently, these structures         to make this procedure easier, such as cannulation of the
must be explored both clinically and radiologically before       tract with a 2-0 or 3-0 monofilament suture probe or injec-
proceeding to surgery. They can be located above, between        tion with methylene blue. Spinal accessory and the hypo-
or below the facial nerve and are classified as Type I or II.    glossal and vagus nerve are at risk of injury, in particular in
Type I lesions course lateral to the facial nerve, and mani-     surgery of relapses because of scar. It can also be useful to
fest as swelling near the ear. Type II lesions pass medial to    explore the tract from the tonsillar fossa using a finger to
the facial nerve, and can present as a preauricular, infra- o    better expose it 5,43 (Tab I).
postauricular swelling anterior to the sternocleidomastoid       Third and fourth cleft anomalies
muscle 4,5,43. It has been observed that fistulas with respect   Third and fourth branchial pouches form the pharynx below
to the sinuses run more frequently in depth than nevus and       the hyoid bone. Sinuses and fistulae deriving from these
that young people have more frequent lesions deep to the         structures are rare and usually enter the pyriform sinus. The
seventh cranic nerve. Furthermore, open lesions in the ex-       fourth arch anomalies on the right-side loop around the sub-
ternal auditory meatus are usually superficial to the nerve.     clavian artery and enter the pharynx at the pyriform apex of
In any case, it is advisable to always locate the main trunk     cervical oesophagus, On the left, they loop around the aor-
of the facial nerve early during surgery 46.                     tic arch and ascend into the neck to enter the pyriform apex
To avoid and treat a recurrence of first arch anomalies, it      of the cervical oesophagus 4,42,43. The clinical manifestation
is necessary to excise the skin and cartilage of the exter-      of the third arch cyst causes hypoglossal nerve palsy in case
nal auditory canal involved by the mass. If the tract goes       of infection or lower neck swallowing.
medial to the tympanic membrane, a second surgical pro-          For the diagnosis of anomalies of the second and third
cedure may be necessary to remove it. In the latter case,        branchial arch, in addition to CT and sometimes ultrasound
the tract lies deep to the facial nerve or can split around      and MRI, a barium oesophagogram can be useful with a
it 43,46,47. Disease relapse is common, and the average num-     sensitivity of up to 80% 49. For anomalies of the fourth arch,



                                                                                                                                      S81
P.G. Meliante et al.




together with CT, endoscopic examination must be per-               2
                                                                         Maheshwar AA, Kim EY, Pensak ML, et al. Roof of the para-
                                                                         pharyngeal space: defining its boundaries and clinical implica-
formed with particular attention to the piriform sinus with              tions. Ann Otol Rhinol Laryngol 2004;113:283-288. https://doi.
which the fistula could communicate 50.                                  org/10.1177/000348940411300405
The surgical treatment is the excision of the mass, usually         3
                                                                         Riffat F, Dwivedi RC, Palme C, et al. A systematic review of 1143
with a cervicotomy approach. Fourth arch anomalies often                 parapharyngeal space tumors reported over 20 years. Oral Oncol
                                                                         2014;50:421-430. https://doi.org/10.1016/j.oraloncology.2014.02.007
require hemithyroidectomy to completely excise the mass             4
                                                                         Enepekides DJ. Management of congenital anomalies of the neck. Fa-
and, sometimes, resection of thyroid cartilage to expose the             cial Plast Surg Clin North Am 2001;9:131-145.
pyriform sinus 50 (Tab. I).                                         5
                                                                         Waldhausen JHT. Branchial cleft and arch anomalies in children.
                                                                         Semin Pediatr Surg 2006;15:64-69. https://doi.org/10.1053/j.
                                                                         sempedsurg.2006.02.002
Conclusions                                                         6
                                                                         Spiro RH. Salivary neoplasms: overview of a 35-year experience
                                                                         with 2,807 patients. Head Neck Surg 1986;8:177-184. https://doi.
There is increasing evidence in the scientific literature of             org/10.1002/hed.2890080309
recurrence of benign latero-cervical pathologies even more          7
                                                                         Psychogios G, Rueger H, Jering M, et al. Ultrasound can help to in-
than 10 years from the first excision. Communication with                directly predict contact of parotid tumors to the facial nerve, correct
the patient is essential to obtain the necessary compliance              intraglandular localization, and appropriate surgical technique. Head
                                                                         Neck 2019;41:3211-3218. https://doi.org/10.1002/hed.25811
for long-term follow-up, which is of utmost importance to           8
                                                                         Witt RL, Eisele DW, Morton RP, et al. Etiology and management of re-
promptly recognise and treat recurrencies.                               current parotid pleomorphic adenoma. Laryngoscope 2015;125:888-
Surgical excision of recurrencies should be performed by                 893. https://doi.org/10.1002/lary.24964
experienced surgeons because they usually require high              9
                                                                         Batsakis JG. Recurrent mixed tumor. Ann Otol Rhinol Laryngol
                                                                         1986;95:543-544. https://doi.org/10.1177/000348948609500520
expertise, and need some precautions to avoid further re-           10
                                                                         Zbären P, Tschumi I, Nuyens M, et al. Recurrent pleomorphic ad-
interventions. Radiation therapy has shown to be effective               enoma of the parotid gland. Am J Surg 2005;189:203-207. https://doi.
in the treatment of recurrent pleomorphic adenomas and to                org/10.1016/j.amjsurg.2004.11.008
reduce the growth of unresectable paraganglioma.                    11
                                                                         Krolls SO, Boyers RC. Mixed tumors of salivary glands.
                                                                         Long-term follow-up. Cancer 1972;30:276-281. https://doi.
                                                                         org/10.1002/1097-0142(197207)30:1276
Conflict of interest statement                                      12
                                                                         Laccourreye H, Laccourreye O, Cauchois R, et al. Total con-
The authors declare no conflict of interest.                             servative parotidectomy for primary benign pleomorphic ad-
                                                                         enoma of the parotid gland: a 25-year experience with 229
                                                                         patients.     Laryngoscope       1994;104:1487-1494.        https://doi.
Funding                                                                  org/10.1288/00005537-199412000-00011
This research did not receive any specific grant from fund-         13
                                                                         Niparko JK, Beauchamp ML, Krause CJ, et al. Surgical treatment of
ing agencies in the public, commercial, or not-for-profit                recurrent pleomorphic adenoma of the parotid gland. Arch Otolaryn-
sectors.                                                                 gol Head Neck Surg 1986;112:1180-1184. https://doi.org/10.1001/
                                                                         archotol.1986.03780110056007
                                                                    14
                                                                         Makeieff M, Venail F, Cartier C, et al. Continuous facial nerve
Author contributions                                                     monitoring during pleomorphic adenoma recurrence surgery. La-
PGM: writing original draft, supervision; AC: writing                    ryngoscope       2005;115:1310-1314.       https://doi.org/10.1097/01.
original draft and critical revision of the manuscript; AM:              MLG.0000166697.48868.8C
                                                                    15
                                                                         Shah JP, Patel SGH, Singh B. Jatin Shah’s head and neck surgery and
supervision; DA: literature review; FC: literature review;               oncology. Fifth edition. Philadelphia: Elsevier; 2019.
MR: critical revision of the manuscript; MdV: supervision.          16
                                                                         Psychogios G, Bohr C, Constantinidis J, et al. Review of surgical
                                                                         techniques and guide for decision making in the treatment of benign
Ethical consideration                                                    parotid tumors. Eur Arch Otorhinolaryngol 2021;278:15-29. https://
                                                                         doi.org/10.1007/s00405-020-06250-x
Not applicable.                                                     17
                                                                         Work WP, Batsakis JG, Bailey DG. Recurrent benign mixed tumor
                                                                         and the facial nerve. Arch Otolaryngol 1976;102:15-19. https://doi.
Availability of data and materials                                       org/10.1001/archotol.1976.00780060061006
The datasets used and/or analysed during the current study          18
                                                                         Mc Loughlin L, Gillanders SL, Smith S, et al The role of adjuvant
are available from the corresponding author on reasonable                radiotherapy in management of recurrent pleomorphic adenoma of
                                                                         the parotid gland: a systematic review. Eur Arch Otorhinolaryngol
request.                                                                 2019;276:283-295. https://doi.org/10.1007/s00405-018-5205-z
                                                                    19
                                                                         Soteldo J, Aranaga N. Metastasizing pleomorphic adenoma of the
                                                                         parotid gland. Ecancermedicalscience 2017;11:758. https://doi.
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