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29 March 2024: Articles  Germany

Diagnostic Challenges and Imaging Considerations for Intraparotid Facial Nerve Schwannoma: A Case Report and Literature Review

Challenging differential diagnosis, Diagnostic / therapeutic accidents, Unusual setting of medical care, Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)

Jeton Luzha1ABDEF*, Markus Kopp2BCD, Marco Wiesmüller2BCD, Lava Taha3BCD, Robin Rupp ORCID logo3BCD, Konstantinos Mantsopoulos3BCD, Michael Koch3BCD, Heinrich Iro3BCDE, Matti Sievert3ABCDEF

DOI: 10.12659/AJCR.942870

Am J Case Rep 2024; 25:e942870

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Abstract

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BACKGROUND: A mass in the parotid gland usually indicates parotid gland neoplasia. Warthin tumors or pleomorphic adenomas are common differential diagnoses. Less frequently, other differential diagnoses and sites of origin are considered. Schwannomas are rare, benign tumors in the head and neck region. Even more rarely, these tumors occur in the intraparotid course of the facial nerve. In the following, we report about 2 patients in whom a mass in the right parotid gland was found incidentally during magnetic resonance imaging (MRI).

CASE REPORT: We reviewed data from the literature on intraparotid facial nerve schwannomas (IPFNS) and compared them with those from our cases. The focus was on data such as clinical history, clinical symptoms, electroneurography, and various imaging modalities, such as ultrasonography and MRI combined with diffusion-weighted imaging.

CONCLUSIONS: It is challenging to distinguish facial nerve schwannomas from other neoplasms. Patient’s history, clinical symptoms, MRI examination with diffusion-weighted imaging, and high-resolution ultrasound imaging are decisive factors for diagnosis and should be performed when IPFNS is suspected. Diagnosis and therapy for IPFNS remain challenging. A wait-and-scan approach could be an option for patients with small tumors and good facial nerve function. On the other hand, patients with advanced tumors associated with limited facial nerve function can benefit from surgical approaches or stereotactic radiosurgery.

Keywords: Nerve Sheath Neoplasms, Parotid Gland, Facial Nerve Diseases, Facial Paralysis

Introduction

A rare site of origin for schwannomas is the parotid gland. Schwannomas arise from the Schwann cells of myelinated cranial or peripheric nerves. They are benign encapsulated tumors originating from the neuroectoderm [1,2]. Usually, head and neck schwannomas arise in the vagus nerve, sympathetic trunk, or vestibular nerve. Facial nerve schwannomas (FNS) are rare and uncommon neoplasms of the seventh cranial nerve. Most FNS arise in the skull base. Extratemporal growth is described in only 9% of cases [3,4]. There is a strong association between FNS and neurofibromatosis types 1 and 2 [2].

The management of FNS is exceptionally complicated because the diagnosis is usually only made intraoperatively. In addition, resection of the tumor could lead to facial nerve palsy, with significant esthetic consequences. On the other hand, if surgical treatment is delayed, tumor progression can lead to severe but rare complications, such as the development of hydrocephalus [5]. Therefore, the decision as to when therapy should be initiated and in what form it should be carried out should be carefully considered [6].

Case Reports

The first case was a 28-year-old male patient with a mass in the right parotid gland incidentally diagnosed during epilepsy imaging. The patient presented to our clinic for a more detailed evaluation. The facial nerve function was normal. However, magnetic resonance imaging (MRI) showed a cystic mass along the course of the facial nerve in the mastoid segment and right parotid gland, with minimal contrast affinity (Figures 1, 2), which was not suspicious for malignancy. We immediately examined the right parotid gland, which revealed a large, sharply circumscribed, lobulated, partially septated mass with dorsal acoustic enhancement without internal perfusion (Figure 3). The subsequent ultrasound and MRI examinations showed a constant size and aspect. Electromyographic measurement of the facial nerve showed unremarkable findings (House-Brackmann grade I).

The second case involved a 34-year-old female patient who noticed a progressive mass on the right mandibular angle for several weeks. Neurological deficits and other symptoms were also denied. To further evaluate the patient’s concerns, we performed ultrasonography of the head and neck region (Figure 4), an MRI scan of the head (Figures 5, 6), and an electromyographic measurement of the facial nerve. Surprisingly, the findings were similar to those of the first patient.

The various therapeutic options were carefully discussed with the patients. Without clinical symptoms and size constancy, both patients opted for a wait-and-scan approach.

Discussion

Schwannomas (also called neurinomas) are benign tumors that originate from myelin-forming cells and are characterized by generally slow growth. The symptoms depend on the growth behavior of the location and size of the tumor and on the function of the corresponding nerve. Schwannomas can occur anywhere in the body where peripheral nerves are located, but they are most common in the head, face, and extremities. Vestibular schwannoma is the most common, accounting for over 90% of cases. In contrast, non-vestibular cranial nerve schwannomas are rare lesions and account for only 5% to 10% of cases. These include, in descending order, trigeminal nerve schwannomas, lower cervical nerve schwannomas, FNS, and, in very few cases, oculomotor nerve schwannomas. FNS can arise anywhere along the course of the facial nerve and have been classified according to their sites of origin: the cerebellopontine angle and the labyrinthine, geniculate, tym-panic, mastoid, and parotid segment. In particular, intraparotid facial nerve schwannomas (IPFNS) are extremely scarce and account for approximately 14.6% of FNS [6,7]. Due to their extracranial course, they are often mistaken for more conventional tumors, such as pleomorphic adenomas [8,9]. Since there are often no clinical symptoms, like in our patients, preoperative diagnosis is complex, and there are no standard diagnostic criteria. Facial nerve paresis could occur in both benign and malignant parotid tumors. According to Marchioni et al, 72 of 79 patients (91.1%) with a diagnosis of IPFNS had normal facial nerve function. The probability of facial nerve palsy in IPFNS is approximately 19% [2]. This is due to the absence of nerve compression in a bony canal in a solely extratemporal tumor location.

Ultrasonography is a standard imaging modality in diagnosing parotid gland tumors. However, as described in case reports, IPFNS present as hypoechogenic, with sharply marked lesions with dorsal acoustic enhancement, and therefore, can be mis-diagnosed as neoplasms of salivary gland tissue. The presented cases illustrate that extended imaging should be considered, especially in space-occupying lesions in broad contact with the mastoid process. The criterion standard of imaging modality is an MRI scan [2]. In concordance with our case report, imaging features suggestive of facial nerve schwannoma include well-circumscribed margins, displacement of adjacent structures, cystic formation, and heterogeneous relatively strong signal intensity, especially on T2-weighted MR images [10]. Due to the rare occurrence of IPFNS and clinically and radiologically uncharacteristic features, preoperative diagnosis is challenging. Marchioni et al could not identify defining radiological imaging features after reviewing 79 cases of patients with IPFNS [9]. In the association of the tumor to the fallopian canal, the diagnosis of IPFNS by MRI is more likely. A computed tomography scan can strengthen this finding to detect bony remodeling in the fallopian canal as an indication of growth tendency [11,12]. It is challenging to distinguish facial nerve sheath tumors from other neoplasms, such as vestibular nerve schwannomas or meningiomas located in the cerebellopontine angle or petrous bone. A hint that can indicate IPFNS is the involvement of the geniculate ganglion or the various segments of the facial nerve, such as its labyrinthine, tympanic, or mastoid course [12].

Another diagnostic method is fine needle aspiration cytology (FNAC). FNAC performed preoperatively often leads to incorrect diagnoses and inconclusive results [8,9]. Other studies showed that IPFNS could be detected preoperatively by FNAC in only 15 of 67 patients (22%). A recent case report showed that in only 31.6% of cases, FNS could be detected by FNA [10]. According to many authors, there are better methods than FNAC [9,10]. A more reliable way to diagnose IPFNS is an intraoperative diagnosis through surgical excision and histopathologic evaluation [9,10]. Adequate treatment of IPFNS is challenging for surgeons. Surgical diagnosis confirmation carries many risks for the patient regarding aesthetics and quality of life. Patient age, facial nerve function, growth progression, and tumor location are important factors that should be considered in the decision-making process [11]. Numerous authors consider a House-Brackmann grade of IV or worse as an indication for surgery [11,13]. Other important surgical indications are complications such as hearing loss, development of hydrocephalus, or brain stem compression in cases of intratemporal manifestation [14]. In cases with a House-Brackmann grade of III or better and uncritical tumor location, in which no other symptoms are present, and there is a strong suspicion of IPFNS radiologically, we agree with other authors that conservative techniques such as wait and scan is the method of choice [1,13,14]. When considering observation as a method of choice, it is essential to know that facial schwannomas have a reported annual growth rate of 2.0 mm per year. Most schwannomas remain constant in size for years without progression. Doshi et al showed that of 14 patients with a House-Brackmann grade III or better, 12 remained without clinical or radiographic progression after 13 years of observation [11]. A study by McMonagle et al strengthens this conclusion. The authors demonstrated that 20 of 53 patients with facial nerve schwannoma were followed up for 8 years and clinically showed good facial nerve function [11]. Thus, as in our case report, a wait-and-scan approach could be a good option for selected patients.

Stereotactic radiosurgery is another established treatment option in selected patients [14–16]. Multiple studies have shown that stereotactic radiosurgery provides reasonable short-term tumor control rates. In a meta-analysis of 45 patients, tumor control was achieved in 93% of cases after 2 years. Sheehan et al showed, after reviewing 42 cases, that 90% of the patients were in progression-free survival after 5 years. The mean marginal dose of radiation was 12.5 Gray. These findings agreed with the study of Hasegawa et al, in which after 5 years, the progression-free survival rate was 92% [11,16]. The probability of post-radiogenic, new-onset worsening of facial nerve function after doses less than 14 Gray was less than 5% [11]. However, it is essential to mention that radiosurgery has 2 significant drawbacks. First, the possibility of complete tumor removal eliminates full histologic evaluation. On the other hand, surgical therapy offers the possibility of patient-specific facial nerve rehabilitation. Second, surgical therapy following radiotherapy is associated with increased complications. Numerous case reports elucidate IPFNS, delineating diverse methodologies and offering intriguing deductions. For example, De Ceulaer et al documented a 68-year-old patient with pre-existing impairment of the facial nerve’s frontal branch. A diagnosis of IPFNS was established based on medical history, symptoms, and MRI examination. Intraoperative frozen section examination confirmed the diagnosis, leading to surgical resection with nerve reconstruction, using a nerve graft. Postoperatively, facial nerve function partially recovered, reaching House-Brackmann grade II [17]. The 2018 case report of Simone et al presented 2 instances of IPFNS. In the first case, a parotidectomy was performed, involving tumor stripping from the nerve. The second case underwent nerve resection with complex reconstruction using hypoglossal-facial neurorraphy. Similar to our findings, the authors emphasized the challenge of preoperative diagnosis but stressed its crucial nature. Postoperative paralysis was expected, and FNAC often lacked diagnostic accuracy. A 24-month follow-up revealed House-Brackmann grade II in temporal-ocular and grade III in facial-cervical branches for the first case, while the second case showed House-Brackmann grade III [18]. Marchioni et al reported on a 74-year-old patient presenting with facial nerve palsy (House-Brackmann grade II). During parotidectomy, the intraoperative sacrifice of the facial nerve occurred and was subsequently reconstructed using a great auricular nerve graft. Immediate postoperative assessment revealed complete facial nerve palsy. A 6-month followup indicated House-Brackmann grade V. The authors concluded that tumor extent and involvement of different facial nerve branches are crucial for prognosis and treatment planning [19].

In our case presentation of IPFNS, we emphasize unique diagnostic challenges and the pivotal role of trans-cervical ultrasound imaging. These 2 cases highlight the complexity of IPFNS diagnosis, particularly in the absence of standard diagnostic criteria, and the potential for misdiagnosis as more conventional salivary gland tumors. Our findings underscore the necessity of extended imaging for accurate diagnosis and management of IPFNS, especially when lesions are in close contact with the mastoid process. This approach is crucial for distinguishing IPFNS from other neoplasms, a challenge accentuated by their rare occurrence and non-characteristic clinical and radiological features.

Conclusions

Diagnosis and therapy for IPFNS remain challenging. When deciding on therapy, it must be considered that significant consequences for the patient and his quality of life can result. Therefore, whether and when surgery is indicated must be weighed. A vital aspect highlighted is the emphasis on ultrasound as the preferred imaging modality for masses in the parotid gland. The differential diagnosis of IPFNS is particularly crucial. Examiners should be familiar with the sonographic findings of these pathologies to initiate further diagnostic imaging when IPFNS is suspected. Our report underscores this point by demonstrating that trans-cervical ultrasound is critical for initial assessment and subsequent diagnostic procedures. The treatment decision is based on facial nerve function, patient age, tumor location and size, and last but not least, the patient’s preferences. An observational wait-and-scan approach is an option for patients with small tumors and normal facial function. On the other hand, surgical approaches or stereo-tactic radiosurgery can be vital for patients with already limited facial nerve function.

Figures

Magnetic resonance imaging scan of the head. (A, B) T2-weighted very hyperintense, homogenous, almost cystic lesion, with (C, D) only minor peripheral and partly septal contrast-enhancement. (E, F) The lesions extend into the right facial canal’s widened mastoid segment and (B, D) present with a mass in the extratemporal segment. (A, B) T2-weighted fat-suppressed inversion recovery sequence in coronal view. (C–E) T1-weighted fat-suppressed dixon sequence with contrast-enhancement in (C, D) coronal and (E) axial view. (F) High-resolution T2-weighted SPACE fat-suppressed sequence in axial view.Figure 1.. Magnetic resonance imaging scan of the head. (A, B) T2-weighted very hyperintense, homogenous, almost cystic lesion, with (C, D) only minor peripheral and partly septal contrast-enhancement. (E, F) The lesions extend into the right facial canal’s widened mastoid segment and (B, D) present with a mass in the extratemporal segment. (A, B) T2-weighted fat-suppressed inversion recovery sequence in coronal view. (C–E) T1-weighted fat-suppressed dixon sequence with contrast-enhancement in (C, D) coronal and (E) axial view. (F) High-resolution T2-weighted SPACE fat-suppressed sequence in axial view. Apparent diffusion coefficient (ADC). High ADC supports the diagnosis of a facial schwannoma (average ADC value: 2.1×10–3 mm2/s).Figure 2.. Apparent diffusion coefficient (ADC). High ADC supports the diagnosis of a facial schwannoma (average ADC value: 2.1×10–3 mm2/s). Ultrasound image of the right parotid gland in (A) transversal and (B) sagittal plane. Both images show an intraparenchymal, sharply circumscribed, anechoic, homogeneous, lobulated mass with dorsal sound enhancement extending from the mastoid. The mass measures 13.9×14.8×17.4 mm. GLP righ – parotid gland; UK – mandible; MM – musculus masseter.Figure 3.. Ultrasound image of the right parotid gland in (A) transversal and (B) sagittal plane. Both images show an intraparenchymal, sharply circumscribed, anechoic, homogeneous, lobulated mass with dorsal sound enhancement extending from the mastoid. The mass measures 13.9×14.8×17.4 mm. GLP righ – parotid gland; UK – mandible; MM – musculus masseter. Ultrasound findings in the (A) transversal and (B) sagittal plane. Ultrasound images reveal a mass of similar morphologic ultrasound criteria as previously described: intraparenchymal in the parotid gland showing a sharply circumscribed, anechoic, dorsal ultrasound enhancement, measuring 19.5×18.3×26.5 mm.Figure 4.. Ultrasound findings in the (A) transversal and (B) sagittal plane. Ultrasound images reveal a mass of similar morphologic ultrasound criteria as previously described: intraparenchymal in the parotid gland showing a sharply circumscribed, anechoic, dorsal ultrasound enhancement, measuring 19.5×18.3×26.5 mm. Magnetic resonance imaging scan of the head. (D) T2-weighted moderate hyperintense, homogenous lesion with (A–C) only minor peripheral and part central contrast enhancement. (A–D) The lesions again extend into the widened mastoid segment of the right facial canal and present with a mass in the extratemporal segment. (A–C) T1-weighted contrast-enhanced magnetization prepared-rapid gradient echo (MP-RAGE) sequence in 3-dimensional reconstruction (sagittal, coronal, axial). (D) High-resolution T2-weighted constructive interference in steady state (CISS) sequence in axial view without contrast enhancement.Figure 5.. Magnetic resonance imaging scan of the head. (D) T2-weighted moderate hyperintense, homogenous lesion with (A–C) only minor peripheral and part central contrast enhancement. (A–D) The lesions again extend into the widened mastoid segment of the right facial canal and present with a mass in the extratemporal segment. (A–C) T1-weighted contrast-enhanced magnetization prepared-rapid gradient echo (MP-RAGE) sequence in 3-dimensional reconstruction (sagittal, coronal, axial). (D) High-resolution T2-weighted constructive interference in steady state (CISS) sequence in axial view without contrast enhancement. Apparent diffusion coefficient (ADC). High ADC supports the diagnosis of a facial schwannoma (average ADC value: 2.1×10–3 mm2/s).Figure 6.. Apparent diffusion coefficient (ADC). High ADC supports the diagnosis of a facial schwannoma (average ADC value: 2.1×10–3 mm2/s).

References:

1.. Seo BF, Choi HJ, Seo KJ, Jung SN, Intraparotid facial nerve schwannomas: Arch Craniofacial Surg, 2019; 20; 71-74

2.. Gumussoy M, Ekmekci S, Intraparotid facial nerve schwannoma in a nine-year-old patient: Diagnosis, classification, and surgical approach stages: J Craniofac Surg, 2019; 30(2); 516-18

3.. Damar M, Dinç AE, Şevik Eliçora S, Facial nerve schwannoma of parotid gland: Difficulties in diagnosis and management: Case Rep Otolaryngol, 2016; 2016; 3999685

4.. Kreeft A, Schellekens PPA, Leverstein H, Intraparotid facial nerve schwannoma. What to do?: Clin Otolaryngol, 2007; 32(2); 125-29

5.. Pathapati D, Barla K, Dayal M, Facial nerve schwannoma: The rare/ great mimicker of vestibular schwannoma/neuroma: Indian J Radiol Imaging, 2021; 31; 510-13

6.. Suárez C, López F, Mendenhall WM, Trends in the management of non-vestibular skull base and intracranial schwannomas: Cancer Manag Res, 2021; 13; 463-78

7.. Kitama T, Hosoya M, Noguchi M, Intratemporal facial nerve schwannomas: A review of 45 cases in a single center: Diagnostics (Basel), 2022; 12(8); 1789

8.. Alicandri-Ciufelli M, Marchioni D, Mattioli F, Critical literature review on the management of intraparotid facial nerve schwannoma and proposed decision-making algorithm: Eur Arch Oto-Rhino-Laryngology, 2009; 266(4); 475-79

9.. Zhang GZ, Su T, Xu JM, Cheng ZQ, Clinical retrospective analysis of 9 cases of intraparotid facial nerve schwannoma: J Oral Maxillofac Surg, 2016; 74(8); 1695-705 [Erratum in: J Oral Maxillofac Surg. 2016;74(11):2330]

10.. Bewley AF, Azhdam AM, Borrelli M, Intraparotid facial nerve schwannoma mimicking primary parotid neoplasm: Ear, Nose Throat J, 2021; 100(6); 881-83

11.. Quesnel AM, Santos F, Evaluation and management of facial nerve schwannoma: Otolaryngol Clin North Am, 2018; 51(6); 1179-92

12.. Moon JH, Chang WS, Jung HH, Gamma Knife surgery for facial nerve schwannomas: J Neurosurg, 2014; 121(2); 116-22

13.. Eshraghi AA, Oker N, Ocak E, Management of facial nerve schwannoma: A multicenter study of 50 cases: J Neurol Surg B Skull Base, 2019; 80(4); 352-56

14.. Rotter J, Victor M, Graffeo CS, Surgery versus radiosurgery for facial nerve schwannoma: A systematic review and meta-analysis of facial nerve function, postoperative complications, and progression: J Neurosurg, 2021; 135(2); 542-53

15.. Comps JN, Tuleasca C, Goncalves-Matoso B, Upfront Gamma Knife surgery for facial nerve schwannomas: Retrospective case series analysis and systematic review: Acta Neurochir, 2018; 160(5); 987-96

16.. Sheehan JP, Kano H, Xu Z, Gamma Knife radiosurgery for facial nerve schwannomas: A multicenter study: J Neurosurg, 2015; 123(2); 387-94

17.. De Ceulaer J, Decat M, Reychler H, Intraparotid facial nerve schwannoma: Case report and literature review: B-ENT, 2012; 8(3); 225-28

18.. Simone M, Vesperini E, Viti C, Intraparotid facial nerve schwannoma: Two case reports and a review of the literature: Acta Otorhinolaryngol Ital, 2018; 38(1); 73-77

19.. Marchioni D, Alicandri Ciufelli M, Presutti L, Intraparotid facial nerve schwannoma: Review and classification proposal: J Laryngol Otol, 2007; 21(8); 707-12

Figures

Figure 1.. Magnetic resonance imaging scan of the head. (A, B) T2-weighted very hyperintense, homogenous, almost cystic lesion, with (C, D) only minor peripheral and partly septal contrast-enhancement. (E, F) The lesions extend into the right facial canal’s widened mastoid segment and (B, D) present with a mass in the extratemporal segment. (A, B) T2-weighted fat-suppressed inversion recovery sequence in coronal view. (C–E) T1-weighted fat-suppressed dixon sequence with contrast-enhancement in (C, D) coronal and (E) axial view. (F) High-resolution T2-weighted SPACE fat-suppressed sequence in axial view.Figure 2.. Apparent diffusion coefficient (ADC). High ADC supports the diagnosis of a facial schwannoma (average ADC value: 2.1×10–3 mm2/s).Figure 3.. Ultrasound image of the right parotid gland in (A) transversal and (B) sagittal plane. Both images show an intraparenchymal, sharply circumscribed, anechoic, homogeneous, lobulated mass with dorsal sound enhancement extending from the mastoid. The mass measures 13.9×14.8×17.4 mm. GLP righ – parotid gland; UK – mandible; MM – musculus masseter.Figure 4.. Ultrasound findings in the (A) transversal and (B) sagittal plane. Ultrasound images reveal a mass of similar morphologic ultrasound criteria as previously described: intraparenchymal in the parotid gland showing a sharply circumscribed, anechoic, dorsal ultrasound enhancement, measuring 19.5×18.3×26.5 mm.Figure 5.. Magnetic resonance imaging scan of the head. (D) T2-weighted moderate hyperintense, homogenous lesion with (A–C) only minor peripheral and part central contrast enhancement. (A–D) The lesions again extend into the widened mastoid segment of the right facial canal and present with a mass in the extratemporal segment. (A–C) T1-weighted contrast-enhanced magnetization prepared-rapid gradient echo (MP-RAGE) sequence in 3-dimensional reconstruction (sagittal, coronal, axial). (D) High-resolution T2-weighted constructive interference in steady state (CISS) sequence in axial view without contrast enhancement.Figure 6.. Apparent diffusion coefficient (ADC). High ADC supports the diagnosis of a facial schwannoma (average ADC value: 2.1×10–3 mm2/s).

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923