Case Reports in Otolaryngology

Case Reports in Otolaryngology / 2018 / Article

Case Report | Open Access

Volume 2018 |Article ID 1413960 | https://doi.org/10.1155/2018/1413960

James H. Kim, Nathan Tu, Bozena Barbara Wrobel, "Paraganglioma Presenting as a Nasal Septal Mass: Case Report and Literature Review", Case Reports in Otolaryngology, vol. 2018, Article ID 1413960, 7 pages, 2018. https://doi.org/10.1155/2018/1413960

Paraganglioma Presenting as a Nasal Septal Mass: Case Report and Literature Review

Academic Editor: Richard T. Miyamoto
Received07 Aug 2018
Accepted19 Nov 2018
Published06 Dec 2018

Abstract

Objectives. To describe a rare case of a paraganglioma arising from the nasal septum and review the diagnosis and management of paragangliomas in the nasal cavity and paranasal sinuses. Methods. We present a case of a 70-year-old female presenting with persistent nasal congestion and obstruction. Nasal endoscopy revealed a posterior septal mass approaching the sphenoid sinuses and partially obstructing the nasopharynx. A biopsy of the mass was taken, and histologic analysis confirmed a diagnosis of paraganglioma. Results. The patient underwent an endoscopic resection of the tumor. There has been no evidence of disease recurrence after 3 months of follow‐up. Conclusions. Paragangliomas arising from the nasal septum are exceedingly rare, but should be considered in the differential diagnosis in patients presenting with nasal septal masses. These tumors are typically benign, although few cases of malignant sinonasal paragangliomas have been reported. Treatment requires surgical excision with close follow-up as several cases of tumor recurrence have been reported.

1. Introduction

Paragangliomas are rare, slow-growing neuroendocrine tumors arising from cells of neural crest origin. They typically originate from the adrenal gland, but occur extra-adrenally in 5–10% of cases [1]. In the head and neck region, paragangliomas make up only 0.6% of all tumors, most commonly in the carotid body and jugular-tympanic regions [13]. Though rare, there are few reported cases of paragangliomas arising in the nasal cavity and paranasal sinuses [48]. To our knowledge, there has only been one previously reported case of a paraganglioma originating from the nasal septum [8].

2. Case Report

A 70-year-old female complaining of persistent nasal congestion and obstruction presented to our clinic for evaluation. She denied any headache or epistaxis. Nasal endoscopy was performed which showed a posterior septal mass approaching the sphenoid sinuses bilaterally and partially obstructing the view of the nasopharynx. The overlying mucosa was intact except for a small area superiorly which showed a soft granulomatous mass protruding into the left nasal cavity.

The patient was taken to the operating room for septoplasty with biopsy of the mass at an outside institution. Microscopic examination of the biopsy specimen demonstrated clusters of epithelioid-appearing cells separated by bands of fibrillary stroma. The epithelioid cells were noted to have abundant amphophilic cytoplasm, uniform, rounded nuclei with “salt and pepper” chromatin, and small nucleoli. No mitotic activity, invasion, necrosis, or calcification was seen. Immunohistochemical staining demonstrated positivity for neuron-specific enolase (NSE), chromogranin A, synaptophysin, and CD56 cell markers within the epithelioid cells. Fibrillary cells were positive for NSE, chromogranin A, S-100, glial fibrillary acid protein (GFAP), and CD56 cell markers. Based on the histological appearance and immunohistochemical staining, a diagnosis of paraganglioma was made.

The patient was referred to our institution for further management. Preoperative CT imaging showed a smoothly marginated, soft tissue density mass centered at the posterior nasal septum with extension into the nasopharynx and bulging into the right sphenoid sinus (Figure 1). Severe thinning and smooth remodeling of the anterior wall of the sphenoid sinus and anterior clivus were seen. MRI imaging demonstrated hyperintense signaling of the mass on T1-weighted images with a peripheral rim of hypointense signaling on T2-weighted imaging suggestive of a capsule.

An endoscopic resection of the mass was performed. Intraoperatively, a large mass was seen in the posterior aspect of the septum, bulging into the bilateral nasal cavities and extending into the sphenoid sinuses (Figure 2). Erosion of the bone of the rostrum and anterior face of the sphenoid were also seen. Complete resection of the mass was achieved through a posterior septectomy and bilateral sphenoidotomy with tissue removal. Postoperative histologic analysis of the specimen was consistent with a paraganglioma (Figure 3).

The patient has been symptom free without local recurrence 3 months following tumor resection.

3. Discussion

Paragangliomas arising from the nasal cavity and nasal sinuses are extremely rare. Only 48 total cases of paragangliomas occurring in the nasal cavity or paranasal sinuses have been reported to date, occurring in patients with ages ranging between 8 and 89 years old, with an average age of 49 years old. These tumors are slightly more prevalent amongst females (60.4%). Twelve were reported to be malignant in nature, demonstrating either intracranial extension or metastasis to the cervical lymph nodes, brain, and bone. These masses were most frequently reported to originate from the ethmoid sinuses, middle turbinate, and maxillary sinuses. Other less common locations of origin reported include the superior and inferior turbinates, and the sphenoid and frontal sinuses. To the best of our knowledge, there has only been one other reported case of a paraganglioma originating from the nasal septum (Table 1) [8].


CaseAuthorYearSexAgeLocationBiological behaviorTherapyFollow-up

1Harkins [9]1957F52Ethmoid sinusBenignExc, RT, lig ECANED at 1 year
2Moran [10]1962F89Nasal cavityBenignExc, ligation of external carotid arteryNED at 2 years
3Lack [3]1976F50High nasal vaultBenign (local recurrence x3)Exc, ligation of external carotid arteryNED at 11 years following final exc
4F50Middle turbinateBenignExcNo f/u
5M8Middle turbinateBenignExcNED at 7 years
6Gosavi [11]1978M65Middle turbinateBenignExc, RT (incomplete cycle)NED at 2 years
7Apple [12]1982F50Ethmoid, sphenoid, and maxillary sinusesBenign; ectopic ACTH productionExc, RT, hormonal pharmacotherapyNED at 6 years
8Koegel [13]1982F71Maxillary and sphenoid sinusesMalignant (extension into temporal lobe); catecholamine secretionRTNo follow-up available
9Himelfarb [14]1985M41Middle turbinateBenignExcNED at 1 year
10Straehler [15]1985M42Maxillary sinusBenignExcNo follow-up available
11Ueda [16]1985F31Maxillary and ethmoid sinusesBenignExcNED at 2.5 years
12Watson [17]1988M56Inferior turbinateBenignExc, RTNED at 3 years
13Branham [18]1989F25Maxillary and ethmoid sinusesMalignant (brain metastasis)Exc, RT, CTDOD at 3 years
14Kuhn [19]1989M62Superior nasal cavity, ethmoid sinusBenignExcNED at 1 year
15Shimoda [20]1989M58Ethmoid sinus and bilateral frontal fossaMalignant (cervical lymph nodes metastasis)Exc, RTNED at 2 years
16Talbot [21]1990F17Maxillary sinusBenignEmbolization of internal maxillary artery, excNED at 3 months
17Nguyen [22]1995F32Ethmoid sinusMalignant (bone metastasis)Exc, RTDOD at 15 years
18Biswas [23]1999F45Middle turbinateBenignExcNED at 1 month
19Sharma [24]1999M33Frontal sinus (primary); orbit, optic nerve, cavernous sinus, maxillary and ethmoid sinuses (recurrent)Malignant (recurrence, intracranial invasion)CT, RT, exc; vessel embolization, exc 8 months later for recurrenceDOD at 19 months
20Welkoborsky [25]2000F54Nasal cavityBenignExcNED at 3 years
21F56Ethmoid sinusMalignant (brain metastasis)Exc, RTDOD at 28 months
22M57Ethmoid sinusBenignExc, RTNED at 2 years
23Scott [26]2001M24Nasal cavity, ethmoid sinusBenignExcNED at 15 months
24Lecuna [27]2002F72Ethmoid sinus (primary); maxillary sinus (recurrent)Malignant (recurrence, submandibular lymph nodes metastasis)ExcNo follow-up available
25Ketabchi [28]2003F72Nasal cavityBenignExcNED at 8 months
26Mouadeb [29]2003M72Ethmoid sinusBenignExcNED at 4 years
27Lieberum [30]2003M64Frontal and ethmoid sinusesBenign; ectopic ACTH productionExcNED at 2 years
28Askar [31]2003M47Middle turbinateBenignExcNED at 2 years
29Rocha [32]2005M45Nasal cavityBenignExcNED at 5 months
30Liess [33]2007F64Sphenoid sinus, sella, cavernous sinusBenignPartial exc, RTResidual tumor stable at 2 years
31Morales [7]2007F41Sphenoid and ethmoid sinusesBenignNo information availableNo follow-up available
32Jin [34]2008F23Superior turbinateMalignant (questionable transformation simulating Ewing's sarcoma/primitive neuroectodermal tumor)ExcNED at 3 years
33Fasunla [35]2008F39Lateral nasal wallBenignExcNED at 1 year
34Kisser [6]2012F36Maxillary sinusBenignExcNED at 1 year
35Zainine [36]2012F43Middle turbinateBenignExcNED at 2 years
36Papaspyrou [4]2013F33Nasal cavityBenignExcNED at 15 years
37M64Ethmoid sinuses, anterior skull base, frontal lobe dura (primary); ethmoid, sphenoid, cavernous sinus, orbit (recurrent)Malignant (recurrent, intradural metastases)2 exc, RT, CTDOD at 60 months
38F56Ethmoid sinusMalignant (brain metastasis)Exc, RTDOD at 28 months
39F80Nasal cavity, maxilloethmoidal angle, maxillary, ethmoid, and sphenoid sinuses (primary); nasal cavity, anterior skull base, frontal lobe (recurrent)Malignant (recurrent, intracranial invasion)ExcDNOD 17 months after 2nd surgery
40M57Ethmoid sinusBenignExcNED at 2 years
41F54Nasal cavityBenignExcNED at 3 years
42Granato [5]2013F61Nasal cavityBenignExcNED at 2 years
43Michel [37]2013M47Maxillary sinusMalignant (cervical lymph node metastasis)Exc, RTNED at 1 year
44Amiraraghi [38]2013M29Cribriform plate and fovea ethmoidalis presenting as nasal polypsBenignExcNED at 3 years
45Arora [39]2014F40Lateral nasal wallBenignExcNED at 2 years
46Yamaguchi [40]2015F66Ethmoid sinusBenignExcNED at 1 year
47Aydin [41]2015F32Nasal cavity, ethmoid sinusBenign (local recurrence x2)ExcNED 5 years after final exc
48Srivastava [8]2016M60Anterior nasal septumBenignExcNED at 18 months

RT, radiation therapy; CT, chemotherapy; Exc, surgical excision; NED, no evidence of disease; DOD, died of disease.

The most commonly reported symptoms included nasal obstruction, headache, and recurrent epistaxis. The vast majority of reported cases were functionally inactive, with no evidence of catecholamine or adrenocorticotropic hormone (ACTH) secretion. However, there have been a few reported cases of metabolically active tumors secreting ACTH and catecholamines, causing hypertension and other cushingoid features in the affected patient [12, 13, 30]. As such, testing for catecholamine and metanephrines is recommended in symptomatic patients [29, 33].

The origin of paragangliomas in the nasal cavity and paranasal sinuses remains unclear. Although paraganglionic tissue has never been demonstrated in the nasal cavity, a wider distribution of paraganglionic tissue is thought to exist in fetuses and neonates, degenerating after birth [42, 43]. Some authors have suggested that the migration of these embryonic paraganglionic cells accounts for the occurrence of paragangliomas in areas with no known paraganglionic tissue like the nasal cavity [13, 29, 42, 44]. Additionally, it has been shown that paraganglionic tissue exists around the terminal portion of the maxillary artery in infants and could possibly represent a point of origin for paragangliomas in the nasal cavity due to its anatomic proximity [19, 45]. Others have suggested that the paraganglionic tissue may exist in the pterygopalatine fossa due to the close relationship of paraganglionic tissue with arteries and cranial nerves [18, 19, 22].

In contrast, chondrocytes of the cartilaginous septum develop from neural crest cells between the nasal cavities by the ninth week of embryonic development [46, 47]. This is evident by the expression of various neural crest stem cell markers in nasal septum progenitor cells [48]. Several studies have demonstrated that these neural crest-derived chondrocytes in an adult septum retain pluripotent properties, with the capacity for osteogenic, chondrogenic, and neurogenic differentiation [48, 49]. It is possible that the neural crest origin of septal chondrocytes plays an important factor in the development of nasal septal paragangliomas. Despite the neural crest origins of nasal chondrocytes, paragangliomas arising from the nasal septum are extremely rare.

Masses arising from the nasal septum represent a broad spectrum of diagnoses including polyps, chondromas, hematomas, and hemangiomas. Less common septal lesions include sarcomas, melanomas, and other neoplasms [46, 50]. Patients with septal masses typically present with nonspecific symptoms of nasal obstruction or epistaxis, making it difficult to diagnose these masses based on clinical evaluation alone. CT imaging and MR imaging are useful tools in determining exact locations and boundaries of lesions, as well as identifying any erosion or extension into surrounding structures, though imaging features of most masses are nondiagnostic. Diagnosis of such masses requires a combination of clinical history and histopathologic findings. Although paragangliomas of the nasal septum are extremely rare, they should be included in the differential diagnosis when evaluating a septal mass.

The classical histologic appearance of a paraganglioma features an alveolar pattern of well-defined clusters of epithelioid chief cells with round nuclei and eosinophilic cytoplasm. These cells form a “zellballen” pattern of nests separated by vascular stroma and spindle-shaped sustentacular cells [28, 43]. Immunohistochemical staining of paragangliomas generally demonstrates positive markers for NSE, synaptophysin, and chromogranin within the chief cells and S-100 and GFAP within the sustentacular cells [8, 25, 43].

Although the majority of reported nasal paragangliomas are benign, 12 of the cases presented in this review (25.0%) were reported to be malignant, demonstrating intracranial invasion and/or metastasis to the brain, cervical lymph nodes, and bones. It is widely accepted that malignancy cannot be diagnosed by histology alone, but requires evidence of bony invasion or distant metastasis [18, 19, 22]. Certain histological features such as mitotic figures, cellular pleomorphism, necrosis, and vascular invasion can be suggestive of malignancy, though none of these features have been shown to be reliable predictors of malignancy [19, 51]. As such, evaluation with CT imaging and MRI imaging plays a key role in both localization of the primary tumor and identification of local invasion or metastasis.

Almost all of the cases presented in this review were treated surgically. Suggested management of sinonasal paragangliomas involves surgical excision with close follow-up, as several cases of recurrence have been reported [4, 22, 29]. Due to the rich vascularity of these masses, preoperative embolization of the vessels supplying the tumor has been suggested to reduce bleeding during surgery [4, 22, 24]. Many authors suggest the use of radiation therapy to slow the rate of recurrence, although radiation therapy alone has not been shown to be curative [3, 22, 33]. Chemotherapy as a primary treatment for these tumors has been shown to be largely ineffective, though 3 cases reported in this review reported the use of chemotherapy in addition to surgery and radiation therapy in the management of malignant sinonasal paragangliomas [4, 22].

4. Conclusion

We present a rare case of a paraganglioma presenting as a septal mass. To the best of the authors’ knowledge, only one other case of nasal septal paragangliomas has been reported. Clinically, these lesions cause symptoms of obstruction, congestion, headache, and epistaxis. Recognizing that paragangliomas can arise from the nasal septum is crucial to accurately diagnose these tumors when evaluating septal lesions. A diagnosis of a paragangliomas can be confirmed based on the histopathologic imaging. These tumors are usually benign but require additional imaging as malignancy cannot reliably be ruled out on the basis of histology alone. Treatment of these tumors requires surgical resection, though radiation therapy has been to show slow growth and decrease recurrence.

Conflicts of Interest

The authors declare that there are no conflicts of interest or financial disclosures.

Acknowledgments

The authors thank Dr. David Yau for providing the histopathology images and Dr. John Go for reviewing the radiology images.

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