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Bilateral oncocytic cysts of the nasopharynx
Bilateral oncocytic cysts of the nasopharynx THEODORE T. BENKE, MD, ROBERT P. ZIT$CH III, MD, and MARCUS B. NASHELSKY, MD, Columbia, Missouri
IW, ysts of the n a s o p h a r y n x are often incidentally discovered lesions that m a y have relatively little clinical significance. However, they must be distinguished f r o m true n e o p l a s m s of the region. S o m e of these lesions m a y occasionally p r o d u c e s y m p t o m s such as nasal obstruction, post nasal drainage, aural fullness, and diminished hearing. T h e most c o m m o n cysts of t h e n a s o p h a r y n x are retention cysts and the cysts of T o r n w a l d t ' s bursa. Oncocytic cystic lesions are u n c o m m o n in all sites of the u p p e r respiratory tract, although they are most often e n c o u n t e r e d in the larynx. In the nasopharynx, at least five cases of unilateral oncocytic cystic lesions have b e e n described. This article is the first r e p o r t of a patient found to have bilateral oncocytic cysts of the nasopharynx. I n t r a o p e r a t i v e p h o t o g r a p h s and histologic sections d e m o n s t r a t e the characteristic gross and microscopic features of the lesions. CASE REPORT
A 59-year-old man came to the otolaryngology clinic with a 2.5-year history of nasal obstruction, postnasal drainage, and loud snoring. The patient was originally thought to have bilateral inferior turbinate hypertrophy and began receiving intranasal beclomethasone spray, which did not relieve the obstruction. After unsuccessful medical treatment, nasal endoscopy revealed marked, bilateral inferior turbinate hypertrophy and copious clear mucus drainage; no pus was seen. A smooth mass was seen in the nasopharynx emanating from the region of the torus tubarius and fossa of Rosenmuller, bilaterally. This large polypoid mass was also seen in the
From the Division of Otolaryngology, Department of Surgery (Drs. Benke and Zitsch) and the Department of Pathology (Dr. Nashelsky), University of Missouri Hospital and Clinics. Presented at the Annual Meeting of the American Academy of Otolaryngology-Head and Neck Surgery, Minneapolis, Minn., Oct. 2-6, 1993. Received for publication April 21, 1994; accepted July 8, 1994. Reprint requests: Robert P. Zitsch III, MD, Division of Otolaryngology, MA 314, One Hospital Drive, Columbia, MO 65212. OTOLARYN~OLHEADNECKSURG1995;112:321-4. Copyright © 1995 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/95/$3.00 + 0 23/4/58969
oropharynx below the level of the soft palate on the right side. The patient was taken to the operating room and was noted to have bilateral, symmetric nasopharyngeal masses, consistent with cysts (Fig. 1). These arose from the posterolateral nasopharyngeal wall, posterior to the eustachian tube orifice, and were adherent to the soft palate. One cyst was opened and contained thick mucus; no pus was found. Each cyst was completely removed endoscopically and submitted for histopathologic study. The surrounding nasopharyngeal mucosa appeared to be thickened. Each cyst measured approximately 1.4 x 1.0 × 0.6 cm, was tan-pink, and had a smooth wall. Histologic sections of each of cysts were similar and demonstrated tissue fragments externally covered by squamous and respiratory mucosa with scattered lobules of seromucous glands within a loose fibrous stroma. The stroma was essentially free of lymphocytes. The fragments showed a central, large, collapsed and irregular cyst lined by two cell layers of columnar and cuboidal oncocytic cells with abundant granular eosinophilic cytoplasm. The nuclei were small and cytologically benign (Figs. 2 and 3). The patient did well after surgery and was discharged on the day after surgery. Three weeks after surgery, he noted marked improvement in his nasal airway and no snoring. At 3 months endoscopy showed no residual nasopharyngeal cysts. DISCUSSION
N a s o p h a r y n g e a l cysts are generally rare findings, I-3 with the medical literature containing only a few case reports. Currently, n a s o p h a r y n g e a l cysts are classified into midline and lateral types, each grouping having both congenital and acquired cysts? Midline cysts lying d e e p to the pharyngobasilar fascia are congenital and originate from Tornwaldt's bursa, 4 a persistent embryonic c o m m u n i c a t i o n between the anterior or caudal n o t o c h o r d and the n a s o p h a r y n x ? T o r n w a l d t ' s cyst, a congenital midline cyst, occurs in approximately 3% of the population. 5 Midline acquired cysts occur s u p e r f c i a l to the pharyngobasilar fascia 4 and are usually retention cysts. 3 Lateral n a s o p h a r y n g e a l cysts s e e m to be less comm o n than midline c y s t s . 3 T h e most c o m m o n congenital lateral cyst is a branchiogenic cyst, which is thought to arise f r o m the second branchial p o u c h ? 321
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Otolaryngology Head and Neck Surgery February 1995
Fig. I. Transnasal endoscopic view of bilateral nasopharyngeal oncocytic cysts shows nearcomplete removal of left cyst {arrow) and an intact right cyst (arrowhead). The posterior nasal septum {S) and left eustachian tube orifice {E) are seen.
Fig. 2. Low-power photomicrograph demonstrates nasopharyngeal tissuewith an outer squamous covering {arrow)and a central cyst lined with a bilayer of oncocytic ceils (arrowhead). (Hematoxylin and eosin; original magnification × 40.]
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Fig. 3. Higher power photomicrograph demonstrates the cyst lining consisting of a bilayer of oncocytic cells with abundant granular eosinophilic cytoplasm. Underlying stroma is essentially free of lymphocytes. {Hematoxylin and eosin; original magnification × 200.)
The usual acquired lateral cyst is a seromucinous gland retention cyst. 3 Oncocytes are large epithelial cells with a deeply eosinophilic granular cytoplasm on routine hematoxylin and eosin preparations. 6 Ultrastructural studies of these cells with electron microscopy reveal abundant mitochondria; histochemistry demonstrates a high oxidative enzyme activity. 7 Oncocytes are located in several areas in the head and neck including the thyroid gland (Hurthle cells), parathyroid gland (oxyphil cells), and major salivary glands (oncocytes). 7'8 They are also found in the lining epithelium and glands (including minor salivary glands) of the respiratory system. 6,9 Lesions composed on oncocytes in the sinonasal tract and larynx are usually a reactive or hyperplastic response to chronic inflammation or the aging phenomenonF ° Oncocytic lesions in the respiratory tract are either solid or assume a papillary configuration within a cystic structure, the latter being much more common. 9 Malignant oncocytic tumors are rare; H benign neoplasms are more common. Nasopharyngeal oncocytic lesions are either clinically inapparent nests of cells found during panendoscopy and blind biopsy, or they are smooth,
mucosally covered masses, usually found in older patients. 12When they occur in peritubal mucoserous glands, however, they may obstruct the eustachian tube, resulting in otitis media with effusion. 1°,13 Other symptoms caused by nasopharyngeal oncocytic lesions include nasal obstruction, nasal pain, rhinorrhea, postnasal drainage, snoring, aural fullness, hearing loss, clicking in the ear, and otalgia. 9'14-]7 Reports describe a variety of cystic nasopharyngeal oncocytic lesions including oncocytic metaplasia, oncocytic cysts, papillary oncocytic cystadenomas, and extraparotid Warthin's tumors. Erlandson and Tandler 6described nests of oncocytes in the nasopharynx and debated whether this represented oncocytic metaplasia or early oncocytoma. The term extraparotid Warthin's tumor has been used by two authors to describe an oncocytic cystic lesion with a lymphocytic infiltrateJ 4,16Very similar lesions described by other authors have been called oncocytic metaplasia of the nasopharynx and oncocytic cysts of the nasopharynx. 13.15Still another report described an oncocytic cyst as a simple cyst lined by oncocytes and surrounded by normal nasopharyngeal lymphoid tissue. 17 The terminology regarding nasopharyngeal onco-
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cytic lesions is certainly confusing, For solid oncocytic lesions, we prefer the term oncocytoma. For oncocytic cystic lesions without an inflammatory infiltrate, we prefer the term oncocytic cyst. Cohen and Batsakis 1° caution that oncocytic cystic lesions with an inflammatory component should not be called extraparotid Warthin's tumors. The oncocytic cyst with an inflammatory component differs from the Warthin's tumor insofar as the latter has lymphoid follicles usually with germinal centers; the former lesion lacks this organization to its lymphoid component. 15 For these lesions, which resemble a Warthin's tumor, Morin's term, oncocytic papillary cystadenoma, seems most appropriateJ ~ Small areas of oncocytes where a cyst cannot be identified seem appropriately labeled oncocytic metaplasia. SUMMARY
We describe a 59-year-old male patient noted to have bilateral oncocytic cysts of the nasopharynx. These cysts appear to represent retention cysts of peritubal mucoserous glands that have undergone oncocytic metaplasia and would be classified as lateral acquired cysts of the nasopharynx. These cysts are benign but may cause symptoms related to their location in the airway and proximity to the eustachian tube. Endoscopic surgical removal is the treatment of choice. REFERENCES
1. Robinson P. Nasopharyngeal cyst. J Laryngol Otol 1988; 102: 182-3. 2. Badrawy R, Safwat F, Fahmy S. Cysts of the nasopharynx. J Laryngol Otol 1974;88:571-8.
Otolaryngology Head and Neck Surgery February 1995
3. Nicolai P, Luzzago F, Magoldi R, Falchetti M, Antonelli A. Nasopharyngeal cysts. Arch Otolaryngol Head Neck Surg 1989;115:860-4. 4. Guggenheim P. Cysts of the nasopharynx. Laryngoscope 1967;75:2147-68. 5. Gustafson R, Neel H. Cysts and tumors of the nasopharynx. In: Paparella M, Shumriek D, Gluckman J, Meyerhoff W, eds. Otolaryngology. 3rd ed. Philadelphia: WB Saunders Co., 1991:2189-98. 6. Erlandson R, Tandler B. Oncocytes in the nasopharynx. Arch Otolaryngol 1977;103:175-8. 7. Johns M, Batsakis J, Short C. Oncocytic and oncocytoid tumors of the salivary glands. Laryngoscope 1973;83: 1940-52. 8. Johns M, Regezi J, Batsakis J. Oncocytic neoplasms of salivary glands: an ultrastructural study. Laryngoscopy 1977;87: 862-71. 9. Batsakis J. Tumors of the upper respiratory tract and ear. In: Hyams V, Batsakis J, Michaels L, eds. Atlas of tumor pathology. 2nd series, Fascicle 25. Washington, D.C.: Armed Forces Institute of Pathology, 1988:88-90. 10. Cohen M, Batsakis J. Oncocytic tumors (oncocytomas) of minor salivary glands. Arch Otolaryngol 1968;88:97-9. 11. Larson D, Fecher R. Pathologic quiz case 1. Arch Otolaryngol 1976;102:320-2. 12. Morin G, Shank E. Burgess L, Heffner D. Oncocytic metaplasia of the pharynx. OTOLARYNGOLH~AD NECKSURG 1991; 105:80-91. 13. Watson C. Oncocytic metaplasia of the nasopharynx-unusual cause of secretory otitis media. J Laryngol Otol 1990; 104:39-40. 14. Griffiths A, Dekker P. Oncocytic metaplasia of the nasopharynx or extra-parotid Warthin's tumor? J Clin Pathol 1991; 44:1030-2. 15. Ophir D, Lifschitz-Mercer B. Oncocytic cystic lesions of the upper respiratory tract. ENT J 1989;68:237-44. 16. Friedmann I. Adenolymphoma of the salivary glands. J Laryngol Otol 1953;67:165-9. 17. Busuttil A. Oncocytic lesions of the upper respiratory tract. J Laryngol Otol 1976;90:277-88.
IW, ysts of the n a s o p h a r y n x are often incidentally discovered lesions that m a y have relatively little clinical significance. However, they must be distinguished f r o m true n e o p l a s m s of the region. S o m e of these lesions m a y occasionally p r o d u c e s y m p t o m s such as nasal obstruction, post nasal drainage, aural fullness, and diminished hearing. T h e most c o m m o n cysts of t h e n a s o p h a r y n x are retention cysts and the cysts of T o r n w a l d t ' s bursa. Oncocytic cystic lesions are u n c o m m o n in all sites of the u p p e r respiratory tract, although they are most often e n c o u n t e r e d in the larynx. In the nasopharynx, at least five cases of unilateral oncocytic cystic lesions have b e e n described. This article is the first r e p o r t of a patient found to have bilateral oncocytic cysts of the nasopharynx. I n t r a o p e r a t i v e p h o t o g r a p h s and histologic sections d e m o n s t r a t e the characteristic gross and microscopic features of the lesions. CASE REPORT
A 59-year-old man came to the otolaryngology clinic with a 2.5-year history of nasal obstruction, postnasal drainage, and loud snoring. The patient was originally thought to have bilateral inferior turbinate hypertrophy and began receiving intranasal beclomethasone spray, which did not relieve the obstruction. After unsuccessful medical treatment, nasal endoscopy revealed marked, bilateral inferior turbinate hypertrophy and copious clear mucus drainage; no pus was seen. A smooth mass was seen in the nasopharynx emanating from the region of the torus tubarius and fossa of Rosenmuller, bilaterally. This large polypoid mass was also seen in the
From the Division of Otolaryngology, Department of Surgery (Drs. Benke and Zitsch) and the Department of Pathology (Dr. Nashelsky), University of Missouri Hospital and Clinics. Presented at the Annual Meeting of the American Academy of Otolaryngology-Head and Neck Surgery, Minneapolis, Minn., Oct. 2-6, 1993. Received for publication April 21, 1994; accepted July 8, 1994. Reprint requests: Robert P. Zitsch III, MD, Division of Otolaryngology, MA 314, One Hospital Drive, Columbia, MO 65212. OTOLARYN~OLHEADNECKSURG1995;112:321-4. Copyright © 1995 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/95/$3.00 + 0 23/4/58969
oropharynx below the level of the soft palate on the right side. The patient was taken to the operating room and was noted to have bilateral, symmetric nasopharyngeal masses, consistent with cysts (Fig. 1). These arose from the posterolateral nasopharyngeal wall, posterior to the eustachian tube orifice, and were adherent to the soft palate. One cyst was opened and contained thick mucus; no pus was found. Each cyst was completely removed endoscopically and submitted for histopathologic study. The surrounding nasopharyngeal mucosa appeared to be thickened. Each cyst measured approximately 1.4 x 1.0 × 0.6 cm, was tan-pink, and had a smooth wall. Histologic sections of each of cysts were similar and demonstrated tissue fragments externally covered by squamous and respiratory mucosa with scattered lobules of seromucous glands within a loose fibrous stroma. The stroma was essentially free of lymphocytes. The fragments showed a central, large, collapsed and irregular cyst lined by two cell layers of columnar and cuboidal oncocytic cells with abundant granular eosinophilic cytoplasm. The nuclei were small and cytologically benign (Figs. 2 and 3). The patient did well after surgery and was discharged on the day after surgery. Three weeks after surgery, he noted marked improvement in his nasal airway and no snoring. At 3 months endoscopy showed no residual nasopharyngeal cysts. DISCUSSION
N a s o p h a r y n g e a l cysts are generally rare findings, I-3 with the medical literature containing only a few case reports. Currently, n a s o p h a r y n g e a l cysts are classified into midline and lateral types, each grouping having both congenital and acquired cysts? Midline cysts lying d e e p to the pharyngobasilar fascia are congenital and originate from Tornwaldt's bursa, 4 a persistent embryonic c o m m u n i c a t i o n between the anterior or caudal n o t o c h o r d and the n a s o p h a r y n x ? T o r n w a l d t ' s cyst, a congenital midline cyst, occurs in approximately 3% of the population. 5 Midline acquired cysts occur s u p e r f c i a l to the pharyngobasilar fascia 4 and are usually retention cysts. 3 Lateral n a s o p h a r y n g e a l cysts s e e m to be less comm o n than midline c y s t s . 3 T h e most c o m m o n congenital lateral cyst is a branchiogenic cyst, which is thought to arise f r o m the second branchial p o u c h ? 321
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BENKE et al.
Otolaryngology Head and Neck Surgery February 1995
Fig. I. Transnasal endoscopic view of bilateral nasopharyngeal oncocytic cysts shows nearcomplete removal of left cyst {arrow) and an intact right cyst (arrowhead). The posterior nasal septum {S) and left eustachian tube orifice {E) are seen.
Fig. 2. Low-power photomicrograph demonstrates nasopharyngeal tissuewith an outer squamous covering {arrow)and a central cyst lined with a bilayer of oncocytic ceils (arrowhead). (Hematoxylin and eosin; original magnification × 40.]
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Fig. 3. Higher power photomicrograph demonstrates the cyst lining consisting of a bilayer of oncocytic cells with abundant granular eosinophilic cytoplasm. Underlying stroma is essentially free of lymphocytes. {Hematoxylin and eosin; original magnification × 200.)
The usual acquired lateral cyst is a seromucinous gland retention cyst. 3 Oncocytes are large epithelial cells with a deeply eosinophilic granular cytoplasm on routine hematoxylin and eosin preparations. 6 Ultrastructural studies of these cells with electron microscopy reveal abundant mitochondria; histochemistry demonstrates a high oxidative enzyme activity. 7 Oncocytes are located in several areas in the head and neck including the thyroid gland (Hurthle cells), parathyroid gland (oxyphil cells), and major salivary glands (oncocytes). 7'8 They are also found in the lining epithelium and glands (including minor salivary glands) of the respiratory system. 6,9 Lesions composed on oncocytes in the sinonasal tract and larynx are usually a reactive or hyperplastic response to chronic inflammation or the aging phenomenonF ° Oncocytic lesions in the respiratory tract are either solid or assume a papillary configuration within a cystic structure, the latter being much more common. 9 Malignant oncocytic tumors are rare; H benign neoplasms are more common. Nasopharyngeal oncocytic lesions are either clinically inapparent nests of cells found during panendoscopy and blind biopsy, or they are smooth,
mucosally covered masses, usually found in older patients. 12When they occur in peritubal mucoserous glands, however, they may obstruct the eustachian tube, resulting in otitis media with effusion. 1°,13 Other symptoms caused by nasopharyngeal oncocytic lesions include nasal obstruction, nasal pain, rhinorrhea, postnasal drainage, snoring, aural fullness, hearing loss, clicking in the ear, and otalgia. 9'14-]7 Reports describe a variety of cystic nasopharyngeal oncocytic lesions including oncocytic metaplasia, oncocytic cysts, papillary oncocytic cystadenomas, and extraparotid Warthin's tumors. Erlandson and Tandler 6described nests of oncocytes in the nasopharynx and debated whether this represented oncocytic metaplasia or early oncocytoma. The term extraparotid Warthin's tumor has been used by two authors to describe an oncocytic cystic lesion with a lymphocytic infiltrateJ 4,16Very similar lesions described by other authors have been called oncocytic metaplasia of the nasopharynx and oncocytic cysts of the nasopharynx. 13.15Still another report described an oncocytic cyst as a simple cyst lined by oncocytes and surrounded by normal nasopharyngeal lymphoid tissue. 17 The terminology regarding nasopharyngeal onco-
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cytic lesions is certainly confusing, For solid oncocytic lesions, we prefer the term oncocytoma. For oncocytic cystic lesions without an inflammatory infiltrate, we prefer the term oncocytic cyst. Cohen and Batsakis 1° caution that oncocytic cystic lesions with an inflammatory component should not be called extraparotid Warthin's tumors. The oncocytic cyst with an inflammatory component differs from the Warthin's tumor insofar as the latter has lymphoid follicles usually with germinal centers; the former lesion lacks this organization to its lymphoid component. 15 For these lesions, which resemble a Warthin's tumor, Morin's term, oncocytic papillary cystadenoma, seems most appropriateJ ~ Small areas of oncocytes where a cyst cannot be identified seem appropriately labeled oncocytic metaplasia. SUMMARY
We describe a 59-year-old male patient noted to have bilateral oncocytic cysts of the nasopharynx. These cysts appear to represent retention cysts of peritubal mucoserous glands that have undergone oncocytic metaplasia and would be classified as lateral acquired cysts of the nasopharynx. These cysts are benign but may cause symptoms related to their location in the airway and proximity to the eustachian tube. Endoscopic surgical removal is the treatment of choice. REFERENCES
1. Robinson P. Nasopharyngeal cyst. J Laryngol Otol 1988; 102: 182-3. 2. Badrawy R, Safwat F, Fahmy S. Cysts of the nasopharynx. J Laryngol Otol 1974;88:571-8.
Otolaryngology Head and Neck Surgery February 1995
3. Nicolai P, Luzzago F, Magoldi R, Falchetti M, Antonelli A. Nasopharyngeal cysts. Arch Otolaryngol Head Neck Surg 1989;115:860-4. 4. Guggenheim P. Cysts of the nasopharynx. Laryngoscope 1967;75:2147-68. 5. Gustafson R, Neel H. Cysts and tumors of the nasopharynx. In: Paparella M, Shumriek D, Gluckman J, Meyerhoff W, eds. Otolaryngology. 3rd ed. Philadelphia: WB Saunders Co., 1991:2189-98. 6. Erlandson R, Tandler B. Oncocytes in the nasopharynx. Arch Otolaryngol 1977;103:175-8. 7. Johns M, Batsakis J, Short C. Oncocytic and oncocytoid tumors of the salivary glands. Laryngoscope 1973;83: 1940-52. 8. Johns M, Regezi J, Batsakis J. Oncocytic neoplasms of salivary glands: an ultrastructural study. Laryngoscopy 1977;87: 862-71. 9. Batsakis J. Tumors of the upper respiratory tract and ear. In: Hyams V, Batsakis J, Michaels L, eds. Atlas of tumor pathology. 2nd series, Fascicle 25. Washington, D.C.: Armed Forces Institute of Pathology, 1988:88-90. 10. Cohen M, Batsakis J. Oncocytic tumors (oncocytomas) of minor salivary glands. Arch Otolaryngol 1968;88:97-9. 11. Larson D, Fecher R. Pathologic quiz case 1. Arch Otolaryngol 1976;102:320-2. 12. Morin G, Shank E. Burgess L, Heffner D. Oncocytic metaplasia of the pharynx. OTOLARYNGOLH~AD NECKSURG 1991; 105:80-91. 13. Watson C. Oncocytic metaplasia of the nasopharynx-unusual cause of secretory otitis media. J Laryngol Otol 1990; 104:39-40. 14. Griffiths A, Dekker P. Oncocytic metaplasia of the nasopharynx or extra-parotid Warthin's tumor? J Clin Pathol 1991; 44:1030-2. 15. Ophir D, Lifschitz-Mercer B. Oncocytic cystic lesions of the upper respiratory tract. ENT J 1989;68:237-44. 16. Friedmann I. Adenolymphoma of the salivary glands. J Laryngol Otol 1953;67:165-9. 17. Busuttil A. Oncocytic lesions of the upper respiratory tract. J Laryngol Otol 1976;90:277-88.