Case Report
EAJM 2012; 44: 185-7
Complete Nasopharyngeal Stenosis: Presentation of a Rare Case Komplet Nazofarengeal Stenoz: Nadir Bir Olgu Sunumu Hakan Korkmaz1, Omer Tarik Selcuk2, Emel Cadalli Tatar1, Guleser Saylam1, Ali Ozdek1 Clinic of 2nd ENT, Diskapi Yildirim Beyazid Training and Research Hospital, Ankara, Turkey Clinic of ENT, Agri State Hospital, Agri, Turkey
1 2
Abstract
Özet
A sixty-eight years old female patient with complete nasopharyngeal stenosis without any determined etiology is presented. She had complete nasal obstruction as well as obstructive sleep apnea syndrome. She was operated and dense, thick, avascular fibrotic tissues were excised and reconstructed with local flaps and skin graft. Only partial opening was achieved in the long term follow-up.
Komplet nazofaringeal stenoz, yumuşak damak ve posterior farinks kaynaklı submukozal fibrotik doku ile nazofarenksin obstrüksiyonu ile karakterize çok nadir bir durum. Altmış sekiz yaşında bir kadın hasta, 30 yıldır devam eden burun tıkanıklığı, nefes darlığı, yutma güçlüğü, ağzı açık uyuma, horlama ve uyku apnesi şikayetleri ile kliniğimize başvurdu. Fizik muayene de uvula ve velopharyngeal açıklık yoktu ve dişlerinde anomalileri mevcuttu idi. Bu olgu sunumunda nazofaringeal stenoz klinik bulguları ve tedavisi ile sunulmaktadır.
Key Words: Nasopharyngeal stenosis, Sleep apnea, Surgery
Anahtar Kelimeler: Nazofaringeal stenoz, Uyku apnesi, Cerrahi
Introduction Complete nasopharyngeal stenosis (NPS) is a very rare condition that is characterized by the obstruction of the nasopharynx by submucosal fibrotic tissue from the soft palate and posterior pharynx [1]. NPS has recently been reported as a complication of infectious diseases, such as rhinoscleroma, diphtheria, syphilis and tuberculosis. However, NPS is frequently iatrogenic and caused by oropharyngeal surgeries such as uvulopalatopharyngoplasty and aggressive tonsillectomy, which are generally discouraged procedures. Here, we present the clinical findings and treatment of a patient with NPS.
Case Report A sixty-eight-year-old female patient arrived at our clinic with complaints of nasal obstruction, shortness of breath, swallowing difficulty, sleeping with an open mouth, snoring and a 30-year history of sleep apnea A physical examination demonstrated that the uvula and the velopharyngeal opening were absent and her teeth were anomalous (Figure 1). The patient did not have a history of pharyngeal surgery or trauma. Computerized tomography (CT) of the paranasal
sinuses and nasopharynx revealed a 2 cm-thick soft tissue mass obstructing the nasopharynx (Figure 2) [2]. There were widespread infectious mucosal changes in the sinonasal cavity. CT of the thorax demonstrated cylindrical bronchiectasis on the mediobasal and posterobasal segments of the lower lobe of the right lung. The patient underwent polysomnography. Her apnea hypopnea index was 32.8, and her oxygen desaturation index was 30.4. O2 saturation less than 90% was 17.7%. All autoimmune markers, PPD and VDRL tests were negative. A pure tone audiogram identified severe mixed hearing loss in both ears. With a tympanogram, the middle ear pressures were measured as -200 and -400 in the right and left ears, respectively. The patient underwent opening surgery for nasopharyngeal stenosis under general anesthesia with orotracheal intubation. The peroperative findings were consistent with the preoperative findings, and the soft palate was adhered to the posterior pharyngeal wall by thick fibrotic tissue. An incision was made horizontally through the lateral folds at the posterior wall by electrocoagulation (Figure 3). The stenotic tissue was determined to be approximately 1 cm thick, avascular and very dense. With the help of a surgical elevator advanced from the nasal cavity, an opening was provided between the dissection plane and the nasal cavity. The opening was widened laterally on both sides. An approximately 4x6 cm
Received: November 02, 2011 / Accepted: February 09, 2012 Correspondence to: Omer Tarik Selcuk, Clinic of ENT, Agri State Hospital, Agri, Turkey Phone: +90 533 325 44 32 e-mail: [email protected] doi:10.5152/eajm.2012.44
186
Korkmaz et al. Complete Nasopharyngeal Stenosis
EAJM 2012; 44: 185-7
Figure 1. On the physical examination of the patient, the uvula and the velopharyngeal opening were not found. Structural anomalies in her teeth were also observed.
Figure 3. Computerized tomography demonstrated a 2 cm-think soft tissue mass obstructing the nasopharynx at the choanae and stenosis of the distal end of the eustachian tube, a lateral pharyngeal recess and torus tubarius in the nasopharynx.
Discussion
Figure 2. Intraoperative images showing the adhesion and the opening between the soft palate and the posterior wall.
split-thickness skin graft taken from the right anterolateral thigh was sutured to the posterior pharyngeal wall. The anterior and posterior walls of the soft palate mucosae were sutured together, and a 3x2 cm nasopharyngeal opening was obtained. A Foley tube was advanced from the left nasal passage, and the cuff was inflated with saline at the level of the new opening. The postsurgical period was uneventful, and the Foley tube was removed on the 7th postoperative day. During the long-term follow up (12 months), partial stenosis recurred, but a limited passage (1x1 cm) was developed. The patient did not consent to the second surgery.
Acquired NPS is most often observed after palatal surgeries and radiation therapy to the nasopharynx. However, in the past, it was mostly due to complications of infectious diseases, such as rhinoscleroma, diphtheria, syphilis and tuberculosis [1, 3]. In childhood, NPS can also be observed after adenotonsillectomy, but the incidence of this complication in both adults and children is very rare [4-6]. Nasopharyngeal stenosis and complete obstruction have been presented in very few reports. Nasopharyngeal stenosis can also be caused by the autoimmune disease cicatricial pemphigoid. Therefore, the patient was tested for autoimmune markers but was negative. Additionally, PDD and VDRL were found to be within normal limits. We were unable to determine any etiologic factor to explain the acquired NPS. Similarly, Shevas et al. [7] also reported a case of nasopharyngeal stenosis with undetermined etiology. Nasopharyngeal stenosis patients can have several disorders that affect the upper aerodigestive system, including nasal obstruction, sleep apnea, swallowing difficulty, voice dysresonance, and eustachian tube and middle ear problems [5]. Our
EAJM 2012; 44: 185-7
Korkmaz et al. Complete Nasopharyngeal Stenosis
patient had almost all of these complaints. Similar to other NPS patients, our patient also had chronic sinonasal problems and infections. Middle ear effusions and rhinosinusitis can also occur. The most effective diagnostic and therapeutic planning approach is transnasal fiberoptic examination. The first reconstruction attempt in nasopharyngeal stenosis was performed by Nichols in 1896. If nasopharyngeal stenosis originates in the soft palate, the use of a laterally based pharyngeal flap is another currently used method of reconstruction. The success of a laterally based pharyngeal flap depends on the use of healthy pharyngeal tissue in the area of the stenosis [5]. Prosthetic nasopharyngeal stents have also been successfully used in some patients [2]. Chheda et al. [8] performed balloon dilatation in three patients with nasopharyngeal stenosis that occurred after radiotherapy. Van Duyne et al. [9] reported the successful opening of nasopharyngeal stenosis using a carbon dioxide laser. Radial forearm free flaps and microvascular approaches are also other surgical options. In summary, NPS is a very rare condition, and its surgical treatment can be very challenging. The comorbidities can be severe in patients who undergo treatment. There is no standard surgical technique used to treat NPS, but several alternatives can be used in sequence in these patients” or simply “but several alternatives can be used in these patients.
References
Conflict of interest statement: The authors declare that they have no conflict of interest to the publication of this article.
187
1. Chang YC, Cheng YK, Tsai MH. A new stenting technique to manage nasopharyngeal stenosis. J Otolaryngol 2006; 35: 430-1. [CrossRef] 2. Eppley BL, Sadove AM, Hennon D, van Aalst JA. Treatment of nasopharyngeal stenosis by prosthetic hollow stents: Clinical experience in eight patients. Cleft Palate Craniofac J 2006; 43: 374-8. [CrossRef] 3. Jones LM, Guillory VL, Mair EA. Total nasopharyngeal stenosis: treatment with laser excision, nasopharyngeal obturators, and topical mitomycin-c. Otolaryngol Head Neck Surg 2005; 133: 795-8. [CrossRef] 4. Krespi YP, Kacker A. Management of nasopharyngeal stenosis after uvulopalatoplasty. Otolaryngol Head Neck Surg 2000; 123: 692-5. [CrossRef] 5. Giannoni C, Sulek M, Friedman EM, Duncan NO 3rd. Acquired nasopharyngeal stenosis: a warning and review. Arch Otolaryngol Head Neck Surg 1998; 124: 163-7. 6. McLaughlin KE, Jacobs IN, Todd NW, Gussack GS, Carlson G. Management of nasopharyngeal and oropharyngeal stenosis in children. Laryngoscope 1997; 107: 1322-31. [CrossRef] 7. Shevas AT, Kastanioudakis IG, Constantopoulos SH, Assimakopoulos DA. Acquired nasopharyngeal obstruction and “Metsovo lung”. Rhinology 1995; 33: 240-3. 8. Chheda NN, Postma GN. Balloon dilation of an acquired nasopharyngeal stenosis. Otolaryngol Head Neck Surg 2009; 140: 939-41. [CrossRef] 9. Van Duyne J, Coleman JA Jr. Treatment of nasopharyngeal inlet stenosis following uvulopalatopharyngoplasty with the CO2 laser. Laryngoscope 1995; 105: 914-8. [CrossRef]
Copyright of Eurasian Journal of Medicine is the property of Aves Yayincilik Ltd. STI and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
EAJM 2012; 44: 185-7
Complete Nasopharyngeal Stenosis: Presentation of a Rare Case Komplet Nazofarengeal Stenoz: Nadir Bir Olgu Sunumu Hakan Korkmaz1, Omer Tarik Selcuk2, Emel Cadalli Tatar1, Guleser Saylam1, Ali Ozdek1 Clinic of 2nd ENT, Diskapi Yildirim Beyazid Training and Research Hospital, Ankara, Turkey Clinic of ENT, Agri State Hospital, Agri, Turkey
1 2
Abstract
Özet
A sixty-eight years old female patient with complete nasopharyngeal stenosis without any determined etiology is presented. She had complete nasal obstruction as well as obstructive sleep apnea syndrome. She was operated and dense, thick, avascular fibrotic tissues were excised and reconstructed with local flaps and skin graft. Only partial opening was achieved in the long term follow-up.
Komplet nazofaringeal stenoz, yumuşak damak ve posterior farinks kaynaklı submukozal fibrotik doku ile nazofarenksin obstrüksiyonu ile karakterize çok nadir bir durum. Altmış sekiz yaşında bir kadın hasta, 30 yıldır devam eden burun tıkanıklığı, nefes darlığı, yutma güçlüğü, ağzı açık uyuma, horlama ve uyku apnesi şikayetleri ile kliniğimize başvurdu. Fizik muayene de uvula ve velopharyngeal açıklık yoktu ve dişlerinde anomalileri mevcuttu idi. Bu olgu sunumunda nazofaringeal stenoz klinik bulguları ve tedavisi ile sunulmaktadır.
Key Words: Nasopharyngeal stenosis, Sleep apnea, Surgery
Anahtar Kelimeler: Nazofaringeal stenoz, Uyku apnesi, Cerrahi
Introduction Complete nasopharyngeal stenosis (NPS) is a very rare condition that is characterized by the obstruction of the nasopharynx by submucosal fibrotic tissue from the soft palate and posterior pharynx [1]. NPS has recently been reported as a complication of infectious diseases, such as rhinoscleroma, diphtheria, syphilis and tuberculosis. However, NPS is frequently iatrogenic and caused by oropharyngeal surgeries such as uvulopalatopharyngoplasty and aggressive tonsillectomy, which are generally discouraged procedures. Here, we present the clinical findings and treatment of a patient with NPS.
Case Report A sixty-eight-year-old female patient arrived at our clinic with complaints of nasal obstruction, shortness of breath, swallowing difficulty, sleeping with an open mouth, snoring and a 30-year history of sleep apnea A physical examination demonstrated that the uvula and the velopharyngeal opening were absent and her teeth were anomalous (Figure 1). The patient did not have a history of pharyngeal surgery or trauma. Computerized tomography (CT) of the paranasal
sinuses and nasopharynx revealed a 2 cm-thick soft tissue mass obstructing the nasopharynx (Figure 2) [2]. There were widespread infectious mucosal changes in the sinonasal cavity. CT of the thorax demonstrated cylindrical bronchiectasis on the mediobasal and posterobasal segments of the lower lobe of the right lung. The patient underwent polysomnography. Her apnea hypopnea index was 32.8, and her oxygen desaturation index was 30.4. O2 saturation less than 90% was 17.7%. All autoimmune markers, PPD and VDRL tests were negative. A pure tone audiogram identified severe mixed hearing loss in both ears. With a tympanogram, the middle ear pressures were measured as -200 and -400 in the right and left ears, respectively. The patient underwent opening surgery for nasopharyngeal stenosis under general anesthesia with orotracheal intubation. The peroperative findings were consistent with the preoperative findings, and the soft palate was adhered to the posterior pharyngeal wall by thick fibrotic tissue. An incision was made horizontally through the lateral folds at the posterior wall by electrocoagulation (Figure 3). The stenotic tissue was determined to be approximately 1 cm thick, avascular and very dense. With the help of a surgical elevator advanced from the nasal cavity, an opening was provided between the dissection plane and the nasal cavity. The opening was widened laterally on both sides. An approximately 4x6 cm
Received: November 02, 2011 / Accepted: February 09, 2012 Correspondence to: Omer Tarik Selcuk, Clinic of ENT, Agri State Hospital, Agri, Turkey Phone: +90 533 325 44 32 e-mail: [email protected] doi:10.5152/eajm.2012.44
186
Korkmaz et al. Complete Nasopharyngeal Stenosis
EAJM 2012; 44: 185-7
Figure 1. On the physical examination of the patient, the uvula and the velopharyngeal opening were not found. Structural anomalies in her teeth were also observed.
Figure 3. Computerized tomography demonstrated a 2 cm-think soft tissue mass obstructing the nasopharynx at the choanae and stenosis of the distal end of the eustachian tube, a lateral pharyngeal recess and torus tubarius in the nasopharynx.
Discussion
Figure 2. Intraoperative images showing the adhesion and the opening between the soft palate and the posterior wall.
split-thickness skin graft taken from the right anterolateral thigh was sutured to the posterior pharyngeal wall. The anterior and posterior walls of the soft palate mucosae were sutured together, and a 3x2 cm nasopharyngeal opening was obtained. A Foley tube was advanced from the left nasal passage, and the cuff was inflated with saline at the level of the new opening. The postsurgical period was uneventful, and the Foley tube was removed on the 7th postoperative day. During the long-term follow up (12 months), partial stenosis recurred, but a limited passage (1x1 cm) was developed. The patient did not consent to the second surgery.
Acquired NPS is most often observed after palatal surgeries and radiation therapy to the nasopharynx. However, in the past, it was mostly due to complications of infectious diseases, such as rhinoscleroma, diphtheria, syphilis and tuberculosis [1, 3]. In childhood, NPS can also be observed after adenotonsillectomy, but the incidence of this complication in both adults and children is very rare [4-6]. Nasopharyngeal stenosis and complete obstruction have been presented in very few reports. Nasopharyngeal stenosis can also be caused by the autoimmune disease cicatricial pemphigoid. Therefore, the patient was tested for autoimmune markers but was negative. Additionally, PDD and VDRL were found to be within normal limits. We were unable to determine any etiologic factor to explain the acquired NPS. Similarly, Shevas et al. [7] also reported a case of nasopharyngeal stenosis with undetermined etiology. Nasopharyngeal stenosis patients can have several disorders that affect the upper aerodigestive system, including nasal obstruction, sleep apnea, swallowing difficulty, voice dysresonance, and eustachian tube and middle ear problems [5]. Our
EAJM 2012; 44: 185-7
Korkmaz et al. Complete Nasopharyngeal Stenosis
patient had almost all of these complaints. Similar to other NPS patients, our patient also had chronic sinonasal problems and infections. Middle ear effusions and rhinosinusitis can also occur. The most effective diagnostic and therapeutic planning approach is transnasal fiberoptic examination. The first reconstruction attempt in nasopharyngeal stenosis was performed by Nichols in 1896. If nasopharyngeal stenosis originates in the soft palate, the use of a laterally based pharyngeal flap is another currently used method of reconstruction. The success of a laterally based pharyngeal flap depends on the use of healthy pharyngeal tissue in the area of the stenosis [5]. Prosthetic nasopharyngeal stents have also been successfully used in some patients [2]. Chheda et al. [8] performed balloon dilatation in three patients with nasopharyngeal stenosis that occurred after radiotherapy. Van Duyne et al. [9] reported the successful opening of nasopharyngeal stenosis using a carbon dioxide laser. Radial forearm free flaps and microvascular approaches are also other surgical options. In summary, NPS is a very rare condition, and its surgical treatment can be very challenging. The comorbidities can be severe in patients who undergo treatment. There is no standard surgical technique used to treat NPS, but several alternatives can be used in sequence in these patients” or simply “but several alternatives can be used in these patients.
References
Conflict of interest statement: The authors declare that they have no conflict of interest to the publication of this article.
187
1. Chang YC, Cheng YK, Tsai MH. A new stenting technique to manage nasopharyngeal stenosis. J Otolaryngol 2006; 35: 430-1. [CrossRef] 2. Eppley BL, Sadove AM, Hennon D, van Aalst JA. Treatment of nasopharyngeal stenosis by prosthetic hollow stents: Clinical experience in eight patients. Cleft Palate Craniofac J 2006; 43: 374-8. [CrossRef] 3. Jones LM, Guillory VL, Mair EA. Total nasopharyngeal stenosis: treatment with laser excision, nasopharyngeal obturators, and topical mitomycin-c. Otolaryngol Head Neck Surg 2005; 133: 795-8. [CrossRef] 4. Krespi YP, Kacker A. Management of nasopharyngeal stenosis after uvulopalatoplasty. Otolaryngol Head Neck Surg 2000; 123: 692-5. [CrossRef] 5. Giannoni C, Sulek M, Friedman EM, Duncan NO 3rd. Acquired nasopharyngeal stenosis: a warning and review. Arch Otolaryngol Head Neck Surg 1998; 124: 163-7. 6. McLaughlin KE, Jacobs IN, Todd NW, Gussack GS, Carlson G. Management of nasopharyngeal and oropharyngeal stenosis in children. Laryngoscope 1997; 107: 1322-31. [CrossRef] 7. Shevas AT, Kastanioudakis IG, Constantopoulos SH, Assimakopoulos DA. Acquired nasopharyngeal obstruction and “Metsovo lung”. Rhinology 1995; 33: 240-3. 8. Chheda NN, Postma GN. Balloon dilation of an acquired nasopharyngeal stenosis. Otolaryngol Head Neck Surg 2009; 140: 939-41. [CrossRef] 9. Van Duyne J, Coleman JA Jr. Treatment of nasopharyngeal inlet stenosis following uvulopalatopharyngoplasty with the CO2 laser. Laryngoscope 1995; 105: 914-8. [CrossRef]
Copyright of Eurasian Journal of Medicine is the property of Aves Yayincilik Ltd. STI and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
