|Year : 2013 | Volume
| Issue : 1 | Page : 18-21
"Doctor I have a frog in my throat": Bilateral pharyngoceles in a recreational trumpet player
Charles Daultrey1, Steve Colley2, Declan Costello3
1 Warwick Hospital - South Warwickshire NHS Foundation Trust, South Warwickshire, United Kingdom
2 Department of Radiology, University Hospital Birmingham, NHS Foundation Trust, Birmingham, United Kingdom
3 ENT, University Hospital Birmingham, NHS Foundation Trust, Birmingham, United Kingdom
|Date of Web Publication||24-Sep-2013|
Flat 2, The Garden House, 190 Harborne Road, Edgbaston, B15 3JJ Birmingham
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Pharyngocele is defined as "lateral pharyngeal wall herniation located in the piriform recess or at the valleculae" and was first described in 1886. The phenomenon is rare. It is characterised by bulging of the pharynx locally and is associated with recurrent increase in intrapharyngeal pressure, reduced muscle resilience, and increasing age. The case describes a 51-year-old recreational trumpeter with bilateral pharyngoceles that demonstrated when blowing into his instrument, reminiscent of a "bullfrog." Differential diagnosis included "Zenker's diverticulum", (pharyngeal pouch), laryngocele, and jugular venous phlebectasia, which may be confirmed through clinical examination and investigation. The report includes remarkable images seen on examination and images of the swelling seen with plain film radiography, barium swallow, and computed tomography. We discuss the management options available, including repair and excision of the pouch, either as an open procedure or endoscopically, and explore the conservative measures as a basis in the management of even significant pharyngoceles.
Keywords: Bilateral, images, clinical findings, dehiscence of pharyngeal muscles, investigation, laryngocele, lateral pharyngeal diverticulum, management, pharyngocele
|How to cite this article:|
Daultrey C, Colley S, Costello D. "Doctor I have a frog in my throat": Bilateral pharyngoceles in a recreational trumpet player. J Laryngol Voice 2013;3:18-21
| Introduction|| |
Pharyngocele is defined as "lateral pharyngeal wall herniation located in the piriform recess or at the valleculae" and was first described by Wheeler et al. (1886) in a military officer who took great pride in his control of an entire military parade with only his voice.  Only a handful of cases have been reported in the last decade. The phenomenon is characterised by bulging of the pharynx locally and is associated with recurrent increase in intrapharyngeal pressure, reduced muscle resilience, and increasing age.
| Case Report|| |
We present the case of a 51-year-old recreational trumpeter, referred to Queen Elizabeth Hospital voice clinic with the alarming symptom of intermittent neck swelling. His symptoms were demonstrated when blowing into his instrument which resulted in his neck expanding dramatically, reminiscent of a "bullfrog," and it would settle when forced exhalation ceased. His only other symptom was intermittent regurgitation of food with occasional dysphagia, though this was infrequent and did not concern him significantly. He was otherwise fit and well, took no regular medication, and as mentioned earlier, was an active member of a band where he played the trumpet.
Examination in the clinic setting confirmed the "bullfrogging" of his neck with exhalation against pursed lips [Figure 1], although neck and mouth examination at rest was normal. Flexible nasendoscopy was normal at rest, though it showed clear ballooning of the piriform fossa during trumpet manoeuvre [Figure 2].
Further investigations with barium swallow and computed tomography (CT) neck were conducted, which confirmed bilateral pharyngoceles. Neck X-ray confirmed the presence of copious air filling the pharynx during Valsalva manoeuvre, most notably on the left side [Figure 3]. Barium swallow revealed pooling of contrast in the piriform fossa and interestingly revealed the presence of a tight cricopharyngeal bar, which may have been the initial cause of the raised intrapharyngeal pressures and thus had given rise to the defect [Figure 4] and [Figure 5]. CT scanning further confirmed the extent of the defect and its extension within the neck spaces [Figure 6] and [Figure 7].
|Figure 3: Cervical X-ray revealing "bullfrogging" of the neck during Valsalva|
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|Figure 4: Lateral view of barium swallow highlighting prominent cricopharyngeal bar|
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|Figure 5: Anterior view of barium swallow highlighting contrast pooling in left piriform fossa|
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Multiple discussions regarding management were undertaken including conservative option of watch and wait, and medical management with anti-reflux medication to prevent symptoms and to relax cricopharyngeal tension secondary to possible reflux irritation. In fact, the patient managed his symptoms by tying a scarf tightly around his neck when playing the trumpet. Surgical intervention would either aim to release the cricopharyngeal hypertrophy, through endoscopic botox injection or stapled division of the bar, or could include external approach and mesh repair to the defective pharyngeal musculature.
Choice was made to continue with conservative management along with medical treatment of reflux, as the patient's main complaint was concern about the neck swelling and whether he could continue with his trumpet playing. At the last clinic review, he was managing very well and his amateur trumpet playing career was a great success, with him still utilising the scarf as a form of "truss" during playing.
| Discussion|| |
The condition is rare and is characterised by herniation of pharyngeal mucosa through the musculature of the tonsillar fossa, valleculae, or piriform fossa, confounded by mechanical factors and weakness in individuals.  This is especially true in those with regular increased intrapharyngeal pressures, such as glassblowers and wind instrument players, as is the case reported here.
Differential diagnosis must not be confused with "Zenker's diverticulum," herniation of the pharyngeal mucosa between thyropharyngeus and cricopharyngeus, which does not classically expand with raised intrapharyngeal pressure. Laryngocele also forms a possible differential, though this refers to herniation of the laryngeal mucosa through the laryngeal ventricle and is obvious on clinical examination.  Jugular venous phlebectasia may present with similar symptomatic features, with the neck bulging during Valsalva manoeuvre, though clinical examination and nasendscopy can again quickly confirm such a diagnosis. 
True lateral diverticula may be classified into congenital and acquired. Congenital diverticula are believed to be vestiges of the pharyngeal pouches, part of the branchial apparatus in the 4-6 week embryo. Most of these arise from a branchial cleft remnant, commonly second, which opens into the pharynx, at the lower pole of the tonsillar fossa by the posterior faucial pillar.  Acquired diverticula, although rare, are reported more frequently in the literature and are more common in situations of prolonged raised intrapharyngeal pressure, historically found in glass blowers and wind instrument players, as is the case in our report, and was recently reported by Fakhouri et al. in a 32-year-old professional military musician. 
Pharyngoceles are rare, generally occurring in the fifth or sixth decade of life, though they are reported in young adults. In 1979, Norris reported 24 cases of pharyngocele over a 2-year series, with a range of 31-75 years, mean age of 51.8 years, and median of 54, and a strong male predilection of 8:1.  Most of the cases in the series were unilateral; also, in their short series in the year 2011, Brazilian authors Porcaro-Salles et al. reported three cases of unilateral pharyngocele.  Bilateral cases are also reported; Martinez et al. present a case of a 71-year-old male with chance discovery of bilateral pharyngoceles. 
The diagnosis, which may be clinically difficult, can be made with varied radiological and ultrasonographic techniques, though clinical examination may be satisfactory. Chevallier et al. report a lateral cervical neck swelling in a 25 year old, which, on ultrasonography, demonstrated communicating and extending into the piriform fossa, also deforming the thyroid cartilage lamina. 
Our case was managed without surgery and highlights the fact that whilst clinically and radiologically diverticula may appear significant, treating the patient's main concerns and symptoms provided an excellent outcome. Use of a "truss" in the management is not widely described in the literature, though use of a scarf in this case gave excellent symptomatic control during ballooning whilst playing. Interestingly, pharyngoceles were noted in Egyptian Muezzins who sang verses from the Koran from the minarets of mosques. Many were described as developing pharyngoceles, which often became so marked that they required specially designed collars to restrain them. 
Surgical options are reported in the literature and generally involve external approach excision, though with our patient, endoscopic approach to cricopharyngeus release was discussed. Surgery selection requires discussion with the patient and balancing symptomatic relief against the risks of operating, be it as open procedure or endoscopically. Recurrence after surgery also poses a potential complication, as reported by Marcotullio et al. in 2008, highlighting the case of bilateral pharyngoceles that recurred 8 years after surgical repair.  Good oral hygiene, healthy diet, anti-reflux medication, and speech and language team involvement are reasonable conservative measures in the management of even significant pharyngoceles.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]