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Bilateral External Laryngoceles Following Radioiodine Ablation for Graves Disease

Bilateral External Laryngoceles Following Radioiodine Ablation for Graves Disease A 28-year-old trumpet player underwent multiple treatments with radioactive iodine for Graves disease associated with an unusually large goiter. Following his second treatment, the patient developed acute neck pain and swelling. Radiographic studies and a laryngoscopy demonstrated bilateral symptomatic external laryngoceles, a very rare entity, not previously known to be associated with radioiodine treatment or Graves disease. The patient's profession placed him at risk for the development of a laryngocele, but the temporal relationship to goiter regression following radioiodine therapy suggests that this occurred as a result of this treatment. The patient's disease was managed nonsurgically, and he has subsequently done well. This represents the first known association of symptomatic laryngocele with radioiodine treatment for Graves disease.Laryngoceles are uncommon lesions of the larynx in which the appendix of the laryngeal ventricle is abnormally dilated. These lesions may be congenital or acquired and are very rarely symptomatic.Certain groups of individuals are at increased risk for developing acquired laryngoceles, such as glass blowers and professional wind instrument players,presumably a result of increased pressures within the laryngeal lumen. At this point, no clear precipitant for the formation of a laryngocele is known. In this report, we present a case of symptomatic bilateral external laryngoceles occurring in a patient following radioiodine treatment for Graves disease (GD) manifested by a very large goiter.REPORT OF A CASEA 28-year-old professional trumpet player presented to the Endocrine Clinic with several months of weight loss, tremors, palpitations, anxiety, and heat intolerance. He was diagnosed as having GD with an undetectable serum thyroid-stimulating hormone level, elevated free thyroxine, radioactive iodine uptake of 67.5% at 24 hours, and homogeneous technetium Tc 99m scan. His examination demonstrated a large goiter, estimated by palpation at a size in excess of 100 g. He was treated initially with propylthiouracil and subsequently with 10 mCi (370 MBq) of idodine 131. Roughly 8 months later, he continued to be thyrotoxic and was treated with an additional 14 mCi (518 MBq) of iodine 131.Approximately 4 weeks after the second dose of idodine 131, he noticed the sudden onset of anterior neck pain while playing the trumpet. Whenever he played his trumpet subsequently, he experienced neck discomfort and a sensation of a foreign body on both sides of his throat. He modified his playing technique, which alleviated his symptoms. Following this second radioiodine treatment, the patient became hypothyroid and was initiated on thyroid hormone replacement. After several more months, he began to complain of occasional dysphagia, and presented again for evaluation. The neck examination at that time demonstrated a much smaller thyroid, estimated at 15 g. When the patient performed a Valsalva maneuver, an anterior neck mass was clearly visible (Figure 1).Figure 1.External view of patient's neck while playing trumpet, demonstrating external laryngocele.Plain films of the neck were obtained following a barium swallow with the patient performing a Valsalva maneuver (Figure 2and Figure 3), demonstrating bilateral air-filled sacs on either side of the larynx. These sacs were believed to be consistent with bilateral laryngoceles. A laryngoscopy was subsequently performed, demonstrating no internal component of the laryngoceles and no lesions suggestive of laryngeal cancer.Figure 2.Frontal plain radiograph of the neck showing bilateral air-filled laryngoceles during Valsalva maneuver.Figure 3.Lateral plain film of the neck demonstrating air-filled laryngocele during Valsalva and following barium swallow (no contrast medium).Surgery was not pursued in this patient since his symptoms were mild and he was able to avoid expansion of the laryngoceles by modifying his trumpet-playing technique.COMMENTAccording to Ojala,the first known recorded description of a laryngocele was in 1829 by Larrey, Napoleon's army doctor in Egypt, who observed large swellings in the necks of criers chanting the Koran from atop towers for hours on end. Virchow, in 1867, described an abnormal dilation of the saccule of the larynx in a patient with a condition he termed laryngocele ventricularis.A laryngocele represents an abnormal air-filled dilation of the saccule of the lateral ventricle of the larynx that communicates with the lumen of the larynx.The lateral ventricle of the larynx is a fossa extending between the true and false vocal cords. At the anterior end of this ventricle is a saccule that forms the laryngeal appendix, which extends upward between the vestibular fold and the thyroid cartilage. When this appendix dilates to an abnormal size (usually greater than 10 mm), or extends above the border of the thyroid cartilage, it is considered a laryngocele.The laryngocele may also become filled with mucus, which may become infected, termed a laryngopyocele.The laryngeal appendix is present at birth, and in childhood is relatively large. It begins to regress by the end of the sixth year.The etiology of laryngoceles in adults remains somewhat controversial, thought to be either a enlargement of the saccule following repeated pressures enlarging a larger than average saccule or an isolated incident enlarging a congenitally large saccule to its full size.A laryngocele may be defined as internal, external, or mixed. A laryngocele is internal when it lies within the thyroid cartilage,external when it protrudes through the thyrohyoid membrane, and mixed when it has properties of both.Mixed laryngoceles are the most common (44%-50%), followed by internal (30%) and external (20%-26%) laryngoceles.An external laryngocele penetrates through the thyrohyoid membrane near the path defined by the superior laryngeal artery and the internal laryngeal nerve.Symptomatic laryngoceles are a rare disease with an annual incidence of 1 in 2 500 000, of which 85% are unilateral and only 15% bilateral.Asymptomatic laryngoceles are thought to be more common, with autopsy rates of internal laryngoceles being as high as 6%.While the vast majority of laryngoceles are asymptomatic, the most common symptoms that occur are hoarseness, dyspnea, coughing, dysphagia, a globus sensation in the throat, and a lateral neck mass.When the patient performs a Valsalva maneuver, the mass should increase in size. On compressing of the swelling, a hissing sign may be heard, known as Bryce's sign.Our patient presented with the acute onset of this very unusual combination of symptomatic bilateral external laryngoceles. The timing of this following treatment with radioiodine for a large goiter suggests a relationship between the two. Specifically, we postulated that the while the patient's profession predisposed him to the development of bilateral laryngoceles, their lateral expansion was prevented by his large goiter. Treatment with radioiodine then resulted in goiter regression, allowing the overt expansion of these structures. Another interesting hypothesis is that the improved respiratory muscle tone following treatment of the patient's hyperthyroidism allowed more vigorous trumpet playing, which in turn could have caused or exacerbated his laryngoceles. Yet another less likely possibility is that the patient's radioiodine therapy directly affected the compliance of the thyrohyoid membrane through which laryngoceles must expand. Radioiodine therapy for thyrotoxicosis has been known rarely to damage other extrathyroidal neck structures such as the parathyroid glandsand the recurrent laryngeal nerves.The association of laryngoceles in brass instrument players is well known,and almost certainly played a role in our patient. Macfiereported a series of 54 wind instrument bandsmen. Under Valsalva maneuver, 56% demonstrated laryngoceles; 31% were internal and the remaining 69% were mixed. It is worth noting that while most of the patients in this series had laryngoceles, none had external laryngoceles, favoring the concept that our patient had preexisting laryngoceles, but that another factor contributed to these becoming both external and symptomatic.Even with the high likelihood that the laryngoceles were related to his occupational hazard rather than a laryngeal tumor, our patient underwent a laryngoscopy to evaluate for laryngeal cancer. There is an association between laryngoceles and squamous cell carcinoma of the larynx, with a reported incidence of 5% to 28% of patients with laryngoceles.Nearly all patients diagnosed as having laryngoceles should undergo computed tomographic evaluationor laryngoscopy, possibly with biopsy, given this association.Because our patient had few symptoms and was able to modify his trumpeting technique to avoid the formation of laryngoceles, surgery was avoided. Following diagnosis and evaluation with appropriate radiological studies and/or laryngoscopy, minimally symptomatic laryngoceles can be managed conservatively with periodic observation.When surgical intervention is required, laryngoceles can be treated by endoscopic laser marsupialization or surgical excision through an external surgical approach.JRutkaDBirtLaryngocele: a case report and review.J Otolaryngol.1983;12:389-392.GKumarPJBradleyMLWastieCase of the month: what a blow! laryngocele.Br J Radiol.1998;71:799-800.CHubbardLaryngocele—a study of five cases with reference to the radiological features.Clin Radiol.1987;38:639-643.DDMacfieAsymptomatic laryngoceles in wind-instrument bandsmen.Arch Otolaryngol.1966;83:270-275.SEversHHenningsenEBRingelsteinTransient ischemic attacks caused by trumpet playing.Neurology.1998;51:1709-1710.KOjalaExternal, asymptomatic laryngocele without known predisposing factors in a middle-aged man.J Laryngol Otol.1983;97:767-769.MJBabbBMRasgonQuiz case 2: bilateral laryngocele.Arch Otolaryngol Head Neck Surg.2000;126:551, 552-554.KRKattanMAZaheerLaryngocele: radiological diagnosis.JAMA.1980;244:1617-1619.RFCanalisObservations on the simultaneous occurrence of laryngocele and cancer.J Otolaryngol.1976;5:207-212.JBallantyneScott-Browns Diseases of the Ear, Nose, and Throat.5th ed. Vol 5. London, England: Butterworth Press; 1987.PMStellAGMaranLaryngocoele.J Laryngol Otol.1975;89:915-924.DMThomasGJMaddenBilateral laryngoceles.Ear Nose Throat J.1993;72:819-821.DMendelsohnYHertzanuRBJGlassComputed tomographic appearance of laryngoceles.S Afr Med J.1984;65:281-282.PBvan VierzenFBJoostenJJManniSonographic, MR and CT findings in a large laryngocele: a case report.Eur J Radiol.1994;18:45-47.SThawleyCysts and tumors of the larynx.In: Zorab R, ed. Otolaryngology.Philadelphia, Pa: WB Saunders Co; 1991:2307-2368.IBRosenJAPalmerJRowenSCLukInduction of hyperparathyroidism by radioactive iodine.Am J Surg.1984;148:441-445.CPWinslowADMeyersHypocalcemia as a complication of radioiodine therapy.Am J Otolaryngol.1998;19:401-403.LRCooverPermanent iatrogenic vocal cord paralysis after I-131 therapy: a case report and literature review.Clin Nucl Med.2000;25:508-510.JCSniezekREJohnsonSGRamirezDKHayesLaryngoceles and saccular cysts.South Med J.1996;89:427-430.Accepted for publication March 20, 2002.The opinions expressed in this article reflect the views of the authors and not the official views of the US Army or the Department of Defense.Corresponding author and reprints: Derek J. Stocker, MD, Endocrine-Metabolic Service, Walter Reed Army Medical Center, Department of Medicine, Washington, DC 20307-5001. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png JAMA Internal Medicine American Medical Association

Bilateral External Laryngoceles Following Radioiodine Ablation for Graves Disease

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American Medical Association
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Copyright 2002 American Medical Association. All Rights Reserved. Applicable FARS/DFARS Restrictions Apply to Government Use.
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2168-6106
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Abstract

A 28-year-old trumpet player underwent multiple treatments with radioactive iodine for Graves disease associated with an unusually large goiter. Following his second treatment, the patient developed acute neck pain and swelling. Radiographic studies and a laryngoscopy demonstrated bilateral symptomatic external laryngoceles, a very rare entity, not previously known to be associated with radioiodine treatment or Graves disease. The patient's profession placed him at risk for the development of a laryngocele, but the temporal relationship to goiter regression following radioiodine therapy suggests that this occurred as a result of this treatment. The patient's disease was managed nonsurgically, and he has subsequently done well. This represents the first known association of symptomatic laryngocele with radioiodine treatment for Graves disease.Laryngoceles are uncommon lesions of the larynx in which the appendix of the laryngeal ventricle is abnormally dilated. These lesions may be congenital or acquired and are very rarely symptomatic.Certain groups of individuals are at increased risk for developing acquired laryngoceles, such as glass blowers and professional wind instrument players,presumably a result of increased pressures within the laryngeal lumen. At this point, no clear precipitant for the formation of a laryngocele is known. In this report, we present a case of symptomatic bilateral external laryngoceles occurring in a patient following radioiodine treatment for Graves disease (GD) manifested by a very large goiter.REPORT OF A CASEA 28-year-old professional trumpet player presented to the Endocrine Clinic with several months of weight loss, tremors, palpitations, anxiety, and heat intolerance. He was diagnosed as having GD with an undetectable serum thyroid-stimulating hormone level, elevated free thyroxine, radioactive iodine uptake of 67.5% at 24 hours, and homogeneous technetium Tc 99m scan. His examination demonstrated a large goiter, estimated by palpation at a size in excess of 100 g. He was treated initially with propylthiouracil and subsequently with 10 mCi (370 MBq) of idodine 131. Roughly 8 months later, he continued to be thyrotoxic and was treated with an additional 14 mCi (518 MBq) of iodine 131.Approximately 4 weeks after the second dose of idodine 131, he noticed the sudden onset of anterior neck pain while playing the trumpet. Whenever he played his trumpet subsequently, he experienced neck discomfort and a sensation of a foreign body on both sides of his throat. He modified his playing technique, which alleviated his symptoms. Following this second radioiodine treatment, the patient became hypothyroid and was initiated on thyroid hormone replacement. After several more months, he began to complain of occasional dysphagia, and presented again for evaluation. The neck examination at that time demonstrated a much smaller thyroid, estimated at 15 g. When the patient performed a Valsalva maneuver, an anterior neck mass was clearly visible (Figure 1).Figure 1.External view of patient's neck while playing trumpet, demonstrating external laryngocele.Plain films of the neck were obtained following a barium swallow with the patient performing a Valsalva maneuver (Figure 2and Figure 3), demonstrating bilateral air-filled sacs on either side of the larynx. These sacs were believed to be consistent with bilateral laryngoceles. A laryngoscopy was subsequently performed, demonstrating no internal component of the laryngoceles and no lesions suggestive of laryngeal cancer.Figure 2.Frontal plain radiograph of the neck showing bilateral air-filled laryngoceles during Valsalva maneuver.Figure 3.Lateral plain film of the neck demonstrating air-filled laryngocele during Valsalva and following barium swallow (no contrast medium).Surgery was not pursued in this patient since his symptoms were mild and he was able to avoid expansion of the laryngoceles by modifying his trumpet-playing technique.COMMENTAccording to Ojala,the first known recorded description of a laryngocele was in 1829 by Larrey, Napoleon's army doctor in Egypt, who observed large swellings in the necks of criers chanting the Koran from atop towers for hours on end. Virchow, in 1867, described an abnormal dilation of the saccule of the larynx in a patient with a condition he termed laryngocele ventricularis.A laryngocele represents an abnormal air-filled dilation of the saccule of the lateral ventricle of the larynx that communicates with the lumen of the larynx.The lateral ventricle of the larynx is a fossa extending between the true and false vocal cords. At the anterior end of this ventricle is a saccule that forms the laryngeal appendix, which extends upward between the vestibular fold and the thyroid cartilage. When this appendix dilates to an abnormal size (usually greater than 10 mm), or extends above the border of the thyroid cartilage, it is considered a laryngocele.The laryngocele may also become filled with mucus, which may become infected, termed a laryngopyocele.The laryngeal appendix is present at birth, and in childhood is relatively large. It begins to regress by the end of the sixth year.The etiology of laryngoceles in adults remains somewhat controversial, thought to be either a enlargement of the saccule following repeated pressures enlarging a larger than average saccule or an isolated incident enlarging a congenitally large saccule to its full size.A laryngocele may be defined as internal, external, or mixed. A laryngocele is internal when it lies within the thyroid cartilage,external when it protrudes through the thyrohyoid membrane, and mixed when it has properties of both.Mixed laryngoceles are the most common (44%-50%), followed by internal (30%) and external (20%-26%) laryngoceles.An external laryngocele penetrates through the thyrohyoid membrane near the path defined by the superior laryngeal artery and the internal laryngeal nerve.Symptomatic laryngoceles are a rare disease with an annual incidence of 1 in 2 500 000, of which 85% are unilateral and only 15% bilateral.Asymptomatic laryngoceles are thought to be more common, with autopsy rates of internal laryngoceles being as high as 6%.While the vast majority of laryngoceles are asymptomatic, the most common symptoms that occur are hoarseness, dyspnea, coughing, dysphagia, a globus sensation in the throat, and a lateral neck mass.When the patient performs a Valsalva maneuver, the mass should increase in size. On compressing of the swelling, a hissing sign may be heard, known as Bryce's sign.Our patient presented with the acute onset of this very unusual combination of symptomatic bilateral external laryngoceles. The timing of this following treatment with radioiodine for a large goiter suggests a relationship between the two. Specifically, we postulated that the while the patient's profession predisposed him to the development of bilateral laryngoceles, their lateral expansion was prevented by his large goiter. Treatment with radioiodine then resulted in goiter regression, allowing the overt expansion of these structures. Another interesting hypothesis is that the improved respiratory muscle tone following treatment of the patient's hyperthyroidism allowed more vigorous trumpet playing, which in turn could have caused or exacerbated his laryngoceles. Yet another less likely possibility is that the patient's radioiodine therapy directly affected the compliance of the thyrohyoid membrane through which laryngoceles must expand. Radioiodine therapy for thyrotoxicosis has been known rarely to damage other extrathyroidal neck structures such as the parathyroid glandsand the recurrent laryngeal nerves.The association of laryngoceles in brass instrument players is well known,and almost certainly played a role in our patient. Macfiereported a series of 54 wind instrument bandsmen. Under Valsalva maneuver, 56% demonstrated laryngoceles; 31% were internal and the remaining 69% were mixed. It is worth noting that while most of the patients in this series had laryngoceles, none had external laryngoceles, favoring the concept that our patient had preexisting laryngoceles, but that another factor contributed to these becoming both external and symptomatic.Even with the high likelihood that the laryngoceles were related to his occupational hazard rather than a laryngeal tumor, our patient underwent a laryngoscopy to evaluate for laryngeal cancer. There is an association between laryngoceles and squamous cell carcinoma of the larynx, with a reported incidence of 5% to 28% of patients with laryngoceles.Nearly all patients diagnosed as having laryngoceles should undergo computed tomographic evaluationor laryngoscopy, possibly with biopsy, given this association.Because our patient had few symptoms and was able to modify his trumpeting technique to avoid the formation of laryngoceles, surgery was avoided. Following diagnosis and evaluation with appropriate radiological studies and/or laryngoscopy, minimally symptomatic laryngoceles can be managed conservatively with periodic observation.When surgical intervention is required, laryngoceles can be treated by endoscopic laser marsupialization or surgical excision through an external surgical approach.JRutkaDBirtLaryngocele: a case report and review.J Otolaryngol.1983;12:389-392.GKumarPJBradleyMLWastieCase of the month: what a blow! laryngocele.Br J Radiol.1998;71:799-800.CHubbardLaryngocele—a study of five cases with reference to the radiological features.Clin Radiol.1987;38:639-643.DDMacfieAsymptomatic laryngoceles in wind-instrument bandsmen.Arch Otolaryngol.1966;83:270-275.SEversHHenningsenEBRingelsteinTransient ischemic attacks caused by trumpet playing.Neurology.1998;51:1709-1710.KOjalaExternal, asymptomatic laryngocele without known predisposing factors in a middle-aged man.J Laryngol Otol.1983;97:767-769.MJBabbBMRasgonQuiz case 2: bilateral laryngocele.Arch Otolaryngol Head Neck Surg.2000;126:551, 552-554.KRKattanMAZaheerLaryngocele: radiological diagnosis.JAMA.1980;244:1617-1619.RFCanalisObservations on the simultaneous occurrence of laryngocele and cancer.J Otolaryngol.1976;5:207-212.JBallantyneScott-Browns Diseases of the Ear, Nose, and Throat.5th ed. Vol 5. London, England: Butterworth Press; 1987.PMStellAGMaranLaryngocoele.J Laryngol Otol.1975;89:915-924.DMThomasGJMaddenBilateral laryngoceles.Ear Nose Throat J.1993;72:819-821.DMendelsohnYHertzanuRBJGlassComputed tomographic appearance of laryngoceles.S Afr Med J.1984;65:281-282.PBvan VierzenFBJoostenJJManniSonographic, MR and CT findings in a large laryngocele: a case report.Eur J Radiol.1994;18:45-47.SThawleyCysts and tumors of the larynx.In: Zorab R, ed. Otolaryngology.Philadelphia, Pa: WB Saunders Co; 1991:2307-2368.IBRosenJAPalmerJRowenSCLukInduction of hyperparathyroidism by radioactive iodine.Am J Surg.1984;148:441-445.CPWinslowADMeyersHypocalcemia as a complication of radioiodine therapy.Am J Otolaryngol.1998;19:401-403.LRCooverPermanent iatrogenic vocal cord paralysis after I-131 therapy: a case report and literature review.Clin Nucl Med.2000;25:508-510.JCSniezekREJohnsonSGRamirezDKHayesLaryngoceles and saccular cysts.South Med J.1996;89:427-430.Accepted for publication March 20, 2002.The opinions expressed in this article reflect the views of the authors and not the official views of the US Army or the Department of Defense.Corresponding author and reprints: Derek J. Stocker, MD, Endocrine-Metabolic Service, Walter Reed Army Medical Center, Department of Medicine, Washington, DC 20307-5001.

Journal

JAMA Internal MedicineAmerican Medical Association

Published: Sep 23, 2002

References