AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI N E AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
Available online at www.sciencedirect.com
ScienceDirect www.elsevier.com/locate/amjoto
Airway management in an infant with a giant vallecular cyst David A. Reiersen⁎, Anil A. Gungor LSU-Health Shreveport Department of Otolaryngology/Head & Neck Surgery
ARTI CLE I NFO
A BS TRACT
Article history:
Review vallecular cysts and report the surgical management of a vallecular cyst of unusual
Received 11 October 2013
size with near-complete obliteration of the airway. This case report describes an unusually large mucus retention cyst in an 8-week-old infant that was diagnosed during induction of general anesthesia and prevented visualization of airway. Intubation was performed after rapid aspiration of the cyst contents. Conclusion: Vallecular cysts can present as a life-threatening obstruction in infants and complicate the establishment of an airway after induction of anesthesia. In our case, rapid thinking and aspiration of the cyst contents helped establish the airway for definitive CO2 laser excision. © 2014 Elsevier Inc. All rights reserved.
1.
Introduction
Vallecular cysts are a rare entity and typically present with respiratory and feeding difficulties due to obstruction and pressure at the laryngeal inlet. The difficulties may manifest as inspiratory stridor, chest wall retraction, increase work-of-breathing, apneas, cyanosis, and failure to thrive. Large cysts are more likely to be diagnosed at younger ages because larger cysts are more likely to cause symptoms that will lead to subsequent diagnostic work-up and treatment. Large vallecular cysts have also been described in the literature to be diagnosed at the time of induction of general anesthesia resulting in a difficult endotracheal intubation due to obstruction of the glottis. Adult vallecular cysts have been described as asymptomatic. When seen in adults the symptoms are subtle and include globus and voice changes. The contrasting difference in symptoms owes itself to the difference in the relative size of the airway [1]. A vallecular cyst has been classified as a ductal cyst by DeSanto in 1970. The ductal cyst, also known as mucus retention cysts, results from retention of mucus within an obstructed collecting duct of a submucosal gland. Ductal cysts are superficial and can occur at any site within the
laryngopharynx (excluding the free edge of the vocal fold) due to the presence of mucosal glands. Most ductal cysts are < 1 cm diameter however they may be significantly larger. The cells of the cyst are of the ductal cell type (squamous or respiratory epithelium) and not of acinar cells (supporting origin from dilation of ducts and not of distended glands) [2]. Ductal cysts at the tongue base and vallecula are the most common in children [3]. DeSanto described that among the ductal cysts found about the epiglottis, 48 of the 67 cysts occurred at the lingual surface of the epiglottis. These cyst are superficial, resulting from obstruction of the submucosal glands, and are confined to the submucosal layer. Mucus production results in further expansion of the ductal cyst. Symptoms result from direct obstruction of the airway directly or from distortion of the epiglottis and the laryngeal inlet [1]. Vallecular cysts have been described to have a high incidence of recurrence with simple needle aspiration. Mitchel in 1987 described 3 of 3 cyst recurring following simple aspiration. Vallecular cysts are typically removed by opening widely to prevent recurrence [4]. Carbon dioxide laser ablation of the inner epithelial lining of the cyst wall has been described to reduce risk of recurrence [3,5]. Patients are
⁎ Corresponding author. E-mail address: [email protected] (D.A. Reiersen). 0196-0709/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.amjoto.2013.11.001
262
AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
Fig. 1 – Vallecular cyst as visualized during flexible laryngoscopic exam (A). Vallecular cyst visualized by direct laryngoscopic exam (B).
generally described to have an immediate improvement in symptoms and post-operative care is limited to that needed for the patient’s comorbid conditions (such and reflux or laryngomalacia). Of the associated anomalies, laryngomalacia is the most common [1].
2.
Case report
An 8 week old male infant presented to the emergency room with failure to thrive, weak cry, progressive inspiratory stridor,
Fig. 2 – After needle aspiration and intubation the cyst was further opened and drained of its contents (A-D).
AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI N E AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
263
Fig. 3 – The open cyst was fully marsupialized and cyst wall was removed with use of CO2 laser (A-D).
and respiratory distress. Prior medical history was significant for reflux and suspected laryngomalacia. The patient was taken back to the operating room for an airway evaluation due to the above findings. The patient underwent flexible fiberoptic laryngoscopy which revealed a large cystic lesion of the vallecular that was mobile and obstructing visualization of the airway beyond the oropharynx (Fig. 1A). Direct laryngoscopy and bronchoscopy were utilized to evaluate the extent of the cystic lesion (Fig. 1B).
Due to the size of the vallecular cyst it was difficult ventilating the patient. This required immediate aspiration of the cyst contents with an 18 gauge needle prior to securing the airway. The patient was then suspended with Infant Lindholm laryngoscope to fully address the vallecular cyst with biopsy and marsupialization of the cyst wall with a CO2 laser (Figs. 2-4). Post-operatively the patient experienced immediate relief from the airway obstruction. Following the procedure, the
Fig. 4 – The floor of the cyst was ablated with CO2 laser to prevent recurrence (A-C).
264
AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
patient displayed no signs of respiratory distress and feeding habits normalized. At two month follow-up, the child’s weight had increased from below the 3rd percentile to the 50th percentile. Follow-up flexible laryngoscopic examination revealed a normal airway with no evidence of recurrence or other abnormality at 2 month follow-up.
3.
Discussion
Thorough airway evaluation is indicated in an infant with upper airway symptoms such as weak cry and stridor. This is especially true when symptoms are coincided with difficulty feeding and failure to thrive. When performing an evaluation of the airway it is important to expect difficulty with the airway and be prepared to overcome lesions that may make bag mask ventilation and endotracheal intubation difficult. In this case, preparation and quick response allowed for successful aspiration of the cyst contents and safe intubation
which was necessary to subsequently treat the condition of a large vallecular cyst that was obstructing and distorting the laryngeal inlet.
REFERENCES
[1] Hsieh WS, Yang PH, Wong KS, Li HY. Vallecular cyst: an uncommon cause of stridor in newborn infants. Eur J Pediatr 2000;159:79–81. [2] DeSanto LW, Devine KD, Weiland LH. Cysts of the larynx— classification. Laryngoscope 2009;80:145–76. [3] Wong KS, Li HY, Huang TS. Vallecular cyst synchronous with laryngomalacia: presentation of two cases. Otolaryngol Head Neck Surg 1995;113:621–4. [4] Mitchell DB, Irwin BC, Bailey CM, et al. Cysts of the infant larynx. J Laryngol Otol 1987;101:833–7. [5] Ku AS. Vallecular cyst: report of four cases-one with co-existing laryngomalacia. J Laryngol Otol 2000;114: 224–6.
Available online at www.sciencedirect.com
ScienceDirect www.elsevier.com/locate/amjoto
Airway management in an infant with a giant vallecular cyst David A. Reiersen⁎, Anil A. Gungor LSU-Health Shreveport Department of Otolaryngology/Head & Neck Surgery
ARTI CLE I NFO
A BS TRACT
Article history:
Review vallecular cysts and report the surgical management of a vallecular cyst of unusual
Received 11 October 2013
size with near-complete obliteration of the airway. This case report describes an unusually large mucus retention cyst in an 8-week-old infant that was diagnosed during induction of general anesthesia and prevented visualization of airway. Intubation was performed after rapid aspiration of the cyst contents. Conclusion: Vallecular cysts can present as a life-threatening obstruction in infants and complicate the establishment of an airway after induction of anesthesia. In our case, rapid thinking and aspiration of the cyst contents helped establish the airway for definitive CO2 laser excision. © 2014 Elsevier Inc. All rights reserved.
1.
Introduction
Vallecular cysts are a rare entity and typically present with respiratory and feeding difficulties due to obstruction and pressure at the laryngeal inlet. The difficulties may manifest as inspiratory stridor, chest wall retraction, increase work-of-breathing, apneas, cyanosis, and failure to thrive. Large cysts are more likely to be diagnosed at younger ages because larger cysts are more likely to cause symptoms that will lead to subsequent diagnostic work-up and treatment. Large vallecular cysts have also been described in the literature to be diagnosed at the time of induction of general anesthesia resulting in a difficult endotracheal intubation due to obstruction of the glottis. Adult vallecular cysts have been described as asymptomatic. When seen in adults the symptoms are subtle and include globus and voice changes. The contrasting difference in symptoms owes itself to the difference in the relative size of the airway [1]. A vallecular cyst has been classified as a ductal cyst by DeSanto in 1970. The ductal cyst, also known as mucus retention cysts, results from retention of mucus within an obstructed collecting duct of a submucosal gland. Ductal cysts are superficial and can occur at any site within the
laryngopharynx (excluding the free edge of the vocal fold) due to the presence of mucosal glands. Most ductal cysts are < 1 cm diameter however they may be significantly larger. The cells of the cyst are of the ductal cell type (squamous or respiratory epithelium) and not of acinar cells (supporting origin from dilation of ducts and not of distended glands) [2]. Ductal cysts at the tongue base and vallecula are the most common in children [3]. DeSanto described that among the ductal cysts found about the epiglottis, 48 of the 67 cysts occurred at the lingual surface of the epiglottis. These cyst are superficial, resulting from obstruction of the submucosal glands, and are confined to the submucosal layer. Mucus production results in further expansion of the ductal cyst. Symptoms result from direct obstruction of the airway directly or from distortion of the epiglottis and the laryngeal inlet [1]. Vallecular cysts have been described to have a high incidence of recurrence with simple needle aspiration. Mitchel in 1987 described 3 of 3 cyst recurring following simple aspiration. Vallecular cysts are typically removed by opening widely to prevent recurrence [4]. Carbon dioxide laser ablation of the inner epithelial lining of the cyst wall has been described to reduce risk of recurrence [3,5]. Patients are
⁎ Corresponding author. E-mail address: [email protected] (D.A. Reiersen). 0196-0709/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.amjoto.2013.11.001
262
AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
Fig. 1 – Vallecular cyst as visualized during flexible laryngoscopic exam (A). Vallecular cyst visualized by direct laryngoscopic exam (B).
generally described to have an immediate improvement in symptoms and post-operative care is limited to that needed for the patient’s comorbid conditions (such and reflux or laryngomalacia). Of the associated anomalies, laryngomalacia is the most common [1].
2.
Case report
An 8 week old male infant presented to the emergency room with failure to thrive, weak cry, progressive inspiratory stridor,
Fig. 2 – After needle aspiration and intubation the cyst was further opened and drained of its contents (A-D).
AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI N E AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
263
Fig. 3 – The open cyst was fully marsupialized and cyst wall was removed with use of CO2 laser (A-D).
and respiratory distress. Prior medical history was significant for reflux and suspected laryngomalacia. The patient was taken back to the operating room for an airway evaluation due to the above findings. The patient underwent flexible fiberoptic laryngoscopy which revealed a large cystic lesion of the vallecular that was mobile and obstructing visualization of the airway beyond the oropharynx (Fig. 1A). Direct laryngoscopy and bronchoscopy were utilized to evaluate the extent of the cystic lesion (Fig. 1B).
Due to the size of the vallecular cyst it was difficult ventilating the patient. This required immediate aspiration of the cyst contents with an 18 gauge needle prior to securing the airway. The patient was then suspended with Infant Lindholm laryngoscope to fully address the vallecular cyst with biopsy and marsupialization of the cyst wall with a CO2 laser (Figs. 2-4). Post-operatively the patient experienced immediate relief from the airway obstruction. Following the procedure, the
Fig. 4 – The floor of the cyst was ablated with CO2 laser to prevent recurrence (A-C).
264
AM ER IC AN JOUR NA L OF OTOLARY NG OLOG Y –H EA D A N D N E CK ME D I CI NE AN D SUR G E RY 3 5 ( 2 0 14 ) 26 1–2 6 4
patient displayed no signs of respiratory distress and feeding habits normalized. At two month follow-up, the child’s weight had increased from below the 3rd percentile to the 50th percentile. Follow-up flexible laryngoscopic examination revealed a normal airway with no evidence of recurrence or other abnormality at 2 month follow-up.
3.
Discussion
Thorough airway evaluation is indicated in an infant with upper airway symptoms such as weak cry and stridor. This is especially true when symptoms are coincided with difficulty feeding and failure to thrive. When performing an evaluation of the airway it is important to expect difficulty with the airway and be prepared to overcome lesions that may make bag mask ventilation and endotracheal intubation difficult. In this case, preparation and quick response allowed for successful aspiration of the cyst contents and safe intubation
which was necessary to subsequently treat the condition of a large vallecular cyst that was obstructing and distorting the laryngeal inlet.
REFERENCES
[1] Hsieh WS, Yang PH, Wong KS, Li HY. Vallecular cyst: an uncommon cause of stridor in newborn infants. Eur J Pediatr 2000;159:79–81. [2] DeSanto LW, Devine KD, Weiland LH. Cysts of the larynx— classification. Laryngoscope 2009;80:145–76. [3] Wong KS, Li HY, Huang TS. Vallecular cyst synchronous with laryngomalacia: presentation of two cases. Otolaryngol Head Neck Surg 1995;113:621–4. [4] Mitchell DB, Irwin BC, Bailey CM, et al. Cysts of the infant larynx. J Laryngol Otol 1987;101:833–7. [5] Ku AS. Vallecular cyst: report of four cases-one with co-existing laryngomalacia. J Laryngol Otol 2000;114: 224–6.
