|
| |
 |
| CASE REPORT |
|
| Year : 2021 | Volume
: 11
| Issue : 1 | Page : 17-20 |
|
Early-onset respiratory distress in a child: A rare presentation
Aishwarya Prateep1, Gowri Priyadharshini Vijayaraj1, Aruna Parameswari2, Prasanna Kumar Saravanam1
1 Department of ENT, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamilnadu, India
2 Department of Anesthesia, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamilnadu, India
| Date of Submission | 14-Oct-2020 |
| Date of Acceptance | 11-Apr-2021 |
| Date of Web Publication | 5-Jul-2021 |
Correspondence Address:
Prasanna Kumar Saravanam
Department of Otorhinolaryngology and Head and Neck Surgery, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jlv.jlv_14_20
 Abstract | | |
A stridulous child with failure to thrive needs to be evaluated and emergent management is required to reduce the morbidity and mortality. The reasons for stridor in early childhood are usually due to laryngomalacia. Laryngeal cyst can pose a potential risk and challenge in management, especially in children. Laryngeal cysts have been described to occur in the glottis, laryngeal pouch, epiglottis, aryepiglottic folds, and subglottic areas. Clinical features of the laryngeal cyst depend on the size and localization of the cyst. They may present with voice change, hoarseness, dysphagia, laryngeal stridor, and dyspnea. Prompt diagnosis and early management can prevent major respiratory compromise. This case reports a 6-month old infant who was referred to us with stridor and failure to thrive since birth. The child was evaluated and a diagnosis of aryepiglottic fold cyst was made and treated surgically. The management protocol and available literature are reviewed and discussed.
Keywords: Failure to thrive, laryngeal cyst, respiratory compromise, stridor
How to cite this article:
Prateep A, Vijayaraj GP, Parameswari A, Saravanam PK. Early-onset respiratory distress in a child: A rare presentation. J Laryngol Voice 2021;11:17-20 |
| Introduction | |  |
The most common causes for early onset of respiratory distress in a child are laryngomalacia, subglottis stenosis, laryngeal webs, and cysts.[1] Most cases are managed conservatively. However, if a child has failure to thrive, definitive intervention is almost always required. “Failure to thrive” is a terminology that is used to describe the failure to gain weight due to inadequate intake of calories or when the child's metabolic needs are not met.[2]
Fiber-optic flexible laryngoscopy and radiological imaging have helped in the evaluation of dynamic upper laryngotracheal airway to identify the cause of stridor and to plan its management. This case report discusses the management of a large aryepiglottic fold cyst that compromised the laryngotracheal inlet in a child that resulted in stridor and failure to thrive.
| Case Report | | |
A 6-month-old infant was referred for an otolaryngologist opinion in view of noisy breathing, difficulty in feeding, and failure to thrive since birth. The child was managed conservatively as “Wheeze Associated Lower Respiratory Infection” syndrome by the pediatrician. Further, it was noted that the child was born at full term by normal vaginal delivery, appropriate for gestational age, cried immediately after birth, and did not have any history of airway intervention. On examination, it was observed that the child was weak and malnourished. The child was in severe respiratory distress and had inspiratory stridor along with severe suprasternal and intercostal retractions [Figure 1].
Flexible fiber-optic laryngoscopy was done which showed a single large, smooth globular mass of size 2 cm × 2 cm in the right supraglottic region obscuring the laryngeal inlet and compromising the airway [Figure 2]. | Figure 2: Preoperative transnasal flexible laryngoscopy showing a smooth globular mass in the supraglottic region obscuring the airway
Click here to view |
A contrast-enhanced computed tomography of the neck was done, which was suggestive of a thin-walled cystic lesion with homogeneous enhancement measuring 1.5 cm × 1.9 cm × 2.9 cm in the region of the right pyriform fossa ext2nding superiorly up to the level of C2 vertebra and inferiorly up to the level of C5 vertebra. The lesion was seen to cross the midline compromising the laryngeal inlet along with the narrowing of the proximal trachea for about 1 cm [Figure 3]a, [Figure 3]b, [Figre 3]c. A diagnosis of a supraglottic cyst arising from the right aryepiglottic fold was made. | Figure 3: (a) Axial section of the contrast-enhanced computed tomography of the neck showing the mass causing narrowing of the airway at the level of supraglottis (b) Sagittal section of contrast-enhanced computed tomography of the neck showing the mass extending from c2 vertebrae to c5 vertebrae. (c) Coronal section of contrast-enhanced computed tomography of the neck showing similar features of airway narrowing at the level of supraglottis
Click here to view |
Treatment
A preoperative assessment for the excision of the cyst was made and discussed with anesthetic team. A high risk for anticipated difficult airway intubation along with the need for emergency tracheostomy was discussed with the attenders. After obtaining informed consent, the child was taken up for excision of the cyst under general anesthesia.
At the time of surgery, the child was preoxygenated and sedated with the help of a C-MAC, transoral endotracheal intubation was attempted. The mass was hugely obscuring the airway, and hence, intubation failed. Using an 18-Gauge modified scalp vein needle, the cyst was aspirated with the help of C-MAC assistance. About 8 ml of fluid was aspirated, thereby reducing the size of the cyst which allowed the visualization of the laryngeal inlet and helped in securing the airway by oral endotracheal intubation [Figure 4]a, [Figure 4]b, [Figure 4]c. A pediatric suspension laryngoscope was introduced and fixed. Using an operating microscope, the entire cyst was excised and marsupialized. A nasogastric tube was inserted. The child was extubated and observed in the pediatric intensive care unit for 24 h. | Figure 4: (a) Visualization of the airway using C-MAC videolaryngoscope during intubation before aspirating the fluid from the cyst. (b) C-MAC image showing a complete reduction in the size after aspiration of fluid from the cyst. (c) C-MAC image showing the successful intubation after creating adequate space by reducing the size of the cyst
Click here to view |
The postoperative period was uneventful. There was no respiratory distress or stridor. After 48 h, the nasogastric feeding tube was removed and the child was started on oral feeds and discharged.
Histopathology was suggestive of benign cyst lined by respiratory epithelium and subepithelial mucous glands. The child was kept on regular follow-up, with no further episodes of stridor or respiratory distress. At 1-month postoperative, flexible fiber-optic laryngoscopy was done, which showed complete resolution of the lesion [Figure 5]. At 4-month postoperative, there were no signs of respiratory distress and the child was feeding normally and had gained weight. | Figure 5: Postoperative transnasal flexible laryngoscopy showing complete regression of size of the cyst at 3-month follow-up
Click here to view |
| Discussion | | |
The most common cause of early-onset stridor in infants and children is laryngomalacia, followed by subglottic stenosis.[3] Congenital laryngeal cysts are relatively uncommon developmental anomalies with an incidence of about 1.82 per lakh live births.[4]
Congenital laryngeal cysts are of two types – ductal and saccular cysts.[5] The most common sites of laryngeal cyst are the glottis (58.2%), laryngeal ventricle (18.3%), epiglottis and vallecula (20.2%), aryepiglottic fold (2.2%), and interarytenoid area (0.7%). These patients usually present with hoarseness, weak cry, feeding difficulties, and early respiratory distress with cyanotic episodes. Most often, children with severe respiratory distress have associated feeding difficulties and also have features of failure to thrive. Infant larynx is different from the adult larynx in terms of shape, position, elasticity, and collapsible nature of the cartilage around the laryngeal inlet.[6] These factors explain why infants are predisposed to airway obstruction. Early evaluation for respiratory distress or stridor helps in identifying the cause and planning the management of stridor. A fiber-optic flexible laryngoscope is an invaluable tool for the dynamic evaluation of the larynx and the upper trachea.[7] Radiological imaging provides vital information on site, extent, and type of lesion. Contrast-enhanced computed tomography helps in differentiating vascular, cystic, and inflammatory lesions.[7] Magnetic resonance imaging has a limited role in the evaluation of a child in severe respiratory distress.
Surgical excision or marsupialization of the cyst is the treatment of choice. Aspiration of the cyst contents has been reported in the literature but has limited value as chances of recurrences with reaccumulation of mucous within the cyst are high.[8] Powered instruments such as light amplification by stimulated emission of radiation have been successfully used in the management of such cases.[9] Cold-knife micro laryngeal surgery is feasible and causes less thermal damage as demonstrated in this case. Large laryngeal cysts can be successfully managed by aspiration of the cyst, thereby allowing visualization of larynx and intubation, thus avoiding tracheostomy and its morbidities.
| Conclusion | | |
This case reports a rare, large aryepiglottic cyst that presented with early-onset stridor and failure to thrive in a child.
Flexible fiber-optic laryngoscopy along with contrast-enhanced computed tomography is an invaluable tool for dynamic evaluation of the airway.
The author emphasizes the importance of teamwork along with anesthesiologists in an effort to establish an airway and avoid tracheostomy.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Clark CM, Kugler K, Carr MM. Common causes of congenital stridor in infants. JAAPA 2018;31:36-40.  |
| 2. | Larson-Nath CM, Goday PS. Failure to thrive: A prospective study in a pediatric gastroenterology clinic. J Pediatr Gastroenterol Nutr 2016;62:907-13. |
| 3. | Pak MW, Woo JK, van Hasselt CA. Congenital laryngeal cysts: Current approach to management. J Laryngol Otol 1996;110:854-6. |
| 4. | Sakakura K, Chikamatsu K, Toyoda M, Kaai M, Yasuoka Y, Furuya N. Congenital laryngeal anomalies presenting as chronic stridor: A retrospective study of 55 patients. Auris Nasus Larynx 2008;35:527-33. |
| 5. | Kakodkara KA, Schroeder JW Jr, Holingerb LD. Laryngeal development, and anatomy pediatric airway surgery. Adv Otorhinolaryngol 2012;73:1-1. |
| 6. | Desanto LW, Devine KD, Weiland LH. Cysts of the larynx – Classification. Laryngoscope 1970;80:145-76. |
| 7. | Kirse DJ, Rees CJ, Celmer AW, Bruegger DE. Endoscopic extended ventriculotomy for congenital saccular cysts of the larynx in infants. Arch Otolaryngol Head Neck Surg 2006;132:724-8. |
| 8. | Suzuki J, Hashimoto S, Watanabe K, Takahashi K. Congenital vallecular cyst in an infant: Case report and review of 52 recent cases. J Laryngol Otol 2011;125:1199-203. |
| 9. | Lim EH, Mohamad H, Sheikh Ab Hamid S. Congenital laryngeal cyst: A report of 2 cases. Egypt J Ear Nose Throat All Sci 2017;18:187-9. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
|
