Journal of Clinical Neonatology

CASE REPORT
Year
: 2019  |  Volume : 8  |  Issue : 4  |  Page : 243--244

A vallecular cyst with near-fatal respiratory distress in a newborn


Selahattin Katar1, Nazan Degirmenci2,  
1 Department of Pediatrics, Dicle University of Medicine, Diyarbakir, Turkey
2 Department of Otolaryngology, Bezmialem University of Medicine, Istanbul, Turkey

Correspondence Address:
Prof. Dr. Selahattin Katar
Department of Pediatrics, Dicle University of Medicine, Diyarbakir
Turkey

Abstract

Vallecular cyst is a rare but potentially near-fatal cause of respiratory distress in newborns. A male infant was born normally. There was respiratory distress. A cystic mass was found in the right parasagittal area of the hypopharynx and right sublingual region. Examination was done with direct laryngoscopy, and the vallecular cyst was surgically removed. Vallecular cyst should be considered as a differential diagnosis in respiratory distress cases.



How to cite this article:
Katar S, Degirmenci N. A vallecular cyst with near-fatal respiratory distress in a newborn.J Clin Neonatol 2019;8:243-244


How to cite this URL:
Katar S, Degirmenci N. A vallecular cyst with near-fatal respiratory distress in a newborn. J Clin Neonatol [serial online] 2019 [cited 2020 Jan 21 ];8:243-244
Available from: http://www.jcnonweb.com/text.asp?2019/8/4/243/268591


Full Text



 Introduction



Vallecular cyst is a rare but potentially near-fatal cause of respiratory distress in newborns.[1] Congenital vallecular cyst-related symptoms such as respiratory distress, feeding problems, and inspiratory stridor occur during the 1st week of life after birth. The most common cause of stridor at the postnatal period is laryngomalacia, and a congenital laryngeal cyst is often misdiagnosed as laryngomalacia. Here, we report a case of vallecular cyst that caused respiratory distress immediately after birth and difficulty in tracheal intubation.

 Case Report



The case is of a male infant born through normal vaginal route at 39 weeks. Immediately after birth, there was respiratory distress, and the patient was referred to our hospital. At the first sight, the patient had cyanosis and suprasternal withdrawal, and hence was immediately intubated. His body weight was 3100 g, height was 50 cm, and head circumference was 35 cm. Other systemic examinations were normal. Chest radiography was normal. On laboratory tests, C-reactive protein was 0.7 mg/dL, hematocrit was 59%, and white blood cell count was 8750/μL. Intubation was difficult because the cyst was causing obstruction. After intubation, the patient was stable, and the diagnosis was thought to be laryngotracheomalacia. After 2 days of follow-up, extubation was performed and respiratory distress occurred again. After that, the patient was intubated immediately and intubation was again difficult. Further examination was planned. Neck ultrasonography (USG) and otorhinolaryngological consultation were performed. On USG examination, a cystic mass of 19 mm in diameter was found in the right parasagittal area of the hypopharynx, starting from the right sublingual region. Flexible laryngoscopic examination revealed a round tissue swelling of 2 cm × 1 cm with a smooth wall at the right side of the hypopharynx. In the surgery room, the examination was done with direct laryngoscopy, and the cyst was surgically removed. On direct laryngoscopy, it was seen that the cyst was narrowing the airway passage, extending to the epiglottis [Figure 1]. Histopathology was compatible with vallecular cyst. There was no respiratory distress at the postoperative 24 h. The patient was discharged at the 7th postoperative day.{Figure 1}

 Discussion



Vallecular cysts are a form of supraglottic cyst accounting for 10.5%–20.1% of all laryngeal cysts.[1],[2] Very few cases have been reported which occurred in newborns.[3] Although congenital vallecular cysts are rare, they may be fatal due to respiratory obstruction. In our case, severe respiratory insufficiency developed due to obstruction immediately after birth. Suprasternal withdrawal, widespread cyanosis, and tachypnea developed at the postpartum 1st h. Most of the time, these cases are considered as transient tachypnea of newborns. Their initial symptoms may be mild, which may lead to delay in recognition or misdiagnosis. The average time for the diagnosis of vallecular cyst varies from 3 to 40 days of life.[1] In a study, 33 cases were reported to have an average time of diagnosis of 2.3 months.[4] However, in the case reported by Cuillier et al., it has been reported that the diagnosis can also be made in the antenatal period.[5] A delayed diagnosis can cause death due to sudden obstruction of airways. In the present case, on the postnatal 2nd day, the cyst was diagnosed with neck USG, and the diagnosis of vallecular cyst was confirmed by direct laryngoscope on the onset of respiratory distress after extubation. The treatment of choice for laryngeal vallecular cyst is endoscopic marsupialization or surgical cyst excision.[6] In our case, the otolaryngologists performed surgical excision. In some cases, postoperative symptoms may recur, but in our case, there were no symptoms after postoperative 1-year follow-up. With this case, we intend to mention the risk of death in newborns due to airway obstruction caused by vallecular cyst. Respiratory distress, especially with suprasternal withdrawal in newborns, and patients with difficult intubation should be considered in terms of vallecular cyst.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient's guardian has given consent for the child's images and other clinical information to be reported in the journal. The patient's guardian understands that the child's name and initials will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1Romak JJ, Olsen SM, Koch CA, Ekbom DC. Bilateral vallecular cysts as a cause of dysphagia: Case report and literature review. Int J Otolaryngol 2010;2010:697583.
2Tsai YT, Lee LA, Fang TJ, Li HY. Treatment of vallecular cysts in infants with and without coexisting laryngomalacia using endoscopic laser marsupialization: Fifteen-year experience at a single-center. Int J Pediatr Otorhinolaryngol 2013;77:424-8.
3Kothandan H, Ho VK, Chan YM, Wong T. Difficult intubation in a patient with vallecular cyst. Singapore Med J 2013;54:e62-5.
4Hsieh LC, Yang CC, Su CH, Lee KS, Chen BN, Wang LT. The outcomes of infantile vallecular cyst post CO2 laser treatment. Int J Pediatr Otorhinolaryngol 2013;77:655-7.
5Cuillier F, Samperiz S, Testud R, Fossati P; Department of Obstetrics and Gynecology, Félix Guyon Hospital, Reunion Island, France. fabrice.cuillier@wanadoo.fr. Antenatal diagnosis and management of a vallecular cyst. Ultrasound Obstet Gynecol 2002;20:623-6.
6Parelkar SV, Patel JL, Sanghvi BV, Joshi PB, Sahoo SK, Sampat N, et al. An unusual presentation of vallecular cyst with near fatal respiratory distress and management using conventional laparoscopic instruments. J Surg Tech Case Rep 2012;4:118-20.