Subglosso-palatal membrane a rare cause of neonatal respiratory distress: A case report.

Subglosso-palatal membrane is a rare cause of acute respiratory distress in newborn. Contrary to the earlier reported cases in which immediate splitting or excision of membrane has been described as measure to relief respiratory distress, we managed our case successfully without any emergency surgical procedure.

INTRODUCTION

Subglosso-palatal membrane is a rare cause of acute respiratory distress in newborn.[1] Immediate splitting or excision of membrane has been described previously as an emergency measure to relief respiratory distress.[2] Herein, we present a case of subglosso-palatal membrane with respiratory distress managed initially without any surgical procedure.

CASE REPORT

A newborn male baby weighing 3.1 kg delivered by cesarean section at 36 + 4 weeks of gestation with Apgar scores, 7 and 8 at five and ten minutes, respectively. The baby developed tachypnea soon after birth and was presented with signs of respiratory distress (tachypnea, nasal flaring, grunting, retractions, and cyanosis) shortly after birth. Physical examination revealed the following conditions of the baby: heart rate 156/min and respiratory rate 75/min with normal breath sounds. On oral examination, a web was found extending from the floor of the mouth to the junction of the soft and hard palates. Lateral extension was from right molar trigone to left molar trigone [Figure 1]. Nasal and oral suction was performed. Rest of examination was normal. Chest radiograph showed diffuse parenchymal infiltrates. Arterial blood gas analysis was normal. Diagnosis of subglosso-palatal membrane with transient tachypnea of the newborn was made and supplemental oxygen was started by nasal cannula at 4 liters/min. Further intravenous fluids and antibiotics were started by maintaining the thermoneutrality. In the next 12 hours, oxygen requirements were reduced, respiratory rate settled and the child started excepting breast feed at 72 hour of life. At 20th day of life, membrane was excised [Figure 2]. Postoperative course was uneventful and baby was discharged.

Figure 1

Subglosso-palatal membrane

Figure 2

Cleft palate after membrane excision

DISCUSSION

The presence of web in the oral cavity is a rare entity. Subglosso-palatal membrane is a fibrous connections extending from floor of mouth to palate and is usually associated other congenital anomalies.[12] Only few cases have been reported in the literature, which are mostly associated with other multiple congenital anomalies. Our case had no other associated congenital anomaly except for the cleft palate.[12] The stomodeum and foregut are separated by the oropharyngeal or buccopharyngeal membrane derived from the prechordal plate, which is a fusion of ectoderm and endoderm without mesodermal penetration during 4th and 5th week of life.[3] The posterior part of maxillary palatal process is located downwards on both sides of tongue.[4] The tongue, floor of the mouth, and palatal shelves develop in close contact with one another during the 5th and 6th weeks of prenatal life. At the 8th week of gestation, the tongue starts to drop while the neck is being extended, and the fibrous connections disappear, with the palatal shelves moving into a horizontal position and eventually fusing in the midline.[3] Remnants of embryologic tissue such as the buccopharyngeal membrane or abnormal adhesions between structures in close proximity[5] could give rise to intraoral bands during these phases of embryologic development.[4]

Respiratory distress and feeding difficulties are the major causes of morbidity in these infants secondary to intraoral synechiae.[14] Our case also presented with respiratory distress. Airway obstruction due to membrane has been reported.[12] Death due to respiratory arrest, bronchopneumonia, and severe dehydration has been documented.[14] On the first day of the life, urgent splitting or resection of the intraoral band under local anesthesia has been described in previous reports to improve respiratory status.[12] Contrary to the earlier reported cases, we managed our case successfully without any emergency surgical procedure. Newborns are preferential nasal breathers.[67] This is due to large tongue that abuts against the hard and soft palates and occludes the passage of air through the mouth and the high position of their epiglottis increases the resistance of the oral airway as well. Hence, any partial obstructing pathology in mouth of an infant should not cause respiratory distress as described previously. In our case, respiratory distress was caused by underlying transient tachypnea of the newborn. It is a benign condition usually seen in term babies born by cesarean section.[8] These babies are well and have only tachypnea with rates as high as 80-100/min. The breathing is shallow and rapid without any significant chest retractions. Hypoventilation is very uncommon, and partial carbon dioxide tensions are usually normal because of the tachypnea. It occurs because of delayed clearance of lung fluid. Management is supportive and prognosis is excellent. X-ray shows clear lung fields with prominent right interlobar fissure with borderline cardiomegaly suggestive of transient tachypnea of the newborn.[9]

Respiratory distress is a common emergency responsible for 30-40% of admissions in the neonatal period.[10] A working diagnosis should be made in the first few minutes of observing the baby and immediate life saving measures should be undertaken until further management plans are drawn up. Respiratory distress in these cases may be the result of associated congenital anomalies and other causes of respiratory distress should be considered. Immediate surgical intervention can be avoided in such cases as it may increase morbidity and mortality. Excision of the membrane can be done once the respiratory distress has settled.

CONCLUSION

Subglosso-palatal membrane is a rare entity. It is not only cause of the respiratory distress in newborn, as described previously, and other causes of respiratory distress should be considered as well. Immediate surgical intervention needs to be avoided in such cases as this may lead to a increase in morbidity and mortality and mortality. Simple excision of the membrane should be done once the respiratory distress has settled.