Anesthetic management of a large cystic hygroma in a newborn.

Cystic hygroma is a congenital benign tumor occurring due to the accumulation of lymph and during its anesthetic management difficulties are known to be encountered. A newborn baby presented with a massive swelling in the front of the neck. It was an antenatally diagnosed case of cystic hygroma with intraoral extension proving to be an anticipated difficult airway. Following inhalational induction, mask ventilation was possible, and the child was successfully intubated. Intra-operative period was uneventful, and the tumor was completely excised. Postoperatively, the child was ventilated for 24 h in view of anticipated airway edema.

INTRODUCTION

Anesthetic management of the pediatric airway can be challenging in neonates and with the addition of a huge cystic hygroma of neck, difficulties are known to be encountered during anesthesia. Cystic hygroma is a congenital benign tumor occurring due to the accumulation of lymph in the jugular lymphatic sacs of nuchal region. The key to a successful management includes identification of the potential problems and considering different options with a selection of an appropriate plan of anesthesia. In this case report, we describe the successful anesthetic management of a newborn with a huge cystic hygroma arising from the neck with intraoral extension.

CASE REPORT

A newborn baby boy weighing 3.5 kg presented with a huge swelling in front of the neck [Figures 1 and 2]. There were no features of respiratory distress and examination revealed a huge cystic swelling in front of the neck measuring 6 cm × 10 cm extending from the angle of the mandible to the clavicle with involvement of under surface of tongue pushing the tongue upward [Figure 3]. The skin over the swelling looked normal, and transillumination test was positive. A 24-gauge intravenous (IV) cannula was already in place over dorsum of the right hand. Chest X-ray was done to exclude intra-thoracic extension of the tumor. Lateral view of X-ray neck and computed tomography (CT) scan showed no evidence of airway compression. Informed consent for anesthesia was taken after explaining the parents about the possibility of difficult intubation. Blood was cross-matched and reserved. Difficult airway cart was kept ready. After shifting to the operating room, standard monitors including electrocardiography, pulse oximeter, and noninvasive blood pressure cuff were attached. Baseline SpO2 recorded was 97% with room air. Following topical anesthesia of the airway with 2% lignocaine viscous smeared using a gloved finger, a check laryngoscopy revealed visible epiglottis with cystic swellings in oropharynx. Preoxygenation was carried out for 3 min. With a visible epiglottis in mind, we proceeded with inhalational induction using sevoflurane with 100% oxygen. After confirming mask ventilation injection atracurium 2 mg and injection fentanyl 8 mcg were given. With the aid of an assistant to lift the swelling, oral intubation was successfully performed at the first attempt using a MAC 1 Macintosh blade laryngoscope with 3.5 mm size uncuffed Portex endotracheal tube (ETT). Tube position was confirmed after confirming bilateral equal air entry. With the airway now secured, nasal intubation was considered in view of anticipated accidental tube dislodgement during surgery [Figure 4]. The oral tube was withdrawn from the vocal cord level once the nasal tube (3.5 mm) with the aid of a Magill's forceps was passed until the glottis level. Tube position was confirmed with EtCO2 and fixed after confirming equal air entry. A 22-gauge IV cannula was secured over dorsum of left foot.

Figure 1

Front view of the child

Figure 2

Side view of the child showing extent of cystic hygroma

Figure 3

Intraoral extension of cystic hygroma

Figure 4

Photo of the child following nasal intubation

Anesthesia was maintained with 66% nitrous oxide in oxygen with controlled ventilation using Jackson Rees modification of Ayre's T piece and top up doses of 0.1 mg/kg atracurium were used as and when necessary. Intra-operative period was uneventful with minimal blood loss. The cysts were multiloculated and excised completely. The child was electively ventilated in view of the possibility of airway edema and was successfully extubated on the 2nd postoperative day.

DISCUSSION

Cystic hygroma is a benign congenital tumor of lymphatic origin which is also called cavernous hemangioma. They contain large multiloculated cysts filled with serous secretions.[1] Clinically, it is a fluctuant lesion which transilluminates. Most often it appears during the 1st year of life but can occasionally present at birth.[2] It is known to coexist with congenital anomalies like Downs and Turners syndrome and also heart defects.[3] The head and neck region are the most common sites of origin, but they can extend into the floor of mouth, pharyngeal wall, airway, and thorax, presenting numerous problems to the anesthesiologist.[45] The most common clinical feature is the presence of a mass. Interference with normal breathing and swallowing are the next symptoms to appear. The anesthesiologist should have knowledge of the possible extension of the cyst into the respiratory tract. One should assess the size and extent of the neck swelling in order to formulate an appropriate plan of airway management. Chest X-ray is mandatory for all cases to exclude the presence of intra-thoracic extension. Tumors located in the mediastinum require further investigations like angiography, CT scan and fluoroscopy. As already mentioned the presence of concurrent disease must be ruled out.[6]

Premedication atropine can be given in a dose of 20 μg/kg to dry secretions, but sedatives should be used with caution because of their potential to worsen airway obstruction.[7] Although some neck masses are known to regress spontaneously, this child had to undergo surgical resection going by the huge size and intraoral extension of the tumor.

The main objective in managing this case was securing the airway. Newborn infants are as it is difficult to intubate, the presence of a huge neck swelling only adds up to the problems. One has to also keep in mind to limit the intubation duration as they are prone for early development of hypoxia.[8] Should a can’t intubate, can’t ventilate situation arise, equipment for emergency airway access like cricothyrotomy kits as well as a standby surgeon to do a tracheostomy should be available.

It is well-known fact that the loss of muscle tone induced by general anesthesia aggravates the pressure effect of a mass leading to airway obstruction and thus inhalational induction remains the preferred technique for management of a difficult pediatric airway. Hence, we proceeded with inhalational mode of induction with the primary aim of maintaining spontaneous ventilation using sevoflurane.[9] Muscle relaxant was given only after confirming face mask ventilation. Different options are available for intubation. Blind nasal intubation requires expertise and can be associated with bleeding which can further compromise the airway. Fiberoptic intubation is a popular technique but its availability and also a considerable amount of skill and experience required to handle it in a neonate limits its use. Tracheostomy under local anesthesia is another option but was not considered in this case because of the distorted anatomical landmarks. Partial aspiration of the cyst can facilitate intubation but can make the surgery more difficult.[10]

Another problem is maintenance of airway during surgery. Being aware of the possibility of accidental dislodgement and extubation of the ETT due to the painting solutions used which can loosen the adhesive plaster and manipulation during surgery, we decided to switch over from an oral to a nasally inserted ETT.[8]

Behavior of cystic hygroma is unpredictable, although spontaneous regression with time occurs in some cases most of them progressively increase in size. The definitive treatment for cystic hygroma is surgery, but repetitive surgeries are often needed due to the infiltrating nature of the condition. We delayed extubation in our patient simply because of the high mortality rate of neonates with large cystic hygroma due to high incidence of postoperative complications like respiratory obstruction secondary to edema of the airway, airway collapse, tongue edema, rapid expansion of residual cyst due to hemorrhage, inflammation and infection of the cyst, and also injury to the recurrent laryngeal nerve. The other important complications include injury to the hypoglossal nerve, facial nerve, and internal jugular vein.[11]

To avoid and manage these potential airway complications adequate knowledge of the nature of the tumor together with communication and coordination between the anesthesiologist, otolaryngologist, and pediatric surgeon as well as presence of an additional experienced anesthesiologist and nursing staff are required.

CONCLUSION

The success in management of a cystic hygroma largely depends upon a detailed and proper preoperative evaluation with emphasis on airway assessment as well as formulation of an appropriate plan for intubation. Equally important is to not hurry extubation considering the potential for postoperative airway-related complications.