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Unexpected Difficult Tracheal Reintubation After Thyroglossal Duct Surgery

Functional Imbalance Aggravated by the Presence of a Hematoma

Van Boven, M. J. MD; Lengele, B. MD; Fraselle, B. MD; Butera, G. MD; Veyckemans, F. MD

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Hyoid bone resection is a common step in thyroglossal cyst surgery. It is generally limited to the median part of the hyoid arch, including the hyoid body between the two lesser horns of the bone. This surgical damage is thought not to induce any functional problem for the patient. However, we describe the case of a child in whom the immediate dynamic consequences of hyoid bone resection, combined with the presence of a local hematoma, resulted in a difficult tracheal reintubation despite the absence of obvious signs of upper airway obstruction. The pathophysiology of this unusual complication emphasizes the central role played by the hyoid bone in the biomechanics of tongue support, mandibular positioning, and laryngeal suspension.

Case Report

A healthy 6-yr-old girl was admitted for excision of a thyroglossal duct cyst diagnosed 2 yr earlier by a small, soft, cystic tumor in front of the hyoid bone. Inhaled anesthesia was induced with halothane in oxygen and nitrous oxide. An intravenous (IV) infusion was given as soon as the child was asleep. Sufentanil 10 micro gram and atracurium 10 mg were then injected IV, and ventilation was controlled with isoflurane in place of halothane. Nasotracheal intubation was very easily performed (uncuffed 5.5-mm inside diameter Portex Registered Trademark tube). Surgery proceeded uneventfully; the body of the hyoid bone was removed, and a small Penrose drain was left in place. The trachea was extubated at the end of the operation. However, the surgical dressing had to be changed twice during the 90-min stay in the recovery room due to persistent oozing from the wound.

Eight hours later, the child was found in her bed in the knee-chest position. She complained in a whisper of pain in the anterior aspect of the neck and was unable to swallow. There were no signs of dyspnea and she tolerated well the supine position. At inspection, the floor of the oral cavity was found to be swollen, forcing the tongue upward, but macroglossia was not present. The presumptive diagnosis of postoperative hemorrhage was established, and it was decided to explore the wound.

The child was immediately brought back to the operating room, and anesthesia was induced IV using the venous cannula inserted in the morning. After preoxygenation and with an assistant applying gentle cricoid pressure, sufentanil 5 mg and propofol 80 mg were administered. Ventilation via a mask was easy, so it was decided to inject 10 mg atracurium IV; ventilation was then controlled until complete muscular relaxation was obtained.

At laryngoscopy with a size 3 Macintosh blade, the pharyngeal and laryngeal structures were difficult to identify: the mucosae were swollen and edematous, pushing and fixing the swollen epiglottis backward and thus hindering any view of the glottis.

At second laryngoscopy, gentle probing of these edematous structures with the distal end of a Steiner needle (atraumatic 3 times 190-mm needle for topical anesthesia; Unimed, Lausanne, Switzerland, ref.: 204.012 D, 2R2) allowed the identification of the arytenoids. Oral endotracheal intubation was thereafter easy. The surgical exploration of the wound allowed the evacuation of a deep hematoma (the size of a cherry) and the hemostasis of a small muscular artery that was bleeding.

It was decided to wait for the resolution of the edema before extubating the trachea; the child therefore remained in the pediatric intensive care unit during the first 12 postoperative hours. The patient went back home without any sequelae 36 h later.

Discussion

Very early in the fetal life, the thyroid gland develops from the base of the tongue and descends in the midline of the neck to its normal position. Later, the duct solidifies and is then known as the His's thyroglossal tract. Any part of the duct may persist along the path of the fibrous tract as an epithelial remnant giving rise to glandular inclusions, thyroglossal cysts, or thyroglossal ductular sinuses communicating with the skin of the neck on the midline [1-3]. According to their embryogenesis, thyroglossal cysts are usually located just below or just above the hyoid bone. Most patients present with a symptomless lump in the midline neck area that rises on protrusion of the tongue.

The treatment of thyroglossal cysts or fistulae consists of radical surgical removal of the embryonic epithelial remnant. This radical excision encompasses the central part of the body of the hyoid bone, which is intimately related to it [4]. No muscular repair is usually performed.

Being completely isolated from the other skeletal elements of the skull, the hyoid bone keeps a relatively stable position within its surrounding soft tissues due to the antagonistic forces of the muscles that are inserted on it. Most of them are connected to the median part of the bone; they relate the position of the hyoid body upward to the mandible and downward to the upper aperture of the thorax. The remaining muscles are inserted on the lateral portions of the bone and track it in a posterosuperior direction, toward the base of the skull [5-7].

During normal fetal growth, the hyoid bone becomes the biomechanical center of a delicate muscular balance, conditioning the functional position of the larynx, in close relationship with lingual statics and mandibular dynamics [2,3,5]. This anatomic balance is broken by the resection of the central part of the hyoid bone; the remaining hyoid arch is released from the action of the main supra- and infrahyoid muscles, and the whole laryngeal apparatus moves in a posterosuperior direction up to the base of the skull [6,7]. This movement may lead to severe airway obstruction in the presence of any additional unfavorable circumstance (e.g., edema, hematoma). Moreover, the caudal displacement of the epiglottic cartilage caused by disruption of the hyoepiglottic membrane (Figure 1, top), combined with the global elevation of the larynx and the local edema, explains the disturbance of the topographic landmarks used during laryngoscopy.

Figure 1
Figure 1:
Top, Balanced action of muscle and ligaments to maintain the normal position of the epiglottis. Bottom, Hyoid bone resection influences the tongue positioning in the oral cavity and contributes to the lowering of the epiglottis.

The body of the hyoid bone also acts as the main skeletal support of the base of the tongue; its resection has severe consequences on the sagital positioning of the tongue and lowers the epiglottis over the laryngeal supraglottic area [6] (Figure 1, bottom).

Finally, the hyoid bone plays an indirect role in the dynamic positioning of the mandible. Its resection induces a global upward and posteriorly directed movement of the mandible. Such a displacement, combined with the positional modifications of the larynx, epiglottis, and tongue, also explains the symptoms and the difficult reintubation encountered in our young patient and the relief obtained by adopting the knee-chest position.

The absence of signs of upper airway obstruction (no inspiratory stridor and good tolerance of supine position) and of macroglossia led us consider that the reintubation would be easy, despite the presence of discrete signs of upper airway involvement (whisper, drooling, knee-chest position); this explains why our management of this case was not classic. The possibility of a repercussion on the epiglottis should have led us to anticipate a difficult intubation and to plan its management with different options [8].

Ideally, the case should have been managed as acute epiglottitis [13]. We did not use a rapid-sequence induction with succinylcholine, as recommended in case of emergency or when there is any doubt about gastric emptiness, because we try to avoid using succinylcholine in children and because cricoid pressure is effective to prevent aspiration of gastric content and gastric insufflation. Although cricoid pressure has been recently shown to obstruct the airway in a patient with a hypertrophied lingual thyroid, it did not appear to contribute to our difficulties in this case [9,10]. The presence of an intrapharyngeal pathology contraindicated the use of a laryngeal mask, which could have aggravated the clinical picture by injuring the swollen epiglottis [11]. In case of macroglossia, the use of a fiberoptic laryngoscope should have been considered; however, one should keep in mind that leading the fiberscope beneath a swollen and fixed epiglottis is sometimes difficult, even in the hands of an expert. In the same way, blind nasotracheal intubation would probably have proven difficult, due to the backward displacement of the epiglottis, or even dangerous if the already edematous pharyngeal structures were injured [12]. Moreover, due to the child's age, the use of some form of sedation would have been necessary. Therefore, in these conditions, a cricothyrotomy should probably have been considered.

Because of these anatomic considerations, we want to emphasize that the hyoid bone is the biomechanical center of the complex musculo-ligamentar balance that ensures the laryngeal suspension, upper airway permeability, lingual statics, and dynamic mandibular positioning. Hyoid bone resection, interrupting some of the main muscular slings involved in this cervicocephalic balance, makes its stability precarious and has possible clinical or anesthetic consequences. When combined, these minor displacements of the tongue, larynx, mandible, and epiglottis may lead to upper airway impairment and difficult reintubation in the presence of edema or hematoma.

Regarding the surgical management of this condition, we advise cervical surgeons to try to reconstruct, as anatomically as possible, the muscular slings widely opened by the resection of the hyoid bone. This procedure seems to be mandatory particularly in young children because of the cephalic position of the cervical viscera, which is another factor favoring upper airway obstruction. It could be unnecessary in the adult, because rapid fibrous scarring of the dead space left behind the resected hyoid bone subsequently replaces it efficiently.

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