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LETTERS TO EDITOR
Year : 2017  |  Volume : 18  |  Issue : 2  |  Page : 92-93
 

Airway management in an infant presenting with congenital bilateral neck lymphangioma and tongue hamartoma


Department of Anaesthesia, BJ Medical College and Sassoon Hospital, Pune, Maharashtra, India

Date of Web Publication12-Dec-2017

Correspondence Address:
Dr. Sushama Raghunath Tandale
E28, Vishnu Sadashiv, Opposite Old Zilha Parishad, New Mangalwar Peth, Pune, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/TheIAForum.TheIAForum_19_17

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How to cite this article:
Tandale SR, Gadre VN, Yadav AH, Vaidya NV. Airway management in an infant presenting with congenital bilateral neck lymphangioma and tongue hamartoma. Indian Anaesth Forum 2017;18:92-3

How to cite this URL:
Tandale SR, Gadre VN, Yadav AH, Vaidya NV. Airway management in an infant presenting with congenital bilateral neck lymphangioma and tongue hamartoma. Indian Anaesth Forum [serial online] 2017 [cited 2019 Aug 25];18:92-3. Available from: http://www.theiaforum.org/text.asp?2017/18/2/92/220552


Sir,

Pediatric airway management is challenging due to its unique anatomical and physiological variations. The presence of congenital lesion involving the airway magnifies the risk. Meticulous history taking, thorough examination, knowledge of the condition and preoperative planning is crucial for successful airway management.

We report a case of 4-month-old, 4 kg female with bilateral neck lymphangioma and tongue hamartoma posted for excision under general anesthesia. She presented with snoring; diagnosed to have bilateral neck swelling in antenatal scan at 30 weeks. Birth history was unremarkable. CT scan revealed bilateral multiple intercommunicating hypodense cystic lesions of the neck extending up to angle of mandible, parotid gland, submandibular gland, sternocleidomastoid muscle and chin [Figure 1]. The largest one was 2.9 cm × 2.6 cm near right angle of mandible. Tongue appeared purple with small lesions and obvious swelling over dorsum. Echocardiography revealed atrial septal defect of 7 mm with left to right shunt. Systemic and hematological examinations were normal. Inhalational anesthesia with spontaneous breathing was planned to administer anesthesia. Standard monitoring was done. Difficult airway cart was kept ready in view of difficult airway secondary to reduced mouth opening and difficult mask ventilation. It included face mask, oropharyngeal, nasopharyngeal airway and laryngeal mask airway of assorted sizes, stylet, endotracheal tube (sizes 2.5, 3, 3.5), laryngoscope with straight and curved blades, cricothyroidotomy, and tracheostomy tray. Intravenous (IV) glycopyrrolate 20 mcg and Ondansetron 0.4 mg was given. After preoxygenation, the child was given IV ketamine 5 mg, and anesthetic plane was deepened with gradually increasing the concentration of sevoflurane up to 8 volume percentage with 100% oxygen through Jackson Rees circuit. Effective mask ventilation was achieved with two hand technique and jaw thrust. The use of airway was avoided to minimize tongue trauma. Gentle laryngoscopy was performed with Macintosh size one blade followed by endotracheal intubation with 3.5 uncuffed tube [Figure 2]. Large epiglottic cyst was visualized during laryngoscopy with Cormack-Lehane grade II. External laryngeal manipulation was not required. Anesthesia was maintained with oxygen, nitrous oxide, and sevoflurane. Analgesia was given with IV fentanyl 5 mcg, paracetamol suppository 80 mg, and incision infiltration with local anesthetics. Surgery was uneventful with minimal blood loss. Unilateral excision was done anticipating airway obstruction due to edema secondary to surgical manipulation and collection of serosanguinous fluid. At the end of surgery, neuromuscular block was reversed and trachea extubated after spontaneous respiratory efforts and recovery of consciousness.
Figure 1: Extensive involvement of bilateral neck due to lymphangioma

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Figure 2: Patients picture before endotracheal intubation and after endotracheal intubation

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Lymphangiomas are benign cystic lesions, representing the sequestration of lymphatic tissue during embryogenesis not communicating with remaining lymphatic channels.[1] Incidence is 1.2–2.8/1000 newborns. About 50% of them are diagnosed at birth and majorities are diagnosed before 2 years of age.[2] Most commonly they appear in cervicofacial region. Their bilateral occurrence is uncommon. The presence of mass, breathing difficulty and feeding difficulty are presenting manifestations in them.[3] They are classified as microcystic (capillary lymphangioma), macrocystic (cavernous lymphangioma), and cystic hygromas according to size of the lymphatic cavities incorporated.[3]

It can be associated with Down's syndrome, Turner's syndrome, Noonan's syndrome, and cardiac anomalies.[2] Mild dysmorphic features and atrial septal defect were noted in the index case. Radio imaging helps to know the extent of lesion, airway involvement and extent of resection. Anesthesiologist must have an alternate plan such as retrograde intubation, cricothyroidotomy, and tracheostomy to secure airway in such situations. Intensive monitoring for airway patency in postoperative period is must. Our child also had tongue hamartoma which demands cautious laryngoscopy to avoid bleeding due to trauma to tongue. It reflects the excessive disorganized growth of tongue tissue merging with the surrounding tissue.[4]

Airway management of infant with lymphangioma and tongue hamartoma requires the information of disease and experience in pediatric airway management. Inhalational induction with spontaneous breathing is a better technique. Difficulty in securing airway, intraoperative blood loss and postoperative airway obstruction should be anticipated.

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.

Acknowledgment

Author would like to acknowlaged Dr. Jayesh Sudhakar Desale M.S., MCH Paediatric Surgery, Assistant Professor, Department of Paediatric Surgery, BJ Medical College and Sassoon Hospital, Pune.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Gupta S, Ahuja P, Rehani U, Singh V. Lymphangioma of cheek region - An unusual presentation. J Oral Biol Craniofac Res 2011;1:47-9.  Back to cited text no. 1
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2.
Anpuram LN, Chakravarthy K., Cherukuri SK, Nalagandla SR. Anaesthetic management of a huge oral lymphangioma in a neonate. J Evol Med Dent Sci 2015;4:15493-5.  Back to cited text no. 2
    
3.
Grasso DL, Pelizzo G, Zocconi E, Schleef J. Lymphangiomas of the head and neck in children. Acta Otorhinolaryngol Ital 2008;28:17-20.  Back to cited text no. 3
    
4.
Hsu YC, Hsu WC. Tongue base hamartoma in a child. J Formos Med Assoc 2012;111:406-7.  Back to cited text no. 4
    


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