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Table of Contents
CASE REPORT
Year : 2020  |  Volume : 4  |  Issue : 2  |  Page : 81-83

Airway obstruction after anesthesia in a 3-month-old baby with lymphangioma


Department of Anesthesiology and Intensive Care, Faculty of Medicine, University of Indonesia, Jakarta, Indonesia

Date of Submission19-Mar-2020
Date of Decision25-Apr-2020
Date of Acceptance29-Apr-2020
Date of Web Publication11-May-2020

Correspondence Address:
Dr. Andi Ade Wijaya Ramlan
Department of Anesthesiology and Intensive Care, Faculty of Medicine, Universitas Indonesia, Jakarta
Indonesia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/BJOA.BJOA_31_20

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  Abstract 

Lymphangiomas are commonly benign. However, in rare cases, it may enlarge and cause airway obstruction. The objective of this clinical case report is to highlight the appropriate course of action in cases of airway obstruction. A 3-month-old boy presented with progressive inspiratory stridor since birth. Computed tomography of the thorax revealed multiple lesions on the bilateral neck, right supraclavicular and right supra-anterior to superior mediastinum. The patient was scheduled for tumor resection and injection of sclerosing agent. Induction was done using sevoflurane, and intubation was carried out and presented without complications. The operation was uneventful. After extubation, retraction in the suprasternal, intercostal, and epigastrium was observed; hence, the patient was reintubated. Extubation was then done the following day after careful positioning in intensive care. In this case, laryngeal edema was caused by the obstruction of the lymphatic drainage, which was present since before the intubation. However, after the surgery, there was worsening of the edema. It may be caused by inflammatory response toward lymphatic drainage, thus worsening lymphatic outflow obstruction.

Keywords: Airway obstruction, general anesthesia, lymphangioma


How to cite this article:
Wijaya Ramlan AA, Ang M, Peddyandhari FS. Airway obstruction after anesthesia in a 3-month-old baby with lymphangioma. Bali J Anaesthesiol 2020;4:81-3

How to cite this URL:
Wijaya Ramlan AA, Ang M, Peddyandhari FS. Airway obstruction after anesthesia in a 3-month-old baby with lymphangioma. Bali J Anaesthesiol [serial online] 2020 [cited 2020 Jul 13];4:81-3. Available from: http://www.bjoaonline.com/text.asp?2020/4/2/81/284190




  Introduction Top


Lymphangioma is a rare congenital malformation of the lymphatic system. Lymphangioma most commonly affects head-and-neck region, followed by axilla, and rarely involves the mediastinum, abdominal organs, retroperitoneum, scrotum, and bone marrow.[1],[2] Lymphangioma is commonly benign; however, in rare cases, it may enlarge and cause significant clinical symptom such as airway obstruction.

Surgical resection remains the mainstay of treatment. However, recurrence and local nerve damage were the most common complications for surgical approach.[3] Kennedy reported that the rate of recurrence of lymphangioma resection was 15%–53%.[4] As lymphangiomas commonly affect various structures, the surgical resection was usually done in several stages, thus exposing the patient with multiple surgical procedures.

Nonsurgical treatment of lymphangioma includes radiotherapy, chemotherapy, and sclerosing agent. Radiotherapy for lymphangioma may lead to various complications, such as esophagitis, tracheitis, and malignancy. Thus, it was abandoned in the 1930s.[5] Chemotherapy was reserved for those unresponsive to other treatment.

Various agents had been proposed as sclerosing drugs. The idea was to damage the epithelium lining of the cyst, accompanied by decreased lymph fluid production, causing the collapse of the cyst. However, sclerosing agent may spread outside the lesion after injection, thus causing damage to surrounding structures and complicated surgical procedure.[6]

Bleomycin was one of the antibiotics with antitumoral activity, with a good outcome reported in 88% of cases.[7],[8] A study by Dubois[9] reported Ethibloc to be a sclerosing agent with a good outcome in 60% of cases; however, it may cause discoloration of the skin weeks to months after injection. Giguère et al.[3] conducted a prospective randomized trial that concluded Picibanil to be effective for the treatment of lymphangioma. 86% of the subjects in the study reported a more than 60% regression of lymphangioma size. In a study by Luzzatto et al., 10 out of 15 patients treated with Picibanil were reported to be completely cured, with shrinkage of the lesion observed in all patients.[6] However, it is to be noted that the long-term impact of Picibanil is still unclear.

In this report, we presented a 3-month-old boy with congenital lymphangioma on the right neck and shoulder who will undergo resection of the tumor and sclerotherapy with bleomycin.


  Case Report Top


A 3-month-old boy with a congenital mass located on the right neck and right shoulder was scheduled for tumor resection and injection of sclerosing agent [Figure 1] and [Figure 2]. The patient had inspiratory stridor that was worsening since birth, without other clinical presentations of airway obstruction. Positioning did not affect upper airway obstruction of the patient. There was no history of dyspnea or cyanosis. Computed tomography of the thorax revealed multiple lesions on the bilateral neck, right supraclavicular and right supra-anterior to the superior mediastinum [Figure 3].
Figure 1: Chest X-ray of the patient

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Figure 2: Thorax computed tomography scan of the patient

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Figure 3: Clinical appearance of the patient

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In this patient, induction was performed using sevoflurane to maintain spontaneous ventilation. Fentanyl 2 μg/kg body weight was given to facilitate intubation, without the use of muscle relaxant. During intubation, slight swelling of the larynx was found. There was no difficulty in visualization of the larynx. Intubation was performed using an endotracheal tube (ETT) with 4.0 internal diameter. The intubation was uneventful. There was slight air leak after the placement of ETT, managed by the application of sterile gauze over the ETT. Surgical resection of the lymphangioma on the right neck and shoulder was performed, followed by injection of the sclerosing agent (bleomycin) into the surgical area and into the mass on the left neck. The surgery was uneventful.

Vital signs were stable throughout the surgery without significant blood loss. Manipulation to the trachea was minimal, and there was no damage to recurrent laryngeal nerve. After the surgery, with adequate spontaneous breathing, the extubation was carried out once the patient was fully awake. However, after extubation, retraction in the suprasternal, intercostals, and epigastrium with inspiratory stridor were observed. Hence, reintubation was performed. Laryngoscopy examination revealed worsening edema of the larynx. Extubation was done in the following day after careful positioning in intensive care unit [Figure 4].
Figure 4: Airway edema was seen during reintubation

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  Discussion Top


Airway obstruction was a devastating complication in lymphangioma involving the head-and-neck region. It may be caused by mechanical compression or by airway edema due to obstruction of lymphatic drainage.[2] This case presents the latter condition, in where obstruction of lymphatic drainage led to the manifestation of laryngeal edema. Clinically, this manifested as inspiratory stridor presenting from birth. Tracheostomy may be performed during severe respiratory distress; however, it was deemed unnecessary in this case.

Surgical excision remained the mainstay of treatment for lymphangioma, followed by aspiration, radiation, and sclerosing agent.[5] Lymphangioma may resolve spontaneously; however, this approach was reserved for rare asymptomatic cases. Surgical excision was indicated in patients exhibiting obstructive symptoms, infection, bleeding, and significant cosmetic deformity.[10]

The surgery was uneventful, without significant changes in vital sign or blood loss. Airway manipulation was minimal, and the recurrent laryngeal nerve was preserved; thus, extubation was done after ensuring the patient had recovered from anesthesia, and adequate breathing was observed. However, following extubation, the patient had worsening airway obstruction compared to the symptoms before the surgery. Reintubation was performed using a smaller tube. Surgical excision of the lymphangioma may cause temporary obstruction of lymphatic drainage, thus resulting in worsening airway edema. After careful head-up positioning in the intensive care unit for 24 h, airway edema was relieved and the patient was successfully extubated.

Airway obstruction after extubation requiring reintubation may be caused by hypoventilation, hypoxemic respiratory failure, inability to protect the airway, inadequate clearance of pulmonary secretion, inadvertent extubation, entrapment, laryngeal edema, laryngeal spasm, injury to airway, and postobstructive pulmonary edema.[11] In this case, the indication for reintubation was laryngeal edema. Laryngeal edema may be caused by the injury to laryngeal mucosa, tight-fitting ETT, and laryngeal or tracheal injury.[12]

Throughout the surgery, airway manipulation was minimal and airway structures and nerves were preserved; thus, injury to larynx and trachea was unlikely. Tight-fitting ETT was unlikely as there was slight leakage of the air right after the first intubation. The first intubation was smooth and uneventful; thus, it was unlikely to cause trauma to the larynx. In this case, laryngeal edema was caused by the obstruction of the lymphatic drainage, which was present since before the intubation. However, after the surgery, there was worsening of edema. It may be caused by inflammatory response toward lymphatic drainage, thus worsening lymphatic outflow obstruction. After careful positioning in the intensive care unit, the obstruction resolved and the patient was successfully extubated.


  Conclusion Top


Even after ensuring extubation criteria were fulfilled, airway obstruction after extubation should be considered, especially in the case of head-and-neck lymphangiomas. Complete preparation of emergency and difficult airway devices has to be done during intubation and extubation, especially in patients with possibility of difficult airway.

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Orvidas LJ, Kasperbauer JL. Pediatric lymphangiomas of the head and neck. Ann Otol Rhinol Laryngol 2000;109:411-21.  Back to cited text no. 1
    
2.
Shimizu J, Taga T, Kishimoto T, Ohta M, Tagawa K, Kunitsu T, et al. Airway obstruction caused by rapid enlargement of cervical lymphangioma in a five-month-old boy. Clin Case Rep 2016;4:896-8.  Back to cited text no. 2
    
3.
Giguère CM, Bauman NM, Sato Y, Burke DK, Greinwald JH, Pransky S, et al. Treatment of lymphangiomas with OK-432 (Picibanil) sclerotherapy: A prospective multi-institutional trial. Arch Otolaryngol Head Neck Surg 2002;128:1137-44.  Back to cited text no. 3
    
4.
Kennedy TL. Cystic hygroma-lymphangioma: A rare and still unclear entity. Laryngoscope 1989;99:1-10.  Back to cited text no. 4
    
5.
Padwa BL, Hayward PG, Ferraro NF, Mulliken JB. Cervicofacial lymphatic malformation: Clinical course, surgical intervention, and pathogenesis of skeletal hypertrophy. Plast Reconstr Surg 1995;95:951-60.  Back to cited text no. 5
    
6.
Luzzatto C, Midrio P, Tchaprassian Z, Guglielmi M. Sclerosing treatment of lymphangiomas with OK-432. Arch Dis Child 2000;82:316-8.  Back to cited text no. 6
    
7.
Yura J, Hashimoto T, Tsuruga N, Shibata K. Bleomycin treatment for cystic hygroma in children. Nihon Geka Hokan 1977;46:607-14.  Back to cited text no. 7
    
8.
Orford J, Barker A, Thonell S, King P, Murphy J. Bleomycin therapy for cystic hygroma. J Pediatr Surg 1995;30:1282-7.  Back to cited text no. 8
    
9.
Dubois J. Lymphangiomas in children: Percutaneous sclerotherapy with an alchoholic solution of zein. Radiology. 1997;204:651-4.  Back to cited text no. 9
    
10.
Sannoh S, Quezada E, Merer DM, Moscatello A, Golombek SG. Cystic hygroma and potential airway obstruction in a newborn: A case report and review of the literature. Cases J 2009;2:48.  Back to cited text no. 10
    
11.
Wittekamp BH, van Mook WN, Tjan DH, Zwaveling JH, Bergmans DC. Clinical review: Post-extubation laryngeal edema and extubation failure in critically ill adult patients. Crit Care 2009;13:233.  Back to cited text no. 11
    
12.
Khemani RG, Hotz J, Morzov R, Flink R, Kamerkar A, Ross PA, et al. Evaluating risk factors for pediatric post-extubation upper airway obstruction using a physiology-based tool. Am J Respir Crit Care Med 2016;193:198-209.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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