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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 4  |  Issue : 2  |  Page : 132-134

Emergency airway management of a patient with Rosai-Dorfman disease


1 Department of Trauma and Emergency, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
2 Department of Anesthesiology and Critical Care, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
3 Department of ENT and NHS, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India

Date of Submission03-Jun-2021
Date of Acceptance02-Jul-2021
Date of Web Publication10-Aug-2021

Correspondence Address:
Dr. Chitta Ranjan Mohanty
Department of Trauma and Emergency, All India Institute of Medical Science, Bhubaneswar - 751 019, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/arwy.arwy_32_21

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  Abstract 


Rosai-Dorfman disease is a rare histiocytic disorder that involves the over-production of non-Langerhans sinus histiocytes. It presents with cervical lymphadenopathy and huge neck swelling that could prove to be a challenging airway and a nightmare for anaesthesiologists. We discuss the airway management of a patient with Rosai-Dorfman disease requiring an emergency tracheostomy as a life-saving measure.

Keywords: Difficult airway, Rosai-Dorfman disease, tracheostomy


How to cite this article:
Mohanty CR, Sahoo S, Ghoshal P, Shaikh Z, Behera KK. Emergency airway management of a patient with Rosai-Dorfman disease. Airway 2021;4:132-4

How to cite this URL:
Mohanty CR, Sahoo S, Ghoshal P, Shaikh Z, Behera KK. Emergency airway management of a patient with Rosai-Dorfman disease. Airway [serial online] 2021 [cited 2021 Dec 2];4:132-4. Available from: https://www.arwy.org/text.asp?2021/4/2/132/323577




  Introduction Top


Rosai-Dorfman disease or sinus histiocytosis with massive lymphadenopathy is a rare histiocytic disorder that involves over-production of non-Langerhans sinus histiocytes that accumulate mostly in lymph nodes but also in other parts of the body and lead to organ damage.[1] Rosai-Dorfman disease with cervical lymphadenopathy and huge neck swelling can produce a challenging airway that can be a nightmare for anaesthesiologists.[2] We present the airway management of a patient with Rosai-Dorfman disease who had a huge neck swelling and sudden onset of breathlessness requiring an emergency tracheostomy as a life-saving measure.


  Case Report Top


A 44-year-old man presented with a huge left-sided neck swelling for the past 1.5 years. It was associated with another swelling on the right side of the neck that arose 2–3 months later, both of which gradually increased in size. He complained of dysphagia, dyspnoea, intermittent oral and nasal bleed and burning sensation of hot and spicy food. He had a history of type 2 diabetes mellitus for 15 years, hypertension for 10 years and a cerebrovascular accident 3 years prior. He was on aspirin 75 mg, clopidogrel 75 mg and rosuvastatin 10 mg every day. On examination, the patient had bilateral neck swellings. There was a 15 cm × 10 cm large swelling over the left cheek and left mandible with minimal extension into the neck. There was another swelling measuring 8 cm x 10 cm on the right side extending from the midline anteriorly to the right posterior triangle in the right supraclavicular region [Figure 1]. The swellings did not move with deglutition. Although the trachea could not be visualised, it was just palpable above the suprasternal notch. The preoperative positron emission tomography-computed tomography (PET-CT) scan image [Figure 2]a showed a nodal mass on the right side pushing the larynx to the left and compressing the right jugular vein. It was extending into the retropharyngeal space, pushing the larynx and oesophagus anteriorly abutting prevertebral fascia (blue arrows). A mass was seen on the left side encasing the left jugular vein. It was pushing the larynx and oesophagus anteriorly and abutting the prevertebral fascia (red arrows). [Figure 2]b shows PET-CT image showing compression of the trachea due to large cervical mass (green arrow).
Figure 1: 44-year-old-male with bilateral neck swellings. The swelling over the left cheek and left mandible with minimal extension into the neck. The other swelling on the right side extends from the midline anteriorly to the right posterior triangle in the right supraclavicular region

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Figure 2: (a) Preoperative positron emission tomographycomputed tomography (PET-CT) scan image showing a nodal mass on the right side pushing the larynx to the left and compressing the right jugular vein. It was extending into the retropharyngeal space, pushing the larynx and oesophagus anteriorly (blue arrows). Another mass on the left side encasing the left jugular vein and pushing the larynx and oesophagus anteriorly (red arrows), (b) PET-CT image showing compression of the trachea due to large cervical mass (green arrow)

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While awaiting surgery, the patient had an episode of sudden-onset breathlessness with a saturation of 92% on room air and a respiratory rate of 28 per min. It was decided to perform an emergency tracheostomy to secure the airway and relieve his distress. In the preoperative assessment, the patient had a mouth opening of 2.5 fingers, a modified Mallampati Class IV and limited neck movements.

Anticipating difficult mask ventilation, difficult intubation and difficult tracheostomy, an awake fibreoptic intubation (AFOI) was planned. The patient was explained the procedure and consent obtained. As the fasting status was not adequate, intravenous (IV) metoclopramide 10 mg was administered. Nebulisation was given with 4 mL of 4% lignocaine and xylometazoline (0.1% w/v) nasal drops were instilled in both the nostrils. Lignocaine (10%) was sprayed onto the posterior pharynx and injection glycopyrrolate 0.2 mg IV was administered as an antisialogogue. Dexmedetomidine infusion was started at 1 μg/kg administered over 10 min followed by an infusion of 0.3 μg/kg/h. The otorhinolaryngology team was standby inside the operation theatre for performing an emergency tracheostomy should the need arise. After 15 min of dexmedetomidine infusion, lignocaine jelly was applied to the nostrils and gentle dilatation was performed with a nasopharyngeal airway. The patient was preoxygenated for 5 min with 100% oxygen. A 6.0 mm ID endotracheal tube (ETT) was preloaded onto a flexible fibreoptic bronchoscope (FOB) which was then introduced via the right nostril and advanced. On the visualisation of the epiglottis, 4 mL of 2% lignocaine was sprayed over the laryngeal structures through the working channel of the FOB to produce topical anaesthesia and obtund airway reflexes. The FOB was advanced through the glottis until the tip reached the carina. The preloaded ETT was introduced after giving fentanyl 150 μg and propofol 100 mg IV. After confirming the presence of at least 3 consecutive capnographic waveforms on the monitor, vecuronium 6 mg IV was administered, bilateral air-entry was checked and the ETT secured [Figure 3]. General anaesthesia was maintained with isoflurane. The tracheostomy was done uneventfully, and correct placement of the tracheostomy tube confirmed by capnography.
Figure 3: Shows the intraoperative image of the patient after the airway secured with a nasotracheal tube

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  Discussion Top


AFOI is the gold standard for the management of an anticipated difficult airway.[3],[4],[5] In our patient, due to anticipated difficult mask ventilation, difficult intubation and difficult tracheostomy, AFOI was opted for. This would allow spontaneous ventilation to be retained until a definitive airway is established and help in the later performance of a tracheostomy. If it were an elective case, cardiopulmonary bypass with femoral venous and arterial cannulation would have been a better choice. In an emergency like this, a procedure such as jet ventilation would have proven extremely useful when emergency surgical neck access was not possible.[6]


  Conclusion Top


We conclude that AFOI would prove a boon in a difficult airway due to extensive neck swellings when spontaneous ventilation must be maintained and no backup plan is available.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the forms, the patient has given his consent for images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Ong YB, Ping LS, Kiang KF, Ching GS, Madatang AB, Samsudin AT. Rosai Dorfman disease: A case report. Int J Otorhinolaryngol Head Neck Surg 2018;4:582-4.  Back to cited text no. 1
    
2.
Zhang J, Chung F, Hung OR. Management of a patient with OSA for total thyroidectomy. In: Hung OR, Murphy MF, editors. Hung's Difficult and Failed Airway Management. Beijing: McGraw-Hill Education; 2018. p. 481-91.  Back to cited text no. 2
    
3.
Johnston KD, Rai MR. Conscious sedation for awake fibreoptic intubation: A review of the literature. Can J Anaesth 2013;60:584-99.  Back to cited text no. 3
    
4.
Sethi S, Mohanty CR. Trachlight-guided intubation in small infant with difficult airway. J Anaesthesiol Clin Pharmacol 2015;31:275-6.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Mohanty CR, Ahmad SR, Behera BK, Bellapukonda S. Difficult and failed airway in small neonates: Lightwand revisited. J Anaesthesiol Clin Pharmacol 2020;36:130-1.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Parthasarathy S, Nag K, Sivashanmugham T, Karthikeyan P, Ravishankar M. Anesthetic management of excision of laryngocele-role of transtracheal jet ventilation. J Anaesthesiol Clin Pharmacol 2018;34:124-5.  Back to cited text no. 6
[PUBMED]  [Full text]  


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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