AMERI CAN JOURNAL OF OTOLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 84–8 6

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Warfarin induced sublingual hematoma: a Ludwig angina mimic☆,☆☆ Ranjan Pathak, MD⁎, Suzanne Supplee, DO, Madan Raj Aryal, MD, Paras Karmacharya, MD Department of Internal Medicine, Reading Health System, West Reading, PA, USA

ARTI CLE I NFO

A BS TRACT

Article history:

Background: Sublingual hematoma is a rare but life-threatening complication of oral

Received 24 July 2014

anticoagulants. It is important to differentiate this from infectious processes like Ludwig's angina. Securing the airway should be a priority and immediate reversal of anticoagulation with close monitoring is required. Case report: We present a case of sublingual hematoma secondary to warfarin therapy without airway compromise which was managed conservatively with reversal of INR with oral vitamin K. Conclusion: Although rare, it is crucial to differentiate sublingual hematomas from infectious processes. Reversal of anticoagulation with low threshold for artificial airway placement in the event of airway compromise is the treatment of choice. © 2015 Elsevier Inc. All rights reserved.

1.

Introduction

Warfarin remains the most commonly prescribed oral anticoagulant in the United States in spite of growing popularity of the newer anticoagulants, with > 25 million warfarin prescriptions in the United States in 2010 [1]. Spontaneous sublingual hematoma is a rare complication which can lead to serious complication like acute airway compromise.

2.

Case report

A 50-year-old female on chronic warfarin therapy for recent pulmonary embolism presented to the emergency department with painful red swelling under her tongue developing rapidly over few hours. About three hours prior, she felt a sore throat with associated right sided neck pain. Shortly ☆

thereafter she developed difficulty swallowing and articulating her speech due to a moderate sized painful mass on the floor of her mouth. She denied recent trauma to the area, epistaxis, melena or hematuria. She did not have any cough, chest pain, difficulty breathing, stridor or drooling. On physical examination, she was afebrile. Her speech was fluent without stridor or respiratory distress. There was slight fullness in the submental region extending inferiorly into the midline, but no ecchymosis of the overlying skin (Fig. 1). Intraoral examination revealed a tense, tender, red-purplish submucosal hematoma measuring 3 cm × 3 cm in the floor of the mouth, displacing the tongue superiorly (Fig. 1). There was no extension of the swelling into the pharynx. Tongue mobility was limited. Laboratory studies revealed a supratherapeutic international normalized ratio (INR) of 5.2. She had a hemoglobin 14.5 g/dL, platelets 269 × 109/L and white cell count 11.9 × 109/L. Her erythrocyte sedimentation rate (ESR) and

Financial disclosures/conflict of interest: None. Funding: None. ⁎ Corresponding author at: Reading Health System, 6th Avenue and Spruce Street, West Reading, PA 19611, USA. Tel.: + 1 484 628 8255; fax: +1 484 628 9003. E-mail address: [email protected] (R. Pathak). ☆☆

http://dx.doi.org/10.1016/j.amjoto.2014.08.008 0196-0709/© 2015 Elsevier Inc. All rights reserved.

AMERI CA N JOURNAL OF OT OLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 84–8 6

Fig. 1 – Sublingual hematoma without evidence of bruising of skin or soft tissue swelling in the neck.

C-reactive protein were normal at 16 mm/h and 0.35 mg/dL respectively. Computed tomography of the neck showed enlargement of the right submandibular gland and possible sialolith with hemorrhage along the floor of the mouth (Fig. 2). Given the absence of airway compromise and systemic signs of infection, the patient was diagnosed with sublingual hematoma. As there were no signs of impending airway compromise, she was managed conservatively with 2.5 mg of oral vitamin K. Her warfarin was held and the INR returned to normal within 48 hours. The hematoma gradually decreased in size with improvement of her tongue mobility over the next 2 days.

3.

Discussion

Warfarin acts as a vitamin K antagonist by binding with the vitamin K 2.3-epoxide reductase in the hepatic microsome and blocking the action of vitamin K dependent factors II, VII, IX, X, protein C and protein S. It is commonly used for chronic anticoagulation in patients with venous thromboembolism and artificial heart valves [2,3]. Warfarin levels are monitored with regular international normalized ratios (INR) with the

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target of 2–3 in venous thromboembolism and 2.5–3.5 in patients with mechanical heart valves. Interaction with drugs like broad spectrum antibiotics, quinidine, salicylate, thyroxine, alcohol and diet often makes anticoagulation with warfarin a challenge. The overall incidence of bleeding in patients on warfarin is about 6.8% [4]. Retropharyngeal, submaxillary and epiglottic hematomas or bleeding has been described in patients on warfarin therapy, but they can be difficult to identify [3]. Sublingual hematomas on the other hand are obvious on direct inspection. In severe cases, the tongue can be displaced superiorly and posteriorly leading to airway compromise and may result in a clinical picture similar to the commonly described Ludwig's angina [3]. It can present with sublingual or neck swelling, sore throat, swallowing difficulty, change in voice, tachypnea or stridor. Ecchymosis in the anterior neck region [3], anoxic brain injury secondary to airway compromise [5], pulmonary edema [6] have also been occasionally described. Differentiation from acute infectious process like Ludwig's angina is crucial as they are managed differently. In the absence of airway compromise necessitating artificial airway, medical therapy with reversal of the coagulopathy with vitamin K or fresh frozen plasma or factor concentrates remains the mainstay of therapy [2,3,7]. The recommended dose of fresh frozen plasma and prothrombin complex concentrate is 4 units with INR greater than 1.5 and 50 units/kg with INR greater than 6 respectively [2]. Although surgical drainage has been described [8], it may not be successful if the bleeding is in the intrinsic muscles of the tongue. Moreover, it carries the risk of increasing soft tissue edema and airway compromise [9]. Lee et al also described the use of medicinal leeches in sublingual hematoma for two hours twice daily with favorable results [9]. Spontaneous resolution usually occurs with normalization of coagulation parameters as in our case [3]. Our patient was managed conservatively with reversal of anticoagulation with vitamin K. Patients with severe airway compromise should be considered a medical emergency and endotracheal intubation may be indicated as life-threatening hemorrhage can occur into the sublingual space rapidly [3]. However, in most cases, intubation may be difficult due to large size of the hematoma necessitating a tracheostomy or cricothyrotomy instead. Prophylactic antibiotics are not indicated as abscess formation usually does not occur [3]. Patients may be restarted on warfarin with regular monitoring of INR once the hematoma resolves [3]. Sublingual hematomas are rare but potentially serious complications of warfarin therapy. It is important to differentiate them from infectious processes. Reversal of anticoagulation with low threshold for artificial airway placement in the event of airway compromise is the treatment of choice.

REFERENCES

Fig. 2 – Enlargement of the right submandibular gland and hemorrhage along the floor of the mouth.

[1] Johnson JA. Warfarin pharmacogenetics: a rising tide for its clinical value. Circulation 2012;125:1964–6. [2] Cashman E, Shandilya M, Amin M. Warfarin-induced sublingual hematoma mimicking Ludwig angina: conservative management of a potentially life-threatening condition. Ear Nose Throat J 2011;90:E1.

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AMERI CAN JOURNAL OF OTOLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 84–8 6

[3] González-García R, Schoendorff G, Muñoz-Guerra MF, et al. Upper airway obstruction by sublingual hematoma: a complication of anticoagulation therapy with acenocoumarol. Am J Otolaryngol 2006;27:129–32. [4] Bloom DC, Haegen T, Keefe MA. Anticoagulation and spontaneous retropharyngeal hematoma. J Emerg Med 2003;24: 389–94. [5] Rosenbaum L, Thurman P, Krantz SB. Upper airway obstruction as a complication of oral anticoagulation therapy. Report of three cases. Arch Intern Med 1979;139:1151–3.

[6] Bachmann P, Gaussorgues P, Pignat JC, et al. Pulmonary edema secondary to warfarin-induced sublingual and laryngeal hematoma. Crit Care Med 1987;15:1074–5. [7] De Moraes HHA, de Santana ST, Camargo IB, et al. Sublingual hematoma after usual warfarin dose. J Craniofac Surg 2013;24:1858–9. [8] Genovesi MG, Simmons DH. Airway obstruction due to spontaneous retropharyngeal hemorrhage. Chest 1975;68:840–2. [9] Lee NJ, Peckitt NS. Treatment of a sublingual hematoma with medicinal leeches: report of case. J Oral Maxillofac Surg 1996; 54:101–3.

Warfarin induced sublingual hematoma: a Ludwig angina mimic.

Sublingual hematoma is a rare but life-threatening complication of oral anticoagulants. It is important to differentiate this from infectious processe...
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