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Lingual hematoma and heparin-induced thrombocytopenia: a case report.

Abstract

Lingual hematoma is a rare but potentially fatal cause of upper airway obstruction. Patients receiving anticoagulants such as heparin can suffer from significant complications of these medications. Not only does heparin exert effects directly on the coagulation cascade, but it has the potential to cause thrombocytopenia by stimulating formation of antibodies against platelets. We present the case of a patient being treated with heparin for a deep-vein thrombosis, who subsequently developed heparin-induced thrombocytopenia and lingual hematoma, necessitating tracheotomy.

Introduction

Lingual hematoma has been described as one cause of upper airway obstruction. (1-16) Because respiratory compromise often occurs precipitously in patients with lingual hematoma, the importance of recognizing this unusual clinical entity early in its course and taking the appropriate steps to secure the airway cannot be overstated. We describe a previously unreported cause of lingual hematoma and subsequent upper airway obstruction requiring surgical intervention.

Case report

A 44-year-old woman with no significant medical history was admitted to an outside hospital for treatment of right lower extremity (RLE) deep-vein thrombosis. She was started on warfarin and intravenous (IV) heparin, but she soon developed bloody diarrhea and abdominal pain. Both anticoagulants were discontinued, and vitamin K was administered. Of note, a nasogastric tube (NGT) was placed and removed to rule out an upper gastrointestinal source of her bleeding. Gastric lavage was negative.

On hospital day 2, an inferior vena cava filter was placed to reduce the likelihood of pulmonary embolism. Later that day, the patient's abdominal pain worsened. Computed tomography (CT) suggested ischemia of the left colon, and she was taken to the operating room (OR). Exploratory laparotomy confirmed left colonic ischemia, and left hemicolectomy was performed, with placement of another NGT. On hospital day 5, the patient had progressive RLE pain and edema, as well as impending gangrene of her right toes; therefore, IV heparin was restarted, and she returned to the OR for RLE venous thrombectomy.

The NGT was removed on hospital day 8, causing paroxysms of violent coughing. Later that day, the patient complained of dyspnea and dysphagia. Progressive ecchymosis and swelling were noted in the floor of her mouth (FOM) and anterior neck, with superior displacement of her tongue. Her voice became hoarse and then muffled, and she developed moderate inspiratory stridor. However, she required minimal supplemental oxygen. IV heparin was discontinued, and an emergent otolaryngology consult was obtained.

Flexible fiberoptic laryngoscopy demonstrated diffuse ecchymosis and edema of the base of the tongue and supraglottis; the patient's true vocal folds could not be visualized. The patient was again taken to the OR, where an uncomplicated tracheotomy was performed under local anesthesia. Protamine was given during the procedure, and methylprednisolone was started postoperatively. Edema of the tongue, FOM, and anterior neck caused the patient's tongue to protrude from her mouth (figure 1).

IV heparin was held for a total of 48 hours but was then restarted to treat a new left lower extremity deep-vein thrombosis. At that time, the patient was transferred to our tertiary care center for continued management. Upon her arrival, a complete thromboembolic workup was initiated. Notably, this workup at the previous institution had been negative. Heparin-induced platelet antibodies were identified on the day after transfer; heparin was discontinued and lepirudin initiated.

Our otolaryngology service was consulted for further management of the patient's airway. Her tongue was soft, enlarged, and protruding from her oral cavity. The FOM was woody and indurated, and the patient had submental swelling and firmness. ACT scan demonstrated diffuse enlargement of the oral tongue consistent with a poorly organized lingual hematoma (figure 2), along with significant edema of the FOM and bilateral submandibular spaces.

The patient had formed a lingual/FOM hematoma and was taken to the OR for incision and drainage. An external incision was made between the mentum and the hyoid bone, and a pocket deep to the geniohyoid muscle was entered, yielding 40 ml of foul-smelling liquefied blood clot. Culture eventually yielded Streptococcus viridans.

A Penrose drain was left in place until postoperative day 5. Tongue edema, FOM fullness, and neck swelling gradually resolved over the next few weeks. Tongue mobility improved slowly as the patient worked with speech pathology. Her tracheotomy tube was decannulated on postoperative day 27, and she was discharged the following day. Within weeks, the patient was taking oral solids and liquids without difficulty. Although there was delayed return of tongue mobility, it returned essentially to baseline by 1-year follow-up.

Discussion

Trauma (1,3,5,7,9,11,15) and anticoagulation with heparin, warfarin, streptokinase, and/or tissue plasminogen activator (2,4,6,8,10,13,14,16) are recognized as the most common causes of lingual hematoma. Additionally, one case has been attributed to severe hypertension. (12) In the present report, the potential causes included heparin-induced thrombocytopenia (HIT), anticoagulation with heparin, trauma secondary to NGT placement, coughing/Valsalva maneuver after NGT removal, and multiple orotracheal intubations.

A diagnosis of H IT is made when both of the following criteria are met: a decrease in platelet count of at least 30%, to less than 150 x 109/L, and a positive serologic test for HIT. (17) To date, HIT has not been reported to be associated with the development of a lingual hematoma. However, in the current case, the drop in platelet count (252 x 109/L on admission to 106 x 109/L on hospital day 5) and the identification of heparin-induced platelet antibody confirmed a diagnosis of HIT. Two elements of HIT could predispose patients to development of hematoma: a quantitative decrease in platelets and an immune-mediated endothelial injury.

[FIGURE 1 OMITTED]

Several traumatic events might have played a role in the evolution of our patient's lingual hematoma. Specifically, she had an NGT placed twice, and she was orally intubated twice. In addition, forceful coughing after removal of the second NGT occurred shortly before formation of the hematoma. Heparin's intrinsic anticoagulant activity also might have led to the lingual hematoma, although the partial thromboplastin time remained subtherapeutic at all times.

Most likely, the lingual hematoma described in the current case was the result of several events occurring in combination. HIT likely predisposed the patient for minor trauma (such as orotracheal intubation or violent coughing) to cause vascular interruption and subsequent hemorrhage into the tongue. Thrombocytopenia and heparin's effect on the coagulation cascade might have then allowed progression of hemorrhage and formation of the large lingual hematoma.

Discussion of lingual hematoma as a cause of upper airway obstruction would not be complete without mention of airway management. One must be aware of the potential for lingual hematoma to rapidly and insidiously compromise the airway by superiorly displacing the tongue and FOM. Of 17 cases of lingual hematoma described in the literature, 5 were surgically managed with tracheotomy and 6 were managed with orotracheal or nasotracheal intubation; the remaining 6 patients were observed expectantly.

[FIGURE 2 OMITTED]

Patients with lingual hematoma can present with greatly varying degrees of airway compromise, ac- counting for the wide variation in airway management approaches. (2) In our case, we agreed that tracheotomy under local anesthesia was the most appropriate course of action. Indeed, the potential for lingual hematoma to completely obstruct the upper airway must not be overlooked.

References

(1.) Chase CR, Herbert KC, Farnham JE. Post-traumatic upper airway obstruction secondary to a lingual artery hematoma. J Trauma 1987;27(8):953-4.

(2.) Cohen AF, Warman SP. Upper airway obstruction secondary to warfarin-induced sublingual hematoma. Arch Otolaryngol Head Neck Surg 1989;115(6):718-20.

(3.) Grossman MD, Karlovitz A. Lingual trauma: The use of medicinal leeches in the treatment of massive lingual hematoma. I Trauma 1998;44(6):1083-5.

(4.) Gupta MK, McClymont LG, El-Hakim H. Case of sublingual hematoma threatening airway obstruction. Med Sci Monit 2003;9(11): 95-7.

(5.) Hing NR, Bowler MD, Byth PL, Daly CG. Lingual haematoma leading to upper airway obstruction. Br I Oral Maxillofac Surg 1985;23(5):322-5.

(6.) Jaffrelot M, Bourrigan P, Cracan A, et al. [Lingual haematoma after thrombolysis for acute myocardial infarction.] Ann Fr Anesth Reanim 2005;24(3):306-7.

(7.) Kattan B, Snyder HS. Lingual artery hematoma resulting in upper airway obstruction. J Emerg Med 1991;9(6):421-4.

(8.) Kaynar AM, Bhavani-Shankar K, Mushlin PS. Lingual hematoma as a potential cause of upper airway obstruction. Anesth Analg 1999;89(6):1573-5.

(9.) Lee NJ, Peckitt NS. Treatment of a sublingual hematoma with medicinal leeches: Report of case. J Oral Maxillofac Surg 1996;54(1): 101-3.

(10.) McMechan SR, Morrow B, Campbell NP. Lingual haematoma after treatment with aheplase (recombinant tissue plasminogen activator) for acute myocardial infarction. Br Heart J 1995;74(2):205.

(11.) Ng KP. Lingual haematoma: Yet another unusual cause of upper airway obstruction. Med J Malaysia 1998;53(1):112-14.

(12.) Prepageran N, Raman R, Ismail SM, Rahman ZA. Spontaneous sublingual hematoma as a complication of severe hypertension: First report of a case. Ear Nose Throat J 2002;81(8):576-8.

(13.) Riggs JE. Sublingual hematoma: An unusual complication of heparin anticoagulation. WV Med J 1986;82(5):131.

(14.) Shaps HI, Snyder GE, Sama AE, Rudolph GS. Airway compromise secondary to lingual hematoma complicating administration of tissue plasminogen activator for acute ischemic stroke. Ann Emerg Med 2001;38(4):447-9.

(15.) Shaw RJ, McNaughton GW. Emergency airway management in a case of lingual hematoma. Emerg Med 1 2001;18(5):408-9.

(16.) Williams PJ, Jani P, McGlashan J. Lingual haematoma following treatment with streptokinase and heparin; anesthetic management. Anaesthesia 1994;49(5):417-18.

(17.) Aster RH. Heparin-induced thrombocytopenia and thrombosis. N Engl J Med 1995;332(2):1374-6.

Geoffrey S. Getnick, MD; Samuel J. Lin, MD; Joseph R. Raviv, MD; William E. Walsh, MD; Kenneth W. Altman, MD, PhD

From the Department of Otolaryngology-Head and Neck Surgery, The Feinberg School of Medicine of Northwestern University, Chicago.

Corresponding author: Geoffrey S. Getnick, MD, 7400 France Avenue South, #107, Edina, MN 55435. Phone: (952) 832-5252; fax (952) 548-5254; e-mail: gsgetnick@yahoo.com
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Article Details
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Title Annotation:ORIGINAL ARTICLE
Author:Getnick, Geoffrey S.; Lin, Samuel J.; Raviv, Joseph R.; Walsh, William E.; Altman, Kenneth W.
Publication:Ear, Nose and Throat Journal
Article Type:Report
Geographic Code:1U3IL
Date:Mar 1, 2008
Words:1658
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