Kamatani, Takaaki

Maeda, Hiromichi

Okabayashi, Takehiro

Toi, Makoto

Yamamoto, Tetsuya

Yamashita, Koichi

Kochi University Repository

1Bilateral ischemic necrosis of the tongue due to disseminated intravascular 
coagulation
Takaaki Kamatani,a Koichi Yamashita,b Takehiro Okabayashi,c Hiromichi Maeda,c
Makoto Toi,d Tetsuya Yamamoto,a
aDepartment of Oral and Maxillofacial Surgery, bDepartment of Anaesthesiology and 
Critical Care Medicine, cDepartment of Surgery, dDepartment of Pathology, Kochi 
Medical School, Kochi University, Japan
Corresponding author: Tetsuya Yamamoto
Department of Oral and Maxillofacial Surgery,
Kochi Medical School, Kochi University
Kohasu, Oko-cho, Nankoku-city, Kochi 783-8505, Japan
TEL: +81-088-880-2423, FAX: +81-088-880-2424
E-mail address: yamamott@kochi-u.ac.jp
Keywords: tongue, necrosis, DIC
Short title: Tongue necrosis due to DIC
* Manuscript
2Abstract
Ischemic necrosis of the tongue is a rare condition because the tongue has a 
rich blood supply. Temporal arteritis appears to be the most frequent cause of tongue 
necrosis.  We present the case of an 82-year-old man who developed bilateral 
ischemic necrosis of the tongue.  The necrosis was considered as a sequela of 
thrombosis of the lingual artery due to disseminated intravascular coagulation.  To 
our knowledge, this is the first reported case of necrosis of the tongue secondary to 
disseminated intravascular coagulation.
Keywords: tongue, necrosis, DIC
3Introduction
Ischemic necrosis of the tongue is a rare condition because the tongue has a 
rich supply of blood from the bilateral lingual arteries; from branches of the facial, 
pharyngeal and palatine arteries; and due to collateral circulation.  Temporal arteritis 
appears to be the most frequent cause of tongue necrosis, with approximately 60 cases 
having been reported in the literature.6  Necrosis of the tongue has also been reported 
subsequent to intra-arterial vasopressin therapy, transient ischemic attack and intestinal 
infarction.4,5,10  Recently, we encountered a patient with rapid development of 
bilateral ischemic necrosis of the tongue secondary to thrombosis of the lingual artery 
due to disseminated intravascular coagulation (DIC).  To our knowledge, this is the 
first reported case of necrosis of the tongue secondary to DIC.
Case report
An 82-year-old obese (body mass index, 25.7 kg/m2) Japanese man was 
referred to our hospital due to bloody stools.  His past medical history included 
Meniere’s disease, lacuna infarction, diabetes mellitus and lung hernia.  Examination 
after admission revealed hepatocellular carcinoma, abdominal aortic aneurysm, adrenal 
tumor, haemorrhoids, tubular adenoma of the colon and prostate cancer.  The patient 
4underwent partial liver resection and extirpation of the adrenal gland.  Nine days after 
the operation, he had a re-operation due to suture failure.  Anaesthesia was induced
with midazolam, and it was maintained with sevoflurane and fentanyl.  The airway 
was secured with a 7.5-mm oral endotracheal tube via direct laryngoscopy, and no 
compression of the tongue by the oral endotracheal tube was noted during the 
operation.  After the second operation, aspiration pneumonia was observed, and the 
patient was transferred to the intensive care unit.  Laboratory examination at 5 days 
after the second operation indicated leukocytosis with neutrophilia, high levels of 
C-reactive protein (CRP), fibrinogen/fibrin degradation products (FDP), D-dimmer, 
activated partial thromboplastin time (APTT) and prothrombin time (PT), low platelet 
count and low activity of anti-thrombin-III (AT-III) (data not shown).  However, the 
erythrocyte sedimentation rate (ESR) was within the normal range, and negative results 
were obtained in the tests for anti-nuclear antibodies, anti-DNA antibodies and 
rheumatoid factors.  A tracheostomy was performed 10 days after the second 
operation under intravenous sedation and local anaesthesia.  On the occasion of 
extubation of the oral endotracheal tube, necrotic tissue was detected on the bilateral 
anterior one-third of the tongue (Fig. 1).  Infarction was suspected; however, the 
patient did not undergo angiography because of his general condition was poor.  
5Although the mouth was cleaned, the signs of necrosis were not diminished. 
Therefore, the non-haemorrhagic necrotic tissues were resected.  Histological 
examination revealed thrombosis of the peripheral tongue artery and vein, and atrophy 
of the muscle with fatty degeneration (Fig. 2).  The final diagnosis was ischemic 
necrosis of the tongue due to thrombosis of the artery subsequent to DIC.  Although 
aggressive medical treatment with intravenous administration of antibiotics, 
anticoagulants and other supportive therapies was started, the patient’s status rapidly 
deteriorated.  He finally died on day 19 after the second operation.
Discussion
DIC is a complex, systemic, thrombohaemorrhagic disorder characterized by 
an imbalance in the haemostatic process, resulting in extensive thrombosis and 
haemorrhage complications.  Laboratory evidences of DIC are procoagulant 
activation, fibrinolytic activation, inhibitor consumption and biomechanical evidence 
of end-organ damage.1,2  DIC is frequently associated with obstetrical disorders, 
malignant diseases and severe infections.9  Pathological findings in patients with DIC 
include major and small vascular thrombosis, and hemorrhage; vascular thrombosis is
frequently observed in the kidney, skin, brain, lung and heart.8  In DIC, most of the 
6thrombosis develops formed in the systemic circulation, and it eventually impinges
upon smaller vessels.7  Since the distal portion of the infarct has no blood supply, it 
does not undergo inflammatory changes and gets mummified.  In this case, based on 
the laboratory and pathological findings, DIC was the most probable cause of the 
ischemic tongue necrosis.  We believe that thrombosis of the tongue is common but 
that it does not result in clinical infarction because of rich collateral circulation, which 
might have been deficient in this particular patient for some reason.
Tongue necrosis is almost always unilateral, and reports of bilateral necrosis 
are rare.6  The most frequent cause of tongue necrosis appears to be temporal arteritis,
which is a common manifestation of systemic arteritis (giant-cell arteritis).  In 70% of 
patients, the physical symptoms included loss of arterial pulsation and a tender, 
thickened and nodular temporal artery.  Other symptoms, such as ischemic ulcers of 
the scalp, hair loss and jaw claudication may also be observed.4  Estimation of the 
ESR and temporal artery biopsy are generally the most informative diagnostic steps.6  
In the present case, although arterial biopsy was not performed, a diagnosis of 
temporal arteritis was not considered because of the normal level of ESR, palpable 
pulsation of the external carotid arteries and temporal arteries and no histological 
findings of lingual arteritis.
7Endotracheal intubation is a rapid, simple, safe and non-surgical technique 
that achieves all the goals of airway management.  The technique protects the lungs 
from aspiration and permits leak-free ventilation during mechanical ventilation.  
Direct airway injury by an endotracheal tube should be considered as a cause of tongue 
necrosis.3  Although the patient gagged and coughed, no bleeding from the tongue 
was observed, and his tongue was not injured during laryngoscopy.  Prolonged 
pressure to the base of the tongue by an endotracheal tube and the use of mouth packs 
and oral airways should also be considered as causes.  However, in the present case, 
the position of the endotracheal tube was changed for several times everyday, and the 
patient did not use mouth packs or oral airways.  Therefore, we considered the 
thrombosis of the lingual artery due to DIC as the cause for the tongue necrosis.
Although ischemic tongue necrosis by causes other than the complication of 
giant-cell arteritis is extremely rare, the possibility of tongue necrosis in patients with 
DIC should be considered by physicians.
8References
1. Aboulafia DM, Aboulafia ED. Aortic aneurysm-induced disseminated 
intravascular coagulation. Ann Vasc Surg 1996: 10: 396-405.
2. Bick RL. Disseminated intravascular coagulation: pathophysiological mechanisms 
and manifestations. Semin Thromb Hemost 1998: 24: 3-18.
3. Divatia JV, Bhowmick K. Complications of endotracheal intubation and other 
airway management procedures. Indian J Anaesth 2005: 49: 308-318.
4. Freedman GL, Hooley JR. Ischemic necrosis of the tongue. Report of a case. 
Oral Surg Oral Med Oral Pathol 1967: 24: 821-824.
5. Libersa P, Loison-Blanchard C, Nawrocki L, Duquesnoy S. Bilateral necrosis of 
the tongue consecutive to cardiac arrest. J Oral Maxillofac Surg  2002: 60:
322-323.
6. Patterson A, Scully C, Barnard N, Griffiths MJ, Eveson JW, Novelli M.  Necrosis 
of the tongue in a patient with intestinal infarction.  Oral Surg Oral Med Oral 
Pathol  1992: 74: 582-586.
7. Robboy SJ, Major MC, Colman RW, Minna JD. Pathology of disseminated 
intravascular coagulation (DIC). Analysis of 26 cases.  Hum Pathol 1972: 3:
327-343.
98. Ueda K, Sugiura M, Ohkawa S, Hiraoka K, Mifune J, Matsuda T, Murakami M. 
Disseminated intravascular coagulation in the aged complicated by acute 
myocardial infarction. Jpn J Med  1981: 20: 202-210.
9. Wada H, Gabazza EC, Asakura H, Koike K, Okamoto K, Maruyama I, Shiku H, 
Nobori T.  Comparison of diagnostic criteria for disseminated intravascular 
coagulation (DIC): diagnostic criteria of the International Society of Thrombosis 
and Hemostasis and of the Japanese Ministry of Health and Welfare for overt DIC. 
Am J Hematol 2003: 74: 17-22.
10. Zelch MG, Geisinger MA, Risius B. Tongue necrosis after intraarterial 
vasopressin therapy.  AJR Am J Roentgenol 1985: 144: 1283-1284.
10
Figure legends
Fig. 1: Anterior and bilateral lingual ischemia with necrosis.
Fig. 2: Thrombosis of the peripheral artery (A) and vein (B).  Hematoxylin and eosin
staining (×100).
Figure 1
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Figure 2
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Bilateral ischemic necrosis of the tongue due to disseminated intravascular coagulation

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