Abstract
Coagulation factor XIII (F-XIII) deficiency is a rare disorder characterized by hemorrhagic diathesis, leading to defective wound healing and prolonged bleeding after trauma. This report describes tooth extraction in a 29-year-old man with F-XIII deficiency. A year earlier, he had experienced odontorrhagia and had been transported to hospital after extraction of the left mandibular third molar. Extensive blood examination was therefore performed this time, revealing F-XIII deficiency (activity level, 25%). Therefore, concentrated F-XIII was administered to increase F-XIII activity before extraction, increasing the F-XIII activity level to 61%. Teeth were then extracted under local anesthesia. Subsequent wound healing was favorable without odontorrhagia. In this time, we described the extremely rare report of F-XIII deficiency diagnosed by examinations immediately before dental treatments that included tooth extraction.
Highlights
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The F-XIII deficiency patients are increasing worldwide.
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First report to detect of F-XIII deficiency on examination immediately preceding dental treatments such as tooth extraction and oral surgery.
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Standardization of methods to deal with F-XIII deficiency should be rapidly considered for dental patient.
1
Introduction
Coagulation factor XIII (F-XIII), also referred to as fibrin stabilizing factor (FSF), was first reported by Laki and Lóránd in 1948 [ ]. The role of F-XIII is to change soluble fibrin to insoluble fibrin [ ]. In addition, F-XIII is associated with the plasmin resistivity of fibrin. F-XIII is therefore considered to be associated with the mechanisms of hemostasis and wound healing [ ]. F-XIII deficiency results in great sensitivity of unstable clots to the fibrinolytic system. F-XIII deficiency can thus result in serious sudden hemorrhage or protracted wound healing [ ].
In the case we describe here, a patient with F-XIII deficiency received injection of concentrated F-XIII preparation prior to tooth extraction.
2
Case report
In October 2015, a 29-year-old man visited our hospital complaining of pain with swelling around the posterior teeth of the right mandible. On initial examination, the patient was 168.0 cm tall and weighed 60.6 kg, with favorable nutrition. No anemia was evident from the palpebral conjunctiva. Physical examination and X-ray of the mouth revealed fracture and mobility of the right mandibular second molar. In addition, the right mandibular third molar was erupting with pericoronitis ( Fig. 1 ). Elicitation of his medical history revealed that when he had undergone extraction of the left mandibular third molar at a dental clinic a year earlier, hemostasis had not been able to be achieved. The patient had then fallen unconsciousness and had been transported and admitted to a dental university hospital. As that time, no abnormal findings had been detected on hematological examination. Hence, tranexamic acid was administered for hemostatic purposes. In terms of family history, his parents had a non-consanguineous marriage of coagulation factor deficiencies, and no members of the family showed any history of bleeding disorders. No abnormalities were identified on hematological, coagulation or biochemical examinations ( Table 1 ). On initial examination, a prosthesis on the right mandibular second molar was removed ( Fig. 2 A), and the decision was made to conduct extensive blood examinations. This revealed low F-XIII activity, at only 25% ( Table 2 ). However, von Willebrand factor was not detected. F-XIII deficiency was therefore diagnosed. Concentrated F-XIII preparation, representing freeze-dried F-XIII derived from human plasma, was prepared before tooth extraction. In addition, a wound protector for hemostasis was prepared for local coagulation. One vial (4 mL) of concentrated F-XIII preparation were injected intravenously the day before and immediately before tooth extraction, respectively. After the second administration of concentrate, F-XIII activity was confirmed to have increased to 61%, the right mandibular second and third molars were extracted under local anesthesia. Absorbable gelatin sponge was inserted into the extraction socket, which was then sutured closed. After extraction, the wound was covered with the wound protector to ensure hemostasis ( Fig. 2 B). On postoperative day 3, F-XIII activity was confirmed to have remained >60% and the patient was discharged from hospital. On postoperative day 7, the extraction socket was removed the stitches ( Fig. 2 C). At one month postoperatively, the extraction socket showed favorable prognosis without protracted wound healing ( Fig. 2 D).
| WBC | 3.9 × 103/μl |
| RBC | 5.46 × 106/μl |
| PLT | 202 × 103/μl |
| PT | 12.5sec |
| PT% | 98% |
| PT-INR | 1.01 |
| APTT | 36.2sec |
| AST | 20U/l |
| ALT | 39U/l |
| BUN | 9.2mg/dl |
| CRE | 0.71mg/dl |
| eGFOcreat | 102.6ml/min/1.73m2 |
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