Sublingual hematoma is characterized by the accumulation of blood within the sublingual and submandibular spaces, resulting in the elevation of the tongue and/or the floor of the oral cavity. This condition can arise from local or systemic factors. Local causes include trauma to the mandible [1,2] or tongue [3,4], as well as surgical procedures [5-7], while systemic causes encompass severe hypertension [8] and the use of anticoagulant medications [9]. Sublingual hematoma is sometimes referred to as “pseudo-Ludwig angina,” a term that reflects the potential for life-threatening airway obstruction associated with the condition [10]. Most documented cases of sublingual hematoma in the literature are related to dental implant placement [5,6]. The prevention of periosteal perforation is a key step in avoiding such complications [11]. However, this may not be feasible during procedures like osteotomy, particularly midline osteotomy, which necessitates breaching the periosteal envelope to permit the movement of bone segments. Although most case reports have attributed sublingual hematoma to implant placement [12], and preventive strategies have been suggested, no instances have been reported of sublingual hematoma associated with midline osteotomy of the mandible. The aim of this case report is to raise awareness about the potential for this complication in the context of osteotomy and to discuss the preventive measures and management options available.

A 43-year-old man presented with a malunited mandibular fracture that necessitated midline osteotomy to correct the mandibular shape and occlusion. The procedure was performed under general anesthesia. A vestibular incision was made to expose the symphysis region, followed by the elevation of a mucoperiosteal flap. A vertical, linear osteotomy line was marked, extending downward from the upper alveolar margin between the central incisors to the inferior border of the mandible. The osteotomy was performed using a surgical handpiece equipped with a #702 surgical cutting bur, which cut through the buccal cortex and into the cancellous bone while sparing the lingual cortex. The osteotomy was completed using an osteotome, and the two halves of the mandible were separated using a Smith spreader. At this point, a brisk bleed was observed in the sublingual region, resulting in rapid elevation of the floor of the mouth and the tongue (Fig. 1). The sublingual region area felt tense and firm. Steady downward intraoral compression of the hematoma, along with extraoral pressure directed superiorly at the chin region, was delivered using a bimanual maneuver for 5 minutes to prevent the hematoma from expanding and to facilitate drainage. The hematoma shrank considerably within minutes. Concurrently, the osteotomized fragments were repositioned and secured with two 2.0-mm miniplates: one at the superior border to avoid injury to the tooth roots and the other at the inferior border. This re-approximation of the hemimandibles prevented further bleeding and growth of the hematoma (Fig. 2). Dental occlusion was restored, and the surgical wound was closed in layers using 3-0 Vicryl suture material. The patient was then extubated without complications. In the immediate postoperative period, the patient’s vitals remained stable. However, the tongue and floor of the mouth turned deep purple, and tongue movements appeared restricted (Fig. 3). The patient was monitored daily with careful attention to vitals and the extent of the swelling (Figs. 4, 5). By the seventh postoperative day, the clinical signs had completely resolved (Fig. 6).

In cases of sublingual hematoma, the hemorrhage is typically attributed to the arterial triad comprising the sublingual, submental, and incisive arteries [13]. Flanagan [14] reported that injury to the sublingual or submental artery can result in the loss of up to 450 mL of blood within 30 minutes. The flow rate, coupled with the anatomy of the oral cavity floor, facilitates the spread of the hematoma into the posterior and inferior sublingual spaces, potentially compromising the airway. Less severe complications associated with sublingual hematoma include drooling, voice changes, sore throat, dysphagia, trismus, neck ecchymosis, tachypnea, and stridor. However, the course of these blood vessels and the consequences of their injury are comparatively poorly understood.

Many surgeons recommend avoiding perforation of the lingual cortex to prevent bleeding and related complications [6]. However, in midline osteotomy, breaching the lingual periosteum is typically inevitable, particularly for procedures requiring transverse expansion of the dental arches. Therefore, the risk of sublingual hematoma must be considered during the planning stage. Mandibular midline osteotomy is indicated for the correction of various clinical conditions, including transverse skeletal discrepancies of the face, long-standing temporomandibular joint dislocation, and malunited mandibular fracture. These procedures require an osteotomy cut that extends from the buccal cortex through to the lingual cortex, enabling the complete separation and mobilization of the hemimandibles to the desired position.

Any violation of the periosteal boundary on the lingual aspect can lead to hemorrhage, with blood flowing unhindered into the sublingual plane due to the lack of a limiting barrier in the form of bone or muscular attachments. To prevent this complication, placing the osteotomy in a paramedian position, which avoids the lingual foramen, has been suggested [25]. In paramedian stepped mandibular osteotomy [26], the upper vertical section of the osteotomy is situated between the ipsilateral central incisor and the lateral incisor or between the lateral incisor and the canine. The middle horizontal section lies between the superior and inferior genial tubercles, where the genioglossus and geniohyoid muscles attach. Above and below these tubercles are the superior and inferior midline lingual foramina [27]. The lower vertical section is positioned between the posterior margin of the mandibular attachment of the digastric muscle anterior belly and the anterior margin of the mental foramen. The osteotomy line is meticulously planned to avoid both the tooth roots and the mental neurovascular bundle. By preserving the midline lingual foramina and their contents, this osteotomy technique helps prevent bleeding. In the present case report, hematoma progression was prevented by achieving hemostasis through the reduction and fixation of fracture fragments. This result stems from the occlusion of any bleeding vessels and the compressive forces exerted by the interfragmentary apposition of the osteotomized fragments. However, a similar approach to hematoma management following genioplasty provided only transient control; the hematoma reappeared in the immediate postoperative period, necessitating intubation [7].

In conclusion, sublingual hematoma requires prompt identification and immediate intervention to prevent complications. Adequate preoperative planning is essential and can be facilitated by the use of virtual surgical models, surgical guides, and computed tomography scans to determine the osteotomy lines before surgery and avoid damaging vital structures near the lingual foramina. Intraoperative navigation may also be employed to assist in guiding the osteotomy procedure. Controlled use of an oscillating saw and osteotome at the lingual cortex can help prevent sublingual hematoma [28]. Management of intraoperative bleeding should adhere to a standardized stepwise protocol, ranging from conservative measures such as the application of pressure to procedures like embolization [29].