Bilateral submandibular gland infection presenting as Ludwig's angina: First report of a case

Ear, Nose & Throat Journal, April, 2001 by Carlo P. Honrado, Samuel M. Lam, Matthew Karen

Abstract

We diagnosed and treated a case of Ludwig's angina in a 45-year-old man who had edema of the floor of month and the tongue along with bilateral submandibular sialadenitis and sialolithiasis. We secured the patient's airway via nasal fiberoptic intubation in the surgical intensive care unit and administered intravenous antibiotics. The edema subsided, and the patient was extubated on the third postoperative day and discharged shortly thereafter. To our knowledge, this is the first reported case of a patient with bilateral submandibular sialadenitis and sialolithiasis presenting as Ludwig's angina. Despite the decreasing incidence of this disease, Ludwig's angina remains an important disease process because a failure to control the airway can have disastrous consequences. Proper diagnosis, airway control, antibiotic therapy, and occasionally surgical management are essential to ensure the safety of the patient.

Introduction

Ludwig's angina is a serious and potentially fatal disease that still receives attention in the otolaryngology and oral surgery literature. In most cases, the primary cause is an odontogenic infection. Other etiologies include peritonsillar and parapharyngeal abscesses, oral lacerations, otitis media, lymphangiomas, and mandibular fractures. [1-6]

Our review of the literature found only sporadic reports of unilateral sialadenitis as a cause of Ludwig's angina. The occurrence of bilateral sialadenitis and sialolithiasis is rare. In fact, in this article we present the first published report of a patient with bilateral submandibular sialadenitis and sialolithiasis presenting as Ludwig's angina.

Case report

A 45-year-old man came to the emergency room complaining of a sore throat, tongue swelling, and bilateral neck and facial swelling. He had no significant medical or surgical history, and he was not taking any medication. He also denied any recent dental work. His temperature on arrival was 100. 1[degrees] F, and the rest of his vital signs were stable. He did not have stridor or cyanosis. His physical examination was significant for tongue swelling with decreased mobility. The floor of his mouth was indurated, swollen, and tender to palpation. He had bilateral submandibular swelling and induration. Laboratory data revealed a white blood cell count (WBC) of 16.7 x 10 [9]/L.

The emergency room personnel consulted an oral surgeon to evaluate the patient for an odontogenic infection, but the surgeon was unable to identify one. A panoramic plain film showed a 1-cm x 5-mm opacity near the body of the right mandible. At that time, the airway was not deemed to be compromised, and the patient was taken for computed tomography (CT) with intravenous contrast. CT revealed the presence of bilateral multiple calculi along the floor of mouth, dilated Wharton's ducts proximal to the calculi, and enlarged submandibular glands with dilated ducts within the glands (figure 1). No discrete abscess or fluid collection was noted.

In view of the CT findings, the otolaryngology service was consulted. The results of the physical examination were confirmed. A flexible fiberoptic nasal examination revealed a normal larynx. A diagnosis of Ludwig's angina was made on the basis of bilateral submandibular sialadenitis with sialolithiasis, which had caused the edema in the floor of mouth and the tongue.

The patient was taken emergently to the operating room for airway protection and incision and drainage. Although his swelling was progressing, it was felt that a nasal fiberoptic intubation was possible in lieu of a tracheotomy (figure 2). After the airway was secured, examination confirmed the presence of palpable stones along the floor of mouth bilaterally and pus expressed from both Wharton's ducts. Blunt dissection of the floor of mouth expressed murky fluid, which was sent for culture. A Penrose drain was placed into the floor of mouth.

The patient remained intubated and was managed post-operatively in the surgical intensive care unit. Intravenous ampicillin/sulbactam (3 g q6h) was initiated. A Gram's stain revealed gram-positive cocci and rare gram-negative rods. Final cultures were positive for a moderate number of Streptococcus salivarius spp., which were sensitive to ampicillin, cefazolin, imipenem, and vancomycin. No anaerobes were isolated. The patient was also evaluated for a systemic etiology for his stones but was found to be normocalcemic.

The patient improved on antibiotics. The swelling of his neck and floor of mouth subsided, and he was extubated on the third postoperative day. The Penrose drain was removed on the following day. The patient was afebrile for 72 hours, and his WBC normalized to 8.0 x [10.sup.9]L. He was sent home on oral amoxicillin/clavulanate (875 mg bid for 2 weeks) and was scheduled to undergo bilateral submandibular gland excisions.

Discussion

The condition we know as Ludwig's angina was mentioned in writings dating back to Hippocrates and Galen. In 1836, German surgeon Wilhelm von Ludwig provided the first detailed description of the disease that now bears his name. [78] Based on his series of cases and autopsies, Ludwig characterized the condition as the "occurrence of a certain type of inflammation of the throat, which, despite the most skillful treatment, is almost always fatal." [9] His autopsy findings included gangrene of the tissues of the submandibular and sublingual spaces and multiple abscesses but a sparing of the lymphatics and salivary glands. [8] The classic description of Ludwig's angina is an inflammation of the cellular tissues that begins around the submandibular gland and subsequently involves the floor of mouth and the neck. Patients who recover do so gradually. Those whose course progressively worsens usually die in 10 to 12 days.