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

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.

The mortality rate reported by Ludwig approached 60%. [9] The mechanism of death was originally attributed to sepsis, but by the 1900s it had become evident that death occurred because of airway obstruction, as pressure on the airway resulted in asphyxia. Another factor that has been implicated in death is the impairment of the medullary respiratory center by acapnia or hypersensitivity of the carotid sinus pressure receptors. [10] The high mortality rate of this disease persisted even after the advent of surgical decompression as a treatment because either the procedure was undertaken too late or the drainage of the infection was inadequate. [11] It was not until the antibiotic era and the more widespread practice of good oral hygiene that the mortality rate dropped to less than l0%. [2,12] In 1982, Patterson et al reported no deaths or complications in a series of 20 patients. [3]

A thorough understanding of the anatomy of the spaces of the deep neck and the fascial planes is a prerequisite for treating this disease process properly. Detailed anatomic descriptions of the fascia and fascial planes have been published by many authors [13,14]--most notably by Grodinsky and Holyoke [15] in 1939. They described the submandibular space as a potential space above the hyoid bone. The submandibular space is made up of both the sublingual space, which lies superior to the mylohyoid muscle, and the submandibular space, which lies below the muscle. These spaces can be considered as one single unit because the free border of the mylohyoid muscle posteriorly allows them to communicate (figure 3). The superficial layer of the deep cervical fascia acts as a barrier to the spread of infection. Along with the mandible and the hyoid bone, the fascia limits the amount of edema that can occur. Any significant swelling that arises in the submandibular space will cause a superior and posterior displacement of the floor of mouth and the tongue. Airway compromise can thus ensue.

The superficial layer of the deep cervical fascia also envelops the submandibular gland. Any infect ion or swelling that occurs in this gland is first contained by this layer. However, any prolonged swelling and inflammation can weaken the fascia and allow the infection to rapidly spread into the submandibular space. [11,16,17]

Our current understanding of Ludwig's angina is that it is a potentially lethal, rapidly spreading cellulitis of the sublingual and submandibular spaces. The clinical features of this inflammation include swelling under the tongue, a wood-like swelling of the neck, and difficulty with speech, deglutition, and occasionally respiration. Grodinsky developed strict criteria for the diagnosis of Ludwig's angina. [14] He said the disease can be recognized by five identifying characteristics: (1) the infection is a cellulitis of the submandibular space, not an abscess; (2) it never involves only one space, and it is usually bilateral; (3) the cellulitis causes gangrene with serosanguineous infiltration and very little or no frank pus; (4) the cellulitis attacks the connective tissue, fascia, and muscles, but not the glandular structures; and (5) the cellulitis is spread by continuity, not by the lymphatics.

An odontogenic disorder is the most common etiology, accounting for approximately 70% of cases [5] Tschiassny described how the roots of the second and third lower molars penetrate the thin inner cortex of the mandible and extend inferiorly to the insertion of the mylohyoid muscle. [1] A periapical abscess can result in an infection of the submandibular space. Mandibular trauma, penetrating injuries of the floor of mouth, otitis media, oral neoplasms, and lymphangiomas have all been reported as potential causes of Ludwig's angina. Despite Grodinsky's strict criteria, sporadic cases of submandibular infections have also been recorded in the development of Ludwig's angina. Bilateral sialadenitis and sialolithiasis in and of itself is a rare entity. [18] However, in our patient, infections clearly arose from both submandibular glands, and stones were present bilaterally. The progression of infection from both glands, coupled with the edema of the floor of mouth and the tongue, forms a picture that is consistent with Ludwig's angina.

Because the incidence of Ludwig's angina has steadily declined, fewer physicians are experienced in diagnosing it and in identifying the etiologic agent. In the case presented here, an otolaryngology consultation was obtained only after evaluation by the emergency room and oral surgery staffs and CT. Airway management should remain the primary therapeutic concern. Stridor, difficulty managing secretions, and cyanosis are the late manifestations of impending airway obstruction. Management should be tailored to each patient and to the experience of the treating physician. Some patients can be managed adequately with intravenous antibiotic therapy and observation in a monitored care setting. In others (e.g., those with a more tenuous airway and those scheduled for surgery), the airway must be secured. Routine orotracheal intubation is usually not feasible in view of the edema and swelling that this disease causes. Fiberoptic nasotracheal intubation is an acceptable method, but it requires an experienced anesthe siologist. Tracheotomy, which has long been considered the gold standard, might be necessary in a severely compromised patient. [19] Aggressive antibiotic therapy and decompression of the submandibular spaces can be instituted once the airway has been deemed secure.

When incision and drainage is performed, the incision can be made intra- and/or extraorally. When the submandibular gland has been identified as the source of infection, it should he removed.[11,16,17] Moreover, Colp has suggested that the removal of the gland will also allow for adequate drainage of the fascial spaces. [11]

One must be cognizant that removal of the gland during an infection can lead to an increase in injury to the hypoglossal or facial nerve.

References

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(3.) Patterson HC, Kelly JH, Strome M. Ludwig's angina: An update. Laryngoscope 1982;92:370-8.

(4.) Finch RG, Snider GE, Sprinkle PM. Ludwig's angina. JAMA 1980;243:l171-3.

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(8.) Hall SF. Ludwig's-like angina (pseudo-angina Ludovici). J Otolaryngol 1984;13:321-4.

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(10.) Lindner HH. The anatomy of the fasciae of the face and neck with particular reference to the spread and treatment of intraoral infections (Ludwig's) that have progressed into adjacent fascial spaces. Ann Surg 1986;204:705-14.

(11.) Colp R. The treatment of deep neck infections of the submaxillary triangle. Surg Clin North Am 1933;13:315-8.

(12.) Toffel M. Harvey SC. Ludwig's angina: Analysis of 46 cases. Surgery 1942;11:841-50.

(13.) Thomas TT. Ludwig's angina: An anatomic, clinical, and statistical study. Am Surg 1908;47:161-83.

(14.) Grodinsky M. Ludwig's angina: An anatomical and clinical study with review of the literature. Surgery 1939;5:678-96.

(15.) Grodinsky M, Holyoke E. The fasciae and fascial spaces of the head, neck, and adjacent regions. Am J Anat 1938;63:367-407.

(16.) Lerner DN, Troost T. Submandibular sialadenitis presenting as Ludwig's angina. Ear Nose Throat J 1991;70:807-9.

(17.) Ramsdell EG. Ludwig's angina: Advantages of submaxillary resection. Surg Clin North Am 1934;14:315-25.

(18.) Lutcavage GJ, Schaberg SJ. Bilateral submandibular sialolithiasis and concurrent sialadenitis: A case report. J Oral Maxillofac Surg 1991;49:1220-2.

(19.) Marple BF. Ludwig angina: A review of current airway management. Arch Otolaryngol Head Neck Surg 1999;125:596-9.