Pediatric Case of Ludwig’s Angina (LA), Pakistan

Ludwig's angina, first define by Wilhelm Fredrick von Ludwig in 1836, is a serious and potentially lethal rapidly expanding cellulitis of soft tissue of the neck and floor of mouth ⁽²⁾with approximately 10% to 17% mortality from this infection in the pediatric population ⁽³⁾.Mortality rate reported previously as high as 54% to 60% ⁽⁴⁾  due to airway obstruction, that is the most fatal complication cause by inflexible fast growing indurated swelling  displacing anatomical structures of oral cavity and oropharynx. The delay in diagnosis and management can lead to difficult airway especially in children due to high position of  the larynx in the neck and like in adult patients we cannot do  technique of choice i.e. blind nasal intubation or awake fiberoptic ⁽⁵⁾. It is still a nightmare for an anesthesiologist as life-threatening acute airway obstruction despite marked reduction in mortality have been observed with early recognition, efficient antibiotics and modern dental care^{. (5)}

LA mostly affects adult population with poor dentition while incidence of LA in the pediatric population 27-30%. LA with identified odontogenic source only 50% of the cases in children but in adults incidence of odontogenic cause 70% to 90%.   LA is two to three times more prevalent in boys ⁽⁶⁾. As many as 1 in 3 to 4 cases of Ludwig’s angina are reported to occur in children ⁽³⁾ . Oral mucosa lacerations, submandibular sialadenitis ⁽⁷⁾ and mandibular fractures are other etiologic sources in children.11 children with systemic diseases, Tetralogy of Fallot herpetic gingivostomatitis, ⁽⁸⁾tongue piercing and lymphatic vascular malformation superinfection as causative factors of LA have been reported. It can also occur without any predisposing or precipitating cause ⁽⁶⁾.

It is essential to know about the complex anatomy of head and neck for better understanding of the disease progression within the potential spaces of neck form by facial layers attachment to neck structures. The submandibular space, that is the primary site of LA is located above the hyoid bone. The superior border is formed by the mucosa of floor of mouth. It is divided superiorly into the sublingual space which is located between the geniohyoid and mylohyoid muscles and inferiorly into the submaxillary space located between the mylohyoid muscle and the superficial fascia and skin. Ludwig’s angina most frequently originates from the second or third mandibular molars ⁽³⁾ and extends into submaxillary space through extension below mylohyoid line. Involvement of floor of mouth displaces tongue posteriorly resulting in life threatening airway obstruction but due to hyoid bone it does not spread inferiorly but can present as on the anterior aspects of the neck resulting in bull neck. The infection spreads along the facial planes with tendency to involve parapharyngeal, retropharyngeal space, superior mediastinum and not via the lymphatic system ⁽³⁾.

Clinical presentation of LA can vary from focal to systemic signs and symptoms which include tongue, throat pain, dysphagia, trismus, dysphonia, drooling from mouth with fever, chills, malaise, decreased oral intake causing dehydration and toxic appearance. Usually have progressive bilateral submandibular and submental neck swelling which on local physical examination can be soft to firm, with or without fluctuation, warm, tender induration of the floor of the mouth and late presentation causes posterior and superior displacement of the tongue obstructing the airway with or without trismus ⁽⁹⁾. Severe Airway compromise can lead to respiratory symptoms like dyspnea, cyanosis, stridor, altered levels of consciousness, labored breathing and oxygen desaturation. LA mostly diagnosed clinically as in our case. If a child comes with firm edematous tongue or floor of the mouth with neck swelling below the mandible than pediatrician must suspect LA as these are the cardinal signs of  progression to airway obstruction.  ⁽¹⁰⁾

Variations of clinical features and severity with time of presentation, atypical cases without predisposing cause can delay the diagnosis and increase mortality. Mostly fever and neck swelling with bilateral submandibular swelling and elevation of the tongue are the presenting complains ⁽¹⁰⁾  like in our case. Plain neck-and-chest  radiographs can help in diagnosis showing swelling, airway compression. Unstable or patient unable to lie supine point-of-care sonographs give the details of collection while computed tomography (CT) scan is recommended before surgical intervention in stable patient which will give information about the extent of soft-tissue inflammation and the infected spaces with respective anatomical location ⁽⁷⁾

Organisms isolated from bacterial cultures after surgical drainage of Ludwig’s abscesses usually showed mixed growth of both aerobic and anaerobic bacteria, including streptococcus, staphylococcus, and Bacteroides. The predominant microorganisms isolated from the discharge after Incision and drainage in pediatric Ludwig’s angina cases most commonly is Streptococcus species ⁽¹⁰⁾ which is reported  40%  (3). In our case MRSA was isolated. Other isolates include gram-negative rods 25% and anaerobes were found 20% of cases while 35% of blood cultures in same patient series came positive. Septic shock can develop after the surgical drainage which was not seen in our case and few Patients may require a second operation to improve the drainage. ⁽¹⁰⁾  Admission with close monitoring is important as sequelae of sepsis and progression in severity can develop in any patient.

Emergency and timely consultation is of prime importance for the management of Ludwig’s angina, immediate initiation of medical therapy is crucial. Early airway establishment via endotracheal tube or tracheostomy is recommended in case of detoriation or signs of airway obstruction⁽³⁾.Although changes in anatomy of airway and mobility of tissue creates difficulty in intubation, in certain cases there are chances of complete airway obstruction with the induction of general anesthesia. ^(11). Team of emergency tracheostomy must always be present during intubation process.

Depending upon the causative organism antibiotic is indicated, severely immunocompromised patients should be treated against methicillin-resistant S. Aureus and resistant gram-negative bacterias. Commonly used antibiotics are high-dose penicillin G, along with metronidazole. In patients who are allergic to penicillin , an alternate is clindamycin hydrochloride. ⁽¹⁰⁾The role of steroids has been reviewed in a study using dexamethasone , even then surgery was required in 27 out of 31 patient cases. ^(12).