Clinical Case Reports - 2022 - Tailor - Parapharyngeal and floor%E2%80%90of%E2%80%90mouth abscess secondary to tonsillar phlegmon A rare.pdf

Received: 17 April 2022 | Revised: 23 August 2022 | Accepted: 26 August 2022 DOI: 10.1002/ccr3.6325 CASE REPORT Parapharyngeal and floor-­of-­mouth abscess secondary to tonsillar phlegmon: A rare and unusual ca...

Received: 17 April 2022 | Revised: 23 August 2022 | Accepted: 26 August 2022 DOI: 10.1002/ccr3.6325 CASE REPORT Parapharyngeal and floor-­of-­mouth abscess secondary to tonsillar phlegmon: A rare and unusual cause of Ludwig's angina Bhavesh V. Tailor | Haran Devakumar | Tharsika Myuran | Dimitrios Ioannidis Department of Otolaryngology, Colchester General Hospital, East Abstract Suffolk and North Essex NHS We present an unusual case of Ludwig's angina secondary to a left tonsillar phleg- Foundation Trust, Colchester, UK mon in a previously fit and well 50-­year-­old woman. This tonsillar phlegmon Correspondence spread along the peritonsillar/parapharyngeal plane to cause a diffuse cellulitis Bhavesh V. Tailor, Department of and collection in the submental, sublingual, and submandibular spaces despite Otolaryngology, Colchester General Hospital, Turner Road, Colchester, empirical intravenous antibiotic therapy. Essex CO4 5JL, UK. Email: [email protected] KEYWORDS abscess, Ludwig's angina, parapharyngeal space, peritonsillar abscess, tonsillitis 1 | I N T RO DU CT ION and consideration of surgical drainage and debridement. Such infections are usually odontogenic in origin.4,5 Peritonsillar infection is a common site of deep neck space We present an unusual case of Ludwig's angina second- infection (DNSI)1 and is often described as part of a spec- ary to a left tonsillar phlegmon. A phlegmon is inflamma- trum of disease from tonsillitis, via peritonsillar cellulitis tion of connective tissue, which in this case spread along and phlegmon, culminating in peritonsillar abscess. Such the peritonsillar/parapharyngeal plane to cause a diffuse presentations are a frequent source of emergency refer- cellulitis and collection in the submental, sublingual, and rals to acute ENT services.2 The mainstay of treatment for submandibular spaces despite empirical intravenous anti- peritonsillar infection is intravenous antibiotics and ste- biotic therapy. roids with attempted drainage of peritonsillar abscess if suspected. In cases of untreated or inadequately treated peritonsillar infection, spread of infection may occur 2 | C ASE REPORT through the deep neck tissues, typically to the parapha- ryngeal or retropharyngeal spaces.3 A previously fit and well 50-­year-­old woman initially pre- Ludwig's angina is a potentially life-­threatening, dif- sented to her general practitioner (GP) with a 3-­day history fuse cellulitis of the soft tissues of the floor-­of-­mouth, of sore throat and given a course of oral penicillin V for involving the sublingual, submental, and submandibular suspected tonsillitis. However, her symptoms progressed spaces. Inflammatory distension of the fascial planes of the rapidly with associated fever and odynophagia, and she neck can progress rapidly, leading to airway obstruction. presented to the emergency department 48 h later. She had Effective treatment involves early recognition to secure no significant medical or surgical history apart from well-­ the airway, intravenous antibiotics and corticosteroids, controlled asthma and denied history of frequent episodes This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. © 2022 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd. Clin Case Rep. 2022;10:e06325.  wileyonlinelibrary.com/journal/ccr3 | 1 of 5 https://doi.org/10.1002/ccr3.6325  | 20500904, 2022, 9, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.6325 by Nat Prov Indonesia, Wiley Online Library on [01/07/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 2 of 5    TAILOR et al. of severe sore throat or tonsillitis and denied dental pain severe pain. Subsequent magnetic resonance imaging or recent dental extraction. The patient was not diabetic (MRI) with contrast demonstrated an inflammatory mass and was not prescribed long-­term corticosteroids or other lesion and multiloculated collection centered on the left immunosuppressive medication. Neck examination re- palatine tonsil, which extended to the left lateral and an- vealed a firm, non-­fluctuant, exquisitely tender swelling terior floor-­of-­mouth, spanning 6.3 cm, in keeping with of the floor-­of-­mouth, left submandibular, and left angle a Ludwig's angina (Figure 2). There was no evidence of of mandible regions, with no associated skin erythema. airway compromise. As for antibiotic therapy, intravenous Examination of the oropharynx was limited by marked metronidazole was continued while intravenous amox- trismus and an elevated tongue. Flexible nasoendoscopy icillin was switched to intravenous co-­amoxiclav on the revealed swelling of the left pharyngeal wall, with no air- advice of the microbiology team. Definitive management way compromise. Blood tests demonstrated neutrophilic entailed hot left tonsillectomy and intraoral abscess inci- leucocytosis and C-­reaction protein level was 343 mg/L. sion and drainage. Preoperatively, two discharging areas The patient was admitted to hospital and received in- were identified at the left floor-­of-­mouth and inferolateral travenous antibiotics, intravenous steroids, fluids, and an- aspect of left tonsillar fossa. Both openings were widened algesia. She was treated with empiric antibiotic therapy intraoperatively to facilitate drainage along with copious for para−/retropharyngeal abscess (amoxicillin, metro- irrigation with saline, and a washout revealed they were nidazole, and gentamicin) as per local hospital protocol. in communication, avoiding the need for a transcervi- An urgent contrast-­enhanced computed tomography (CT) cal drainage approach. A swab was taken, which grew revealed a soft tissue abnormality within the left parapha- Streptococcus anginosus, sensitive to penicillin and eryth- ryngeal space adjacent to the oropharynx but without con- romycin. Tonsil histology showed ulceration and neutro- vincing evidence of enhancement (Figure 1). A diagnosis philic abscess formation. After 48 h postoperatively, her of left tonsillar phlegmon was made. Needle aspiration pain was well-­controlled, she was managing adequate oral of the left peritonsillar space was attempted but failed to intake, and clinical examination demonstrated minimal yield any pus. The maxillofacial team was consulted, who neck swelling and a healthy tonsillar bed. Following dis- deemed an odontogenic cause for the presentation unlikely. cussion with the microbiology team, she was discharged A decision was made to continue medical management. with a 5-­week course of oral co-­amoxiclav and weaning Intravenous gentamicin was stopped on Day 3 following steroid regime. Upon outpatient review in clinic 6 weeks advice from the microbiology team. By the morning of later, the patient had made a complete recovery, with Day 4, she was managing small amounts of oral intake and normal appearances of the floor-­of-­mouth, oropharynx, remained afebrile despite the neck swelling and persistent tongue base and vocal cords on flexible nasoendoscopy. 3 | DISC USSION The peritonsillar space is a potential space between the tonsillar capsule medially and superior pharyngeal con- strictor muscle laterally. Due to the presence of loose con- nective tissue, it is highly susceptible to developing an inflammatory reaction following infection.6 Peritonsillar cellulitis or phlegmon is considered a transition phase of the inflammatory process before abscess formation.7 Adult DNSI may involve multiple spaces leading to se- vere complications, and it is sometimes difficult to trace the primary source of the infection.8 From the periton- sillar space, infections may extend into the para-­and/or retropharyngeal spaces.3 Klug et al.9 reviewed the clinical records of 63 patients with parapharyngeal abscess in a Danish tertiary care center over a 11-­year period and re- F I G U R E 1 Contrast-­enhanced computed tomography ported that 52% of patients had concomitant peritonsillar (CT) imaging demonstrating a soft tissue abnormality within abscess. Rarely, from the parapharyngeal space, infections the left parapharyngeal space adjacent to the oropharynx but of tonsillar origin may spread into adjacent deep neck without convincing evidence of enhancement, suggestive of an spaces such as the submandibular, parotid, and mastica- inflammatory phlegmon. tor spaces.10–­13  | 20500904, 2022, 9, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.6325 by Nat Prov Indonesia, Wiley Online Library on [01/07/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License TAILOR et al.    3 of 5 F I G U R E 2 Magnetic resonance imaging (MRI) demonstrating an inflammatory mass lesion and multiloculated collection centered on the left palatine tonsil, which extends to the left lateral and anterior floor-­of-­mouth. A recent narrative systematic review published in 2020 guidelines based on local epidemiology. The rationale for sought to identify the spectrum of complications in pa- multiple broad-­spectrum antimicrobial therapy in DNSI tients with peritonsillar abscess and identified only one is to ensure adequate cover for all likely pathogens, both case of bilateral submandibular cellulitis (and concurrent aerobic and anaerobic bacteria originating from the pha- parapharyngeal abscess) among 334 published cases of ryngeal flora.14 Our hospital protocol recommended in- peritonsillar abscess with associated complications in the travenous amoxicillin, metronidazole, and gentamicin, literature.3,12 The 75-­year-­old gentleman refused hot ton- although various antibiotic regimes exist depending on sillectomy and subsequently developed submandibular geographical region. Gram-­positive cocci, such as strepto- space abscess, necessitating abscess drainage via a cervi- cocci and staphylococci, are the most common bacterial cal approach.12 Matsuura describes a case of a 68-­year-­old species isolated.14,15 gentleman with ulcerative colitis who presented with However, despite aggressive medical management peritonsillar abscess complicated by Ludwig's angina. The with intravenous antibiotics and steroids, the patient patient initially refused needle aspiration and developed subsequently developed a left parapharyngeal and floor-­ submandibular erythema with swelling 5 days later, re- of-­mouth abscess. It is possible that a small abscess was quiring submandibular incision and drainage as well as a present during the early phases of the patient's illness, but 6-­week course of intravenous and oral antibiotics.13 unfortunately too subtle to detect on CT. The prolonged In the present case, because initial CT imaging of the use of antibiotics without surgical drainage could have neck demonstrated a non-­enhancing soft tissue abnormal- created an antibioma (i.e., sterile abscess) due to local- ity within the parapharyngeal space and attempted nee- ization of pus, often reported in odontogenic infections.16 dle aspiration of the peritonsillar space did not yield pus, This might explain why the patient did not obtain signifi- a diagnosis of tonsillar phlegmon was made. Antibiotic cant relief of symptoms by Day 4 of admission. Although therapy was empirically initiated following microbiology Streptococcus anginosus was subsequently isolated from  | 20500904, 2022, 9, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.6325 by Nat Prov Indonesia, Wiley Online Library on [01/07/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 4 of 5    TAILOR et al. the swab taken intraoperatively, this may have represented ACKNOWLEDGEMENTS contamination from oral commensal bacteria. None. Unusually, the patient did not present with risk factors typically associated with spread of DNSI, such as older CONFLICT OF INTEREST age, diabetes mellitus, immunosuppression, or under- The authors declare that they have no competing or con- lying systemic disease.17,18 There is no standard surgical flict of interests. protocol for managing peritonsillitis with involvement of adjacent deep neck spaces, although hot (or abscess) DATA AVAILABILITY STATEMENT tonsillectomy and intraoral incision and drainage, as per- The data that support the findings of this study are avail- formed in this case, is recommended to reduce risk of fur- able from the corresponding author upon reasonable ther abscess extension.9,12 request. Although there are frequent reports of peritonsillar ab- scess complicated by parapharyngeal extension, the pres- CONSENT ent case is unique in that initial imaging demonstrated a Written informed consent was obtained from the patient tonsillar phlegmon with some mass effect but no convinc- for the publication of this case report and any accompany- ing evidence of collection. This tonsillar phlegmon spread ing images. along the peritonsillar/parapharyngeal plane to cause a diffuse cellulitis and collection in the floor-­of-­mouth de- ORCID spite intravenous antibiotic therapy. Although Ludwig's Bhavesh V. Tailor https://orcid. angina is usually odontogenic in origin, it is important to org/0000-0001-7534-5183 highlight that other etiologies are possible, although less Haran Devakumar https://orcid. common. Throughout the course of admission, there was org/0000-0002-7918-9189 no evidence of airway compromise. 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