As a rare complication of oropharyngeal and cervical infection, descending necrotizing mediastinitis (DNM) is one of the most dreaded forms of mediastinitis, with reported mortality ranging from 16.5% to 50% (1-3). Successful treatment of DNM relies on early diagnosis and aggressive surgical drainage of the mediastinal abscess (2,4). Up to now, very few childhood cases of DNM have been reported.
Video-assisted thoracoscopic surgery (VATS) in children was first reported as an initial diagnostic procedure in the 1970s (5). In recent decades, VATS has become more and more popular for a wide range of applications, from biopsy to advanced thoracic surgery, due to technological advancements in visualization and instrumentation. The advantages of VATS included less wound infection rate, lower postoperative pain, shorter hospital stay, and fewer long-term musculoskeletal sequelae (6). Using VATS, we report herein a successful treatment of a 6-year-old girl with DNM complicated with severe thoracic empyema. We present the following case in accordance with the CARE reporting checklist (available at https://tp.amegroups.com/article/view/10.21037/tp-22-60/rc).
A 6-year-old previously healthy girl presented with odynophagia and dental pain of the right upper molars. One day later, she was admitted to the local general hospital with high fever of 40 ℃. Although broad-spectrum antibiotics were administered, the fever persisted. On the fourth day in the local hospital, dental pain gradually resolved but tachypnea and left chest pain developed. An urgent chest computed tomography (CT) revealed pneumonia, pleural effusion, mediastinal widening, and air bubbles accumulated in the neck (Figure 1). She was transferred immediately to our hospital for intensive care. On admission, her vital signs were temperature 38.6 ℃, pulse rate 138 beats/min, respiratory rate 40 breaths/min, blood pressure 114/69 mmHg, and pulse oximetry of 95% on room air. There was no redness, localized swelling, or enlarged lymph nodes on her neck. Initial laboratory findings were shown in Table 1. Empirical treatment was started with meropenem (60 mg/kg per day) and linezolid (30 mg/kg per day). However, her tachypnea and dyspnea progressed rapidly. An ultrasound-guided left-sided thoracentesis was performed and drained 80 mL of brown sticky pus on the second day in our hospital. The laboratory examinations of drainage revealed low glucose (0.09 mmol/L), high protein levels (41.6 g/L), and neutrophil predominance (90%). Pus culture for only aerobes yielded Streptococcus constellatus. The blood culture was negative. On the third day, a contrast-enhanced CT scan of the neck and chest was performed. Compared with the previous result of the CT scan in her local hospital, the area of mediastinal abscess enlarged clearly, and thoracic empyema was more severe (Figure 2A-2C, Video 1). Air and fluid extended to the upper mediastinum rapidly and accumulated in this region. However, the low-density area of the abscess surrounding the right common carotid artery in the neck diminished (Figure 2D). The patient was therefore diagnosed with DNM complicated with severe empyema at the left side. VATS for left thoracic and mediastinal drainage was performed under general endotracheal anesthesia with two-lung ventilation (cuffed endotracheal tube size: 5 mm) on the next day. With the patient in the right lateral decubitus position, three 5-mm thoracic ports were introduced in the 4th, 7th, and 6th intercostal spaces, in the anterior, middle, and posterior axillary lines, respectively. Intrapleural carbon dioxide insufflation at low pressure of 2–4 mmHg was required for collapse of the ipsilateral lung. To obtain optimal visualization of the whole pleural cavity, each of the three ports could be used as the observation hole, and the other two ports were used as the operation holes. The procedure involved surgical debridement of the mediastinum and pleura with complete excision and decortication of necrotic tissue (Figure 3). Through the incisions of introduced ports, two drains (22F single-lumen chest tubes) were positioned in the left thoracic cavity and the opened mediastinum via the thoracic cavity, respectively. Drainage cultures were negative. Cervicotomy and transcervical drainage of the mediastinum was not performed due to the absence of swelling on the neck during the physical examination and diminishment of cervical abscess in the CT image. Her fever resolved within three days after surgery. As the fluid from the drainage tubes gradually decreased, both drains were removed after confirming by chest X-ray that residual fluid was less noticeable on hospital day 10 (Figure 4A). On the same day, the C-reactive protein (CRP) level decreased to 7.9 mg/L (Table 1), meropenem was changed to ceftazidime at 100 mg/kg per day for the purpose of step-down antibiotic therapy, and the patient was transferred to the department of pediatric infectious diseases from the pediatric intensive care unit in our hospital. She made a full recovery and was discharged without any complications on hospital day 18 after near-normal mediastinal contours on chest radiography was confirmed (Figure 4B).
|Laboratory tests||Reference range||Admission||D6||D10|
|White blood cell count (109/L)||3.5–9.5||4.85||17.36||8.66|
|C-reactive protein (mg/L)||0–8||306.5||95.8||7.9|
|Lactate dehydrogenase (U/L)||120–250||433||–||–|
D6, hospital day 6 (day 2 after surgery); D10, hospital day 10 (day 6 after surgery).
All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Declaration of Helsinki (as revised in 2013). Written informed consent was obtained from the patient’s parent or legal guardian for publication of this case report and accompanying images. A copy or the written informed consent is available for review by the editorial office of this journal.
DNM is a rare but fatal complication of odontogenic or pharyngeal infection, such as Ludwig’s angina, which is an infection of the submandibular space, usually secondary to infection of the second or third lower molar (7). These infections spread along the deep fascial spaces down to the mediastinum via the loose anatomical structures, such as the pretracheal space, perivascular space, and prevertebral space (3). In 1983, Estrera et al. first described DNM, which was found during surgery or post-mortem examinations (1). As DNM is rapidly progressive and highly lethal, prompt diagnosis, broad-spectrum antibiotic therapy, aggressive surgical drainage, as well as intensive medical care are crucial for successful treatment and recovery (8).
The most common symptoms of DNM include fever, odynophagia, cervical swelling, cervical pain, and dyspnea (9). Thoracic or back pain is less frequent. Only based on clinical symptoms and physical examination, DNM cannot be diagnosed early since these symptoms are not specific. Endo et al. reported an adult DNM case, in which a physical examination of the neck showed no abnormalities but a suspected retropharyngeal abscess immediately spread to the mediastinum instead of the cervical area via the prevertebral space (10). Similar to the case above, our patient also had a near-normal appearance of the neck on physical examination. It is therefore crucial to perform a CT scan immediately when DNM is suspected. Moreover, the cervicothoracic CT scan is the most effective tool for diagnosing DNM, assessing the range of mediastinitis, and guiding the surgical procedures (9).
According to previous studies, the most frequently isolated pathogen from pus cultures in children was Streptococcus aureus (11-13). In the present case, pus culture yielded Streptococcus constellatus. Mixed polymicrobial aerobic and anaerobic infections were reported in over 50% of the adult patients with DNM, which indicated broad-spectrum antibiotic therapy is crucial (9,14). Common choices for empiric therapy in adult patients were cephalosporins combined with metronidazole (9,15). In childhood cases, empirical treatment was started with vancomycin or meropenem more commonly (13,16,17).
It is universally accepted that antibiotic therapy alone is insufficient for eradicating the DNM (9). Aggressive surgical debridement and drainage of the mediastinum are fundamental. In adults, the optimal mediastinal drainage can be achieved with a variety of surgical approaches, such as a transcervical approach and transthoracic approaches including a posterolateral thoracotomy, median sternotomy, subxiphoid approach, and video-assisted thoracoscopic approach, but the best surgical approach remains controversial (4,8,18). As a minimally invasive approach to drain the mediastinum, VATS drainage in adults was first described in 1997 (18), and has been reported to be feasible and effective for treating the whole mediastinum (19). Compared with thoracotomy, VATS has fewer wound infection rates, shorter hospital stays, and fewer long-term musculoskeletal complications (6). However, the role of VATS in children with DNM has not been clear since only one case of management of DNM by VATS has been reported to our knowledge (16). Similar to the case above, our patient also developed severe empyema rapidly due to the spreading of pus from the mediastinum to the pleural cavity (Figure 2B,2C). Thanks to VATS, we could perform not only an aggressive surgical debridement of the mediastinum and pleura with complete excision and decortication of necrotic tissue, but also thorough mediastinal drainage by the incisions of introduced ports. However, we decided to avoid performing the cervicotomy or transcervical drainage of the mediastinum because our patient is unique in that there was no swelling on the neck and decreasing size of cervical abscess on CT. As a kind of traditional open surgery, transverse cervicotomy requires the use of a large incision and causes a visible scar in the neck unavoidably (20). Although cervicotomy is recommended as the standard care in adult DNM by most authors (9,14,15), we decided to choose an individual strategy according to clinical features of our young patient and achieved a satisfactory outcome. It should be emphasized that we would not hesitate to perform transcervical drainage if the patient had displayed an incomplete clinical resolution of the neck infection.
In conclusion, this case report suggests that early diagnosis by cervicothoracic CT and multidisciplinary approaches including intensive care, broad-spectrum antibiotics, and aggressive surgical intervention are crucial to reducing morbidity and mortality of DNM. VATS is a minimally invasive and appropriate treatment strategy for children with DNM, especially complicated with thoracic empyema.
We would like to thank the patient and her parents for their support and cooperation in publishing this work.
Funding: This work was supported by Jinan Science and Technology Bureau (grant No. 202019134), and the Shandong Provincial Natural Science Foundation (grant No. ZR2020QH263).
Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://tp.amegroups.com/article/view/10.21037/tp-22-60/rc
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Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tp.amegroups.com/article/view/10.21037/tp-22-60/coif). The authors have no conflicts of interest to declare.
Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Declaration of Helsinki (as revised in 2013). Written informed consent was obtained from the patient’s parent or legal guardian for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.
Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.
- Estrera AS, Landay MJ, Grisham JM, et al. Descending necrotizing mediastinitis. Surg Gynecol Obstet 1983;157:545-52. [PubMed]
- Marty-Ané CH, Berthet JP, Alric P, et al. Management of descending necrotizing mediastinitis: an aggressive treatment for an aggressive disease. Ann Thorac Surg 1999;68:212-7. [Crossref] [PubMed]
- Freeman RK, Vallières E, Verrier ED, et al. Descending necrotizing mediastinitis: An analysis of the effects of serial surgical debridement on patient mortality. J Thorac Cardiovasc Surg 2000;119:260-7. [Crossref] [PubMed]
- Papalia E, Rena O, Oliaro A, et al. Descending necrotizing mediastinitis: surgical management. Eur J Cardiothorac Surg 2001;20:739-42. [Crossref] [PubMed]
- Rodgers BM, Moazam F, Talbert JL. Thoracoscopy in children. Ann Surg 1979;189:176-80. [Crossref] [PubMed]
- Kiblawi R, Zoeller C, Zanini A, et al. Video-Assisted Thoracoscopic or Conventional Thoracic Surgery in Infants and Children: Current Evidence. Eur J Pediatr Surg 2021;31:54-64. [Crossref] [PubMed]
- Albacete Neto A, Coltro PS, Horácio GS, et al. Unilateral pectoralis major muscle flap for the treatment of sternal wounds due to Ludwig's angina. Int Wound J 2018;15:174-7. [Crossref] [PubMed]
- Mihos P, Potaris K, Gakidis I, et al. Management of descending necrotizing mediastinitis. J Oral Maxillofac Surg 2004;62:966-72. [Crossref] [PubMed]
- Ridder GJ, Maier W, Kinzer S, et al. Descending necrotizing mediastinitis: contemporary trends in etiology, diagnosis, management, and outcome. Ann Surg 2010;251:528-34. [Crossref] [PubMed]
- Endo S, Tsubochi H, Nakano T, et al. Descending necrotizing mediastinitis secondary to retropharyngeal abscess without cervical spread. Gen Thorac Cardiovasc Surg 2008;56:25-7. [Crossref] [PubMed]
- Wright CT, Stocks RM, Armstrong DL, et al. Pediatric mediastinitis as a complication of methicillin-resistant Staphylococcus aureus retropharyngeal abscess. Arch Otolaryngol Head Neck Surg 2008;134:408-13. [Crossref] [PubMed]
- Abdel-Haq N, Quezada M, Asmar BI. Retropharyngeal abscess in children: the rising incidence of methicillin-resistant Staphylococcus aureus. Pediatr Infect Dis J 2012;31:696-9. [Crossref] [PubMed]
- Wilson CD, Kennedy K, Wood JW, et al. Retrospective Review of Management and Outcomes of Pediatric Descending Mediastinitis. Otolaryngol Head Neck Surg 2016;155:155-9. [Crossref] [PubMed]
- Palma DM, Giuliano S, Cracchiolo AN, et al. Clinical features and outcome of patients with descending necrotizing mediastinitis: prospective analysis of 34 cases. Infection 2016;44:77-84. [Crossref] [PubMed]
- Wei D, Bi L, Zhu H, et al. Less invasive management of deep neck infection and descending necrotizing mediastinitis: A single-center retrospective study. Medicine (Baltimore) 2017;96:e6590. [Crossref] [PubMed]
- Chen IC, Hsu JH, Dai ZK. Management of descending necrotizing mediastinitis complicated with empyema by video-assisted thoracoscopic surgery. Pediatr Pulmonol 2016;51:E17-20. [Crossref] [PubMed]
- Macarrón CP, Palomino AP, Matute de Cárdenas JA, et al. Descending necrotizing mediastinitis in a child with chickenpox. J Thorac Cardiovasc Surg 2007;133:271-2. [Crossref] [PubMed]
- Roberts JR, Smythe WR, Weber RW, et al. Thoracoscopic management of descending necrotizing mediastinitis. Chest 1997;112:850-4. [Crossref] [PubMed]
- Hsin MK, Yim AP. Video assisted thoracoscopic surgery is a valuable approach for the management of descending necrotizing mediastinitis. Ann Surg 2011;253:1055-author reply 1055-6. [Crossref] [PubMed]
- Guillier D, Moris V, Al Hindi AA, et al. Surgical approaches in neck dissection: Comparing functional, oncologic and aesthetic aspects of transverse cervicotomy to Paul André's approach. Ann Chir Plast Esthet 2018;63:140-7. [Crossref] [PubMed]