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Fatal necrotizing fasciitis following a non-treated mandibular fracture: case report

Fasciíte necrotizante fatal devido a fratura mandibular não tratada: relato de caso

ABSTRACT

Necrotizing fasciitis is a rare and severe infection characterized by extensive and quickly progressing necrosis of the subcutaneous tissue and muscle fascia associated with high mortality rates in the head and neck region. We present a case of fatal necrotizing fasciitis due to an untreated mandibular fracture. Eight days after the trauma, the patient was admitted to the hospital and died on the sixth day of hospitalization.

Indexing terms
Case report; Mandibular fractures; Necrotizing fasciitis

RESUMO

Fasciite necrosante é uma infecção rara e grave caracterizada por necrose extensa e de rápida progressão do tecido subcutâneo e fáscia muscular associada a altas taxas de mortalidade na região da cabeça e pescoço. Apresentamos um caso de fasciite necrosante fatal devido a uma fratura mandibular não tratada. Oito dias após o trauma, o paciente deu entrada no hospital e faleceu no sexto dia de internação.

Termos de indexação
Relato de caso; Fraturas mandibulares; Fasciíte necrotizante

INTRODUCTION

The necrotizing fasciitis (NF) is a rare, severe, devastating, and fulminating infection, characterized by necrosis of the fascia and adjacent soft tissue with quick progression and rapid expansion along the fascial planes [11 Chou PY, Hsieh YH, Lin CH. Necrotizing fasciitis of the entire head and neck: literature review and case report. Biomed J. 2020;43(1):94-98. http://dx.doi.org/10.1016/j.bj.2019.08.002
https://doi.org/10.1016/j.bj.2019.08.002...
].

When the NF occur in the area below the border of the mandible, above the clavicle, and anterior to the trapezius, it is called cervical necrotizing fasciitis (CNF) [22 Gunaratne DA, Tseros EA, Hasan Z, Kudpaje AS, Suruliraj A, Smith MC, et al. Cervical necrotizing fasciitis: systematic review and analysis of 1235 reported cases from the literature. Head Neck. 2018;40(9):2094-2102. http://dx.doi.org/10.1002/hed.25184
https://doi.org/10.1002/hed.25184...
].

There are few reported cases of CNF developing after mandibular fracture [33 Badri AA, Hasheminasab MS, Bolandparva F. Cervical necrotizing fasciitis after surgery of a mandibular fracture. J Craniofac Surg. 2020;31(6):e541-e542. http://dx.doi.org/10.1097/SCS.0000000000006471
https://doi.org/10.1097/SCS.000000000000...
, 44 Chin RS, Kaltman SI, Colella J. Fatal necrotizing fasciitis following a mandibular fracture. J Craniomaxillofac Trauma. 1995;1(3):22-29.], causing this report crucial for professionals who treat maxillofacial trauma.

We present a case where the patient postponed the search for trauma assessment, resulting in fatal CNF due to an untreated mandibular fracture. This case report was prepared following the CARE Guidelines [55 Riley DS, Barber MS, Kienle GS, Aronson JK, von Schoen-Angerer T, Tugwell P, et al. CARE guidelines for case reports: explanation and elaboration document. J Clin Epidemiol. 2017;89:218-235. http://dx.doi.org/10.1016/j.jclinepi.2017.04.026
https://doi.org/10.1016/j.jclinepi.2017....
].

CASE REPORT

A 26-year-old black man was admitted to the emergency hospital with pain in the left hemiface, cervical and thoracic region. He reported a previous untreated mandibular fracture nine days ago. The patient was eupneic, flushed, conscious, oriented, afebrile, and had isochoric and photo reagent pupils. He denied deleterious habits and vices, other comorbidities, and drug allergies.

The facial examination exhibited bilateral submandibular hematoma, sialorrhea, halitosis, poor oral hygiene, trismus, dental malocclusion with mobility, and crackling in the left mandibular body. The Computed Tomography showed complete fracture with subcutaneous emphysema in the adjacent soft tissues.

The laboratory tests showed leukocytosis (11.5 mm3). After seven hours of hospitalization, the patient worsened with tachypnea and respiratory failure, with a saturation of 78%. Thus, the patient was referred to the intensive care unit, where orotracheal intubation was performed.

The next day, the patient was taken to the operating room for cervicectomy, debridement, and drainage of the cervical abscess. During the surgical procedure, the diagnosis of CNF was made due to extensive areas of necrosis of the subplatismatic muscle fascia and deep cervical planes (figure 1). Thus, debridement and necrosectomy of several muscular planes and deep fascia were performed.

Figure 1
A) Edema observed in the cervical region (black arrows). B) Cervical incision made for access (red arrows). C and D ) Exposure and a wide range of necrotic tissue (white arrows).

Antibiotic therapy was made with gentamicin (80mg 8/8 hours) associated with the meropenem (1g 8/8 hours) and vancomycin (500mg 12/12 hours). Laboratory tests showed immature neutrophils at 21% and urea at 48mg/dL.

On the first postoperative day, the patient had sepsis. He was intubated, on mechanical ventilation, hemodynamically unstable, and receiving a noradrenaline vasoactive drug (80 ml/h).

On the second postoperative day, the laboratory tests demonstrated the values: leukocytes (10.5 mm3), immature neutrophils (13%), urea (109 mg / dL), creatinine (2.47 mg / mL) and CRP (448.45 mg / L). In the cervical region, a foul odor and the presence of a necrotic area were noted. The previous antibiotic therapy was suspended and replaced with the prescription of oxacillin (2 g 8/8 h), metronidazole (500 mg 8/8 h), and cefepime (2 g 8/8 h).

On the third postoperative day, the patient had central and peripheral cyanosis with slow capillary filling time. He showed closed extremities and received noradrenaline and adrenaline. The cervical area, with muscles exposed, showed improvement without pus, necrosis, bleeding, and granulation tissue. The team suspended this antibiotic therapy and returned to the initial treatment with an increase in the intravenous dosage of gentamicin (500mg 8/8 h), meropenem (2g 8/8 h), and vancomycin (2g 8/8 h).

On the fourth day, the patient suffered a reversed cardiorespiratory arrest during the night, followed by tachycardia, fever, slowed peripheral perfusion, paleness, and a slightly distended abdomen. He remained in continuous moderate sedation, intubated on mechanical ventilation, and without bowel movements throughout the day. During the night, a new cardiorespiratory arrest occurred, leading to death (figure 2).

Figure 2
Timeline.

The result of the microbiological culture exam was performed from the collection on the second day of hospitalization. The result showed the presence of Staphyloccocus hominis Subsp. hominis. This strain was having sensitivity to daptomycin, linezolid, rifampicin, synercid, tetracillin, and vancomycin. However, the test result was only available to the team after five days of collection.

DISCUSSION

This rare case showed the importance of early diagnoses and treatment for CNF. The patient’s delay in seeking treatment for a sporting accident contributed to the negative outcome. Besides, diagnosing the early stages of CNF concerning other soft tissue infections of odontogenic origin is difficult and often leads to less aggressive treatment, resulting in increased morbidity and mortality [66 Al-Ali MA, Hefny AF, Idris KM, Abu-Zidan FM. Cervical necrotizing fasciitis: an overlooked diagnosis of a fatal disease. Acta Otolaryngol. 2018;138(4):411-414. http://dx.doi.org/10.1080/00016489.2017.1393841
https://doi.org/10.1080/00016489.2017.13...
]. With disease progression and nutrient and vascularization loss, the skin becomes dark, and necrosis sites become apparent. In our case, the initial edema was compatible with mandibular fracture associated with cellulitis without any dark coloration that could evidence FN diagnosis.

Upon admission, the patient was afebrile, which corroborates that 60% may not have a fever [77 Goh T, Goh LG, Ang CH, Wong CH. Early diagnosis of necrotizing fasciitis. Br J Surg. 2014;101(1):e119-125. http://dx.doi.org/10.1002/bjs.9371
https://doi.org/10.1002/bjs.9371...
]. Other symptoms, such as erythema, pain, and edema, were presented in the case and related to the diagnosis. Also, bullae, crepitus, skin necrosis, hypotension are signs that may be present [77 Goh T, Goh LG, Ang CH, Wong CH. Early diagnosis of necrotizing fasciitis. Br J Surg. 2014;101(1):e119-125. http://dx.doi.org/10.1002/bjs.9371
https://doi.org/10.1002/bjs.9371...
], but in the initial evaluation, they had not manifested, making it more challenging to diagnose the entity.

The laboratory risk indicator for necrotizing fasciitis (LRINEC) score can be valuable for early diagnosis [88 Ogawa M, Yokoo S, Takayama Y, Kurihara J, Makiguchi T, Shimizu T. Laboratory risk indicator for necrotizing fasciitis of the oro-cervical region (LRINEC-OC): a possible diagnostic tool for emergencies of the oro-cervical region. Emerg Med Int. 2019;2019:1573453. http://dx.doi.org/10.1155/2019/1573453
https://doi.org/10.1155/2019/1573453...
]. The tool is based on six standard serum parameters: C-reactive protein (CRP), total white cell count, hemoglobin, serum sodium, creatinine, and glucose. Despite its controversial use in literature, a systematic review [99 Bechar J, Sepehripour S, Hardwicke J, Filobbos G. Laboratory risk indicator for necrotising fasciitis (LRINEC) score for the assessment of early necrotising fasciitis: a systematic review of the literature. Ann R Coll Surg Engl. 2017;99(5):341-346. http://dx.doi.org/10.1308/rcsann.2017.0053
https://doi.org/10.1308/rcsann.2017.0053...
] demonstrated that the LRINEC score is a helpful adjunct in the clinical diagnosis of necrotizing fasciitis with a statistically positive correlation. In addition, a modification in the score was proposed with the addition of clinical parameters such as pain, fever, tachycardia, and comorbidities.

The CNF is extremely rare due to the robust blood supply of the cervical area. A large percentage (97.1%) of patients have a past medical history. The association with diabetes mellitus (18.18%), alcoholism (9,09%), underlying malignancy (3%), corticosteroid therapy (1.5%), and HIV (1%) was found in a systematic review and analysis of 1235 reported cases from the literature [22 Gunaratne DA, Tseros EA, Hasan Z, Kudpaje AS, Suruliraj A, Smith MC, et al. Cervical necrotizing fasciitis: systematic review and analysis of 1235 reported cases from the literature. Head Neck. 2018;40(9):2094-2102. http://dx.doi.org/10.1002/hed.25184
https://doi.org/10.1002/hed.25184...
]. Although CNF is more common in obese individuals and the immunocompromised, they can affect healthy individuals without risk factors [1010 Sideris G, Nikolopoulos T, Delides A. Cervical necrotizing fasciitis affects only immunocompromized patients? Diagnostic challenges, treatment outcomes and clinical management of eleven immunocompetent adult patients with a still fatal disease. Am J Otolaryngol. 2020;41(6):102613. http://dx.doi.org/10.1016/j.amjoto.2020.102613
https://doi.org/10.1016/j.amjoto.2020.10...
,1111 Elander J, Nekludov M, Larsson A, Nordlander B, Eksborg S, Hydman J. Cervical necrotizing fasciitis: descriptive, retrospective analysis of 59 cases treated at a single center. Eur Arch Otorhinolaryngol. 2016;273(12):4461-4467. http://dx.doi.org/10.1007/s00405-016-4126-y
https://doi.org/10.1007/s00405-016-4126-...
].

CNF can be caused by odontogenic infection (47%), pharyngolaryngeal (28%), tonsillar (6%), traumatic, iatrogenic, or post-operative (4%) salivary gland (2%), skin (1%) between others [22 Gunaratne DA, Tseros EA, Hasan Z, Kudpaje AS, Suruliraj A, Smith MC, et al. Cervical necrotizing fasciitis: systematic review and analysis of 1235 reported cases from the literature. Head Neck. 2018;40(9):2094-2102. http://dx.doi.org/10.1002/hed.25184
https://doi.org/10.1002/hed.25184...
]. There are few cases reported in the literature whose cause is a mandibular fracture [33 Badri AA, Hasheminasab MS, Bolandparva F. Cervical necrotizing fasciitis after surgery of a mandibular fracture. J Craniofac Surg. 2020;31(6):e541-e542. http://dx.doi.org/10.1097/SCS.0000000000006471
https://doi.org/10.1097/SCS.000000000000...
,44 Chin RS, Kaltman SI, Colella J. Fatal necrotizing fasciitis following a mandibular fracture. J Craniomaxillofac Trauma. 1995;1(3):22-29.]. Of these, Chin et al. (1995)[44 Chin RS, Kaltman SI, Colella J. Fatal necrotizing fasciitis following a mandibular fracture. J Craniomaxillofac Trauma. 1995;1(3):22-29.] had a fatal outcome, and however, unlike our case, the patient was severely immunocompromised elderly. Nevertheless, the treatment was also delayed.

Among the microorganisms identified in the CNF, staphylococcus was found in second place, corresponding to 18.09% of patients. The most common was streptococcus in 61.22% of cases [22 Gunaratne DA, Tseros EA, Hasan Z, Kudpaje AS, Suruliraj A, Smith MC, et al. Cervical necrotizing fasciitis: systematic review and analysis of 1235 reported cases from the literature. Head Neck. 2018;40(9):2094-2102. http://dx.doi.org/10.1002/hed.25184
https://doi.org/10.1002/hed.25184...
]. In this case report, staphylococcus hominis was isolated.

The overall mortality is considered high, ranging from 13.36% [1212 Wang YS, Wong CH, Tay YK. Staging of necrotizing fasciitis based on the evolving cutaneous features. Int J Dermatol. 2007;46(10):1036-1041. http://dx.doi.org/10.1111/j.1365-4632.2007.03201.x
https://doi.org/10.1111/j.1365-4632.2007...
] to 18% [1010 Sideris G, Nikolopoulos T, Delides A. Cervical necrotizing fasciitis affects only immunocompromized patients? Diagnostic challenges, treatment outcomes and clinical management of eleven immunocompetent adult patients with a still fatal disease. Am J Otolaryngol. 2020;41(6):102613. http://dx.doi.org/10.1016/j.amjoto.2020.102613
https://doi.org/10.1016/j.amjoto.2020.10...
]. Wang et al [1212 Wang YS, Wong CH, Tay YK. Staging of necrotizing fasciitis based on the evolving cutaneous features. Int J Dermatol. 2007;46(10):1036-1041. http://dx.doi.org/10.1111/j.1365-4632.2007.03201.x
https://doi.org/10.1111/j.1365-4632.2007...
] proposed a three-stage classification of FN, with the signs and symptoms to the initial stage corresponds to only cutaneous symptoms (tenderness, erythema, and swelling). In the intermediate stage, the patient presents blister or bullae formation (serous fluid), and the last stage (late) presents crackling, skin anesthesia, and necrosis with dusky discoloration [1212 Wang YS, Wong CH, Tay YK. Staging of necrotizing fasciitis based on the evolving cutaneous features. Int J Dermatol. 2007;46(10):1036-1041. http://dx.doi.org/10.1111/j.1365-4632.2007.03201.x
https://doi.org/10.1111/j.1365-4632.2007...
]. The evolution to the third and last stage (sepsis) was rapid.

CONCLUSION

The mandibular fracture can be an etiology of CNF who has a high mortality. The definitive diagnosis must be rapid and the treatment aggressive. The patient’s delay in seeking treatment contributed to the fatal negative outcome.

How to cite this article

REFERENCES

  • 1
    Chou PY, Hsieh YH, Lin CH. Necrotizing fasciitis of the entire head and neck: literature review and case report. Biomed J. 2020;43(1):94-98. http://dx.doi.org/10.1016/j.bj.2019.08.002
    » https://doi.org/10.1016/j.bj.2019.08.002
  • 2
    Gunaratne DA, Tseros EA, Hasan Z, Kudpaje AS, Suruliraj A, Smith MC, et al. Cervical necrotizing fasciitis: systematic review and analysis of 1235 reported cases from the literature. Head Neck. 2018;40(9):2094-2102. http://dx.doi.org/10.1002/hed.25184
    » https://doi.org/10.1002/hed.25184
  • 3
    Badri AA, Hasheminasab MS, Bolandparva F. Cervical necrotizing fasciitis after surgery of a mandibular fracture. J Craniofac Surg. 2020;31(6):e541-e542. http://dx.doi.org/10.1097/SCS.0000000000006471
    » https://doi.org/10.1097/SCS.0000000000006471
  • 4
    Chin RS, Kaltman SI, Colella J. Fatal necrotizing fasciitis following a mandibular fracture. J Craniomaxillofac Trauma. 1995;1(3):22-29.
  • 5
    Riley DS, Barber MS, Kienle GS, Aronson JK, von Schoen-Angerer T, Tugwell P, et al. CARE guidelines for case reports: explanation and elaboration document. J Clin Epidemiol. 2017;89:218-235. http://dx.doi.org/10.1016/j.jclinepi.2017.04.026
    » https://doi.org/10.1016/j.jclinepi.2017.04.026
  • 6
    Al-Ali MA, Hefny AF, Idris KM, Abu-Zidan FM. Cervical necrotizing fasciitis: an overlooked diagnosis of a fatal disease. Acta Otolaryngol. 2018;138(4):411-414. http://dx.doi.org/10.1080/00016489.2017.1393841
    » https://doi.org/10.1080/00016489.2017.1393841
  • 7
    Goh T, Goh LG, Ang CH, Wong CH. Early diagnosis of necrotizing fasciitis. Br J Surg. 2014;101(1):e119-125. http://dx.doi.org/10.1002/bjs.9371
    » https://doi.org/10.1002/bjs.9371
  • 8
    Ogawa M, Yokoo S, Takayama Y, Kurihara J, Makiguchi T, Shimizu T. Laboratory risk indicator for necrotizing fasciitis of the oro-cervical region (LRINEC-OC): a possible diagnostic tool for emergencies of the oro-cervical region. Emerg Med Int. 2019;2019:1573453. http://dx.doi.org/10.1155/2019/1573453
    » https://doi.org/10.1155/2019/1573453
  • 9
    Bechar J, Sepehripour S, Hardwicke J, Filobbos G. Laboratory risk indicator for necrotising fasciitis (LRINEC) score for the assessment of early necrotising fasciitis: a systematic review of the literature. Ann R Coll Surg Engl. 2017;99(5):341-346. http://dx.doi.org/10.1308/rcsann.2017.0053
    » https://doi.org/10.1308/rcsann.2017.0053
  • 10
    Sideris G, Nikolopoulos T, Delides A. Cervical necrotizing fasciitis affects only immunocompromized patients? Diagnostic challenges, treatment outcomes and clinical management of eleven immunocompetent adult patients with a still fatal disease. Am J Otolaryngol. 2020;41(6):102613. http://dx.doi.org/10.1016/j.amjoto.2020.102613
    » https://doi.org/10.1016/j.amjoto.2020.102613
  • 11
    Elander J, Nekludov M, Larsson A, Nordlander B, Eksborg S, Hydman J. Cervical necrotizing fasciitis: descriptive, retrospective analysis of 59 cases treated at a single center. Eur Arch Otorhinolaryngol. 2016;273(12):4461-4467. http://dx.doi.org/10.1007/s00405-016-4126-y
    » https://doi.org/10.1007/s00405-016-4126-y
  • 12
    Wang YS, Wong CH, Tay YK. Staging of necrotizing fasciitis based on the evolving cutaneous features. Int J Dermatol. 2007;46(10):1036-1041. http://dx.doi.org/10.1111/j.1365-4632.2007.03201.x
    » https://doi.org/10.1111/j.1365-4632.2007.03201.x

Edited by

Assistant editor: Marcelo Sperandio

Publication Dates

  • Publication in this collection
    21 Nov 2022
  • Date of issue
    2022

History

  • Received
    06 Sept 2021
  • Accepted
    16 Mar 2022
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