November 30, 2021
J Diagn Treat Oral Maxillofac Pathol 2021;125–134.
Under a Creative Commons license
Savchuk LA, Fesenko II. Obstructive submandibular sialadenitis complicated with severe neck phlegmon: transcutaneous removal of sialolith with a flow of pus: literature review of extraoral complications. J Diagn Treat Oral Maxillofac Pathol 2021;5(11):125–34.
Sialolithiasis, as the most common reason (60–85 percent) of obstructive salivary gland disease, in the rare cases, may be complicated by soft tissues abscess or/and fistula. The purpose of this report is to present a rare case of submandibular gland sialolithiasis complicated with severe neck phlegmon in a 47-year-old Caucasian male. Ultrasonography and transcutaneous removal of sialolith upon the purulent locus lancing are highlighted. Our literature review based on the existed ones with a total 24 complication cases is presented. Wakoh et al`s classification of submandibular gland sialolith-associated fistulas types is analyzed. The ultrasound imaging is still underestimated and not adequately popularized among head and neck and oral and maxillofacial surgeons. Presented case and published reports show the usefulness of this constantly developing diagnostic technique in a combination with knowledge of possible extraoral purulent complications’ and its management.
Sialolithiasis, as the most common reason (60–85 percent)1 of obstructive salivary gland disease, in the rare cases, may be complicated by soft tissues abscess2,3 or/and fistula.4,5 A clinical study by Kishore Kumar et al (2012) of the 200 cases of cutaneous sinuses revealed that only in 0.5 percent of cases was noted cutaneous fistula as a result of submandibular gland calculus.6 Such fistula usually is named by authors as orocutaneous or sialo-cutaneous fistula.5,7
According to our literature search, submandibular gland calculus-associated abscesses/phlegmons are even rarer comparing with sialo-cutaneous fistulae. Purulent floor of the mouth processes as a complication of obstructive salivary stone usually were diagnosed by multi-slice computed tomography (MSCT).3,8 Despite ultrasound (US) are widely described as a diagnostic tool for submandibular sialolithiasis1,9,10 its application in case of calculus-associated cervical fistulae and abscess/phlegmon to our knowledge is presented in very limited publications.11,12
The purpose of this report is to present a rare case of submandibular gland sialolithiasis complicated with severe neck phlegmon in a 47-year-old male. Ultrasound and transcutaneous removal of sialolith upon the purulent locus lancing are highlighted. Our own literature review based on the existed ones with a total 24 complication cases is presented.
A 47-year-old Caucasian gentleman was referred to the Kyiv Regional Clinical Hospital with a left neck and submandibular swelling (Fig 1), fever, difficulty of swallowing. The complaints had been initiated with salivary colics several weeks before. Previously, during the last years, patient noted a painful chewing–associated submandibular swelling at the left side.
US was performed by an experienced doctor of ultrasound diagnostics (L.A.S., her experience – 31 years). Submandibular and neck regions were examined bilaterally according to the protocol (ie, swelled and healthy side). Using linear transducer (12-5 MHz) in a longitudinal submandibular position, the gray scale US (also known as B-mode [brightness mode]) showed hyperechoic semilunar structure (ie, sialolith, measured 0.76 × 0.78 cm) with an artifact of acoustic shadowing behind (Fig 2). Also, in this left submandibular region, the submandibular gland with a hypoechoic parenchyma and the collection of hypoechoic heterogenic content (ie, pus) were visualized. The diagnosis of obstructive sialadenitis caused by sialolith and complicated with submandibular and left neck phlegmon was established.
The phlegmon lancing was done under the local infiltration anesthesia using 8.0 ml of 4 percent Ultracain® D-S solution (Sanofi-Aventis Deutschland GmbH, Frankfurt am Main, Germany) mixed with 12.0 ml of 0.9% saline solution in a 20-ml syringe. Intravenous sedation was done by anesthesiologist`s team. An oblique 3.5-cm incision was planned and done inside the prominent upper neck skin crease (Fig 1C). A 1.2-cm salivary calculus (ie, sialolith) was removed with a flow of pus (~22 ml) upon the finger examination (ie, revision) of the purulent wound (Fig 3). Tubular double drainage was installed after rinsing with an antiseptic solution. Post-operative period associated with infusion and antibacterial therapy and wound rinsing showed gradual decrease of soft tissues inflammatory changes and complaints.
Digestion-associated swelling9 (also known as “mealtime syndrome”13) in the submandibular region is a common disorder at the in- and out-patient oral and maxillofacial units. And typically, this is a manifestation of obstructive sialadenitis caused by sialoliths. Such complications of obstructive sialoliths as pus collection in the cellular spaces and fistulae are not common and its diagnostic features and management strategies are to be analyzed. In a literature review we analyze all purulent extraoral complications’ manifestations (abscesses, phlegmons, sialo-cutaneous fistulae, and tender cutaneous nodules).
Table 1 is presenting our literature review of submandibular sialolithiasis complications and its management strategies based on the reviews of Ha et al (2020),14 Wakoh et al (2021)7.
TABLE 1. Literature Review of Submandibular Sialolithiasis with Purulent Extraoral Complications’ Manifestations and Its Management.
Among all 24 published cases (Table 1) of submandibular sialolithiasis with extraoral complications in the majority of cases (n = 17) there were noted the cutaneous fistulous tracts (in 69.56 percent), collection of pus in cellular space – in 5 cases (in 21.74 percent), and tender cutaneous nodule – in 2 cases (in 8.7 percent).
Among 16 fistulous cases, in two cases both sialo-oral and sialocutaneous fistulae were noted from one calculus, and in one case – the fistula located in a tissues with a resolution of the submandibular abscess.
Among five abscess/phlegmon cases, the abscess with discharging fistula was noted in one case (Wakoh et al)7.
Bridwell et al presented the description and computed tomography of an obstructed submandibular sialolith with abscess mimicking Ludwig's angina.3
Henry Hoffman presented an adult patient with submandibular region abscess due to the submandibular gland sialolithiasis, obstruction disease, and acute infection.8 In the reported institution`s protocol, the step by step management strategy was perfectly highlighted with a purpose to be a helpful guidelines for other practitioners designed to bridge the gap between procedural concepts and their implementation.25
Among five calculus-associated abscesses presentations and descriptions in three cases (Cartwright and Hardingham, Hoffman, and Wakoh et al)2,8,7 a limited collection of pus was noted, and in two cases (Bridwell et al and our case)3 the purulent process was diffuse and localized in two or more anatomical regions.
Worth of attention is the classification proposed by the authors from Japan who classified all submandibular gland sialolith-associated fistulas into four types7:
Sialo-pharyngeal fistula (synonym: sialo-parapharyngeal fistula).27
Wakoh et al presented the case of cutaneous fistula related with ectopic submandibular gland sialolith.7 Ha et al (2020) exhibited a sialo-cutaneous fistula but with no evidence of submandibular gland sialolith. In their cases the diagnosis of submandibular sialadenitis was established and the conservative treatment helped to eliminate the symptoms.14
Cases with intraoral complications of submandibular sialolithiasis (like submandibular duct fistula, etc.) were excluded from this discussion and can be analyzed in further studies. Despite the fact that in the report of Chandak et al (2012) the case with diagnosis of acute submandibular sialadenitis complicated with abscess was established there was no radiological evidence of sialoliths in submandibular gland.28
In the reported cases, some authors combined multiple diagnostic tools, like Jayachandran et al (X-ray, MSCT, MRI),18 others used a single one, like Hoffman (MSCT)8. Among 24 complication cases presented in the Table 1, computed tomography (non-contrast/contrast enhanced) was used in 10 cases, OPG – in three cases, X-ray – in five cases, X-ray fistulography – in three cases, fluoroscopy – in one case, US – in four cases, MRI – in 1 case, sialoendoscopy – in one case, and X-ray sialography – in one case. Thus, despite the fact that in the majority of cases (n = 10) the teams had chosen CT, ultrasonography (n = 4) has an undiscovered potential among surgeons.
US became a vital diagnostic tool for pathology detection in different glands―thyroid,29 lymphatic (ie, lymph nodes),30 and salivary ones31. Katz et al are more than right pointing out that a clinical US is to be indicated as soon as the first symptoms of sialadenitis occur.32 US is more than useful in the detection of structural changes of the salivary gland parenchyma, vascularization, condition of the duct system, sialoliths9,10 and even mucous plugs31. The authors note that approximately 20 to 40% of the salivary stones are not opaque on plain radiography, but most of these sialoliths are visible upon sialography.32 2.0-mm and longer sialoliths can be detected on gray scale US.32
Practitioners must keep in mind the possible calcifications in the area of major salivary glands as potential mimickers of sialoliths. These can be: healed tuberculous lymphadenitis, phleboliths, tonsilloliths, segmental ossification of the stylohyoid ligament.33 So, analysis of the anamnesis data, complaints and radiological features can significantly help in differential diagnostics.
In different countries, the purulent process presented in our case can be described by different terms. In English-speaking countries the term abscess34 is usually used, and in some of East European countries (Ukraine, etc.) the term phlegmon can be used for the similar condition. In general, phlegmon is an acute, clearly not limited purulent inflammation of cellular tissue.35 And abscess is a cavity filled with pus and delimited from the surrounding tissues by a pyogenic membrane.35 In this particular case the term phlegmon was used to describe the spread of the purulent process up to two anatomical regions.
In general, based on our literature search, the transcutaneous removal of submandibular gland sialoliths was reported in 4 cases. And our case became the fifth one. More than rare case of cutaneous exfoliation of the submandibular gland sialolith was recorded Karengera et al (1998).21 Very unusual two cases of the submandibular salivary stones migration in the cutaneous direction was described by Drage et al (2005).13 In 2021, Wakoh et al also highlighted the submandibular gland calculus migration to subcutaneous tissue.7 Calculi removal in those three cases7,13 was similar with our case in which the gland was preserved.
Ultrasound imaging is still underestimated and not enough popularized among head and neck and oral and maxillofacial surgeons. Presented case and published reports show the usefulness of this constantly developing diagnostic technique in a combination with knowledge of possible extraoral purulent complications’ and its management.
The patient provided written consent for the use of his images.
Conceptualization: Fesenko II. Data acquisition: Savchuk LA. Data analysis, interpretation, and drafting of the manuscript: Fesenko II. Critical revision of the manuscript: Savchuk LA, Fesenko II. Approval of the final version of the manuscript: both authors.
We would like to thank Professor Henry T. Hoffman, MD, Iowa City, Iowa, United States for the kind support in preparing the manuscript.
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