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Tensiolito
CASE REPORT
Large tonsillolith associated with the accessory duct of the
ipsilateral submandibular gland: support for saliva
stasis hypothesis
1
I Pirkl, 1B Filipović, 2T Goranović and 1B Šimunjak
1
Department of Otorhinolaryngology—Head and Neck Surgery, University Hospital “Sveti Duh”, Zagreb, Croatia; 2Department
of Anesthesiology and Intensive Care Medicine, University Hospital “Sveti Duh”, Zagreb, Croatia
Tonsillolith is a calcified mass in the tonsil and/or its surrounding tissue, which is considered
to be caused by chronic tonsillitis. However, here we hypothesized that a tonsillolith can also
be formed by chronic saliva stasis in the tonsillar tissue, without any signs of chronic
inflammation. We present the case of a 32-year-old male patient with a large tonsillolith. We
reviewed his medical files, pre-operative imaging and intraoperative findings. During
a standard tonsillectomy, we encountered a large tonsillolith measuring 3.1 3 2.6 cm.
Additionally, a careful dissection of the lower pole of the remaining tonsillar tissue revealed
a large fistulous tract filled with saliva. Post-operative examination of the pre-operative CT
scan found a hypodense fistulous tract extending from the lower tonsillar pole towards the left
submandibular gland, measuring 36 mm in length, which was diagnosed as an accessory duct
of the submandibular gland. To our knowledge, this is the first case of a large tonsillolith
associated with the accessory duct of the ipsilateral major salivary gland. Furthermore, from
the aetiopathological view, this finding supports the saliva stasis hypothesis for formation of
the tonsillolith. However, larger studies, including a detailed radiological analysis as in our
case, are needed to further investigate this possible aetiology of tonsilloliths.
Dentomaxillofacial Radiology (2015) 44, 20150090. doi: 10.1259/dmfr.20150090
Cite this article as: Pirkl I, Filipović B, Goranović T, Šimunjak B. Large tonsillolith associated
with the accessory duct of the ipsilateral submandibular gland: support for saliva stasis hy-
pothesis. Dentomaxillofac Radiol 2015; 44: 20150090.
Introduction
Tonsilloliths or tonsil stones are singular or multiple centimetres in diameter.4 A histological study of excised
calculi found in tonsillar crypts that are located unilat- tonsils found microscopic size in 8% of specimens, while
erally or bilaterally. A review report determined the intermediate size (up to 7 mm) was found in 2%.4 Small
exact location of the tonsillolith, showing that in 69.7% tonsilloliths are usually asymptomatic and have small
of cases they are located in the tonsillar tissue, 21.2% in clinical importance. Large tonsilloliths are extremely
the tonsillar fossa and 9% in the soft palate.1 Tonsillo- rare, measuring several centimetres in diameter.3,5,6
lith prevalence in population ranging from 10 to 77 They result in clinical symptoms such as dysphagia,
years is up to 16%.2 Stones are composed of calcium otalgia, chronic halitosis and foreign body sensations.7,8
salts such as hydroxyapatite or calcium carbonate ap- Differential diagnosis of the large tonsillolith includes
atite, oxalates and other magnesium salts.3 They can phleboliths, lymph node calcifications, tuberculosis and
other chronic granulomatous diseases, elongated styloid
vary in size, measuring from microscopic to a few
process, prominent hamular process, foreign bodies,
Correspondence to: Dr Boris Filipović. E-mail: boris.filipovic@hotmail.com displaced tooth, peritonsillar abscess and neoplasm.1
Received 16 March 2015; revised 7 May 2015; accepted 19 May 2015 Today, a detailed clinical ear, nose and throat (ENT)
Tonsillolith and accessory duct of the salivary gland
2 of 4 I Pirkl et al
examination and a CT scan are sufficient for a proper tract is an accessory duct of the submandibular gland
diagnosis. Treatment is linked to the size and clinical and therefore inspected the sublingual region and pro-
symptoms and usually involves removal of the stone by bed the left orifice of Wharton’s duct. As they appeared
curettage or tonsillectomy under general anaesthesia.7 normal, we placed a suture around the duct at the lower
A large majority of published articles regarding tonsil- pole of the tonsil bed. We found no abnormal adhesions
loliths are focused on size of the particular stone, but between the tonsillar capsule and the superior con-
only a few discuss the pathogenesis of stone formation. strictor muscle. The rest of the surgery and post-
Most authors believe that chronic tonsillitis is a suffi- operative period were uneventful, and the patient
cient factor for localized salt precipitation and forma- recovered well. There was no post-operative swelling in
tion of a tonsillolith.3,5,7 However, some authors the left submandibular region. The stone was hard,
alternatively suggest that the tonsilloliths form as a re- yellowish-white, measuring 3.1 3 2.6 cm, the weight was
sult of saliva stasis in the efferent ducts of the small 6.99 g (Figure 3).
accessory salivary glands located in the oral cavity.9 Additionally, a detailed analysis of the pre-operative
Up to now, we have found only a few reports refer- CT scan showed a well-defined, hypodense fistulous
encing an accessory duct of the submandibular tract measuring 6 mm in width and 36 mm in length,
gland.10–13 This anomaly is defined as a duplication of extending from the lower tonsillar pole towards the left
the main Wharton’s duct and usually running parallel to submandibular gland (Figure 4). It was then diagnosed
it. Its opening can be at the orifice of the gland together as an accessory duct of the submandibular gland
with the main duct or separate in the floor of the oral extending to the tonsil associated with a large
cavity.13 tonsillolith.
A 32-year-old male patient was referred to our ENT Up to now, case reports with large giant tonsilloliths
department by his general practitioner under suspicion were mainly focused on the particular size of the stones,
of left peritonsillar abscess. His main symptoms were localization and clinical presentation.3,5,6 However,
slight dysphagia and foreign body sensation, which aetiopathogenesis of tonsilloliths is still unknown. Most
lasted for 1 year. He had no medical history of chronic authors believe tonsilloliths are formed in cases of
tonsillitis, peritonsillar abscess or any other diseases. chronic tonsillitis; as a result of organic debris, epithelial
ENT examination revealed an intraoral, submucosal, tissue and dead bacteria collections in tonsillar crypts
hard and well-delineated mass protruding from the soft which are a nidus for salt precipitation from the saliva
palate and tonsillar fossa of the left side, measuring secreted in the mouth by major and minor salivary
30 3 20 mm. Overlying mucosa was slightly erythema-
tous but with neither oedema nor displacement of the
uvula. There was no sign of trismus. The rest of the
head and neck examination findings were within normal
limits with no palpable neck lymphadenopathy. Labo-
ratory blood test parameters were normal. CT of the
head and neck showed a large hyperdense oval mass in
the left tonsillar fossa between the palatoglossus and
palatopharyngeus muscles, measuring approximately
30 3 20 mm (Figure 1). In order to exclude a possible
lithiasis in other regions of the body, we performed
ultrasounds of salivary glands, kidney and gallbladder,
and results were within normal values. The diagnosis of
a large left tonsillolith was made and, owing to the size
of the stone, we decided to perform a left tonsillectomy
under general anaesthesia. During surgery, upon ma-
nipulating to release the upper pole, we noticed a flow
of blocked saliva around the stone. Progressing from
the upper to the lower pole of the tonsillar bed, at the
level of lower pole, we opened a fistulous tract filled
with saliva. It was approximately 5 mm in width and 30
mm long (Figure 2), running from the lower pole of the
left tonsil, medial to the mandibular bone and towards
the left submandibular gland. The lumen of the fistula
was wide enough to accommodate the endoscope Figure 1 Coronal CT scan showing hyperdense mass (T) in the left
(Supplementary Video 1). We suspected that this fistula peritonsillar region. It measures approximately 30 3 20 mm (D1 3 D2).
Figure 4 (a) Axial CT scan showing the hypodense fistulous tract of the left side (white arrow), starting at the lower pole of the tonsil, measuring
6 mm in width (A–B). (b, c) Coronal and sagittal CT scans showing the calcified mass, tonsillolith (T) (dark arrow), at the level of the left
peritonsillar region with a hypodense fistulous tract (white arrows) extending from the lower tonsillar pole (A) towards the left submandibular
gland (B), measuring 36 mm in length (A–B).
inflammation. Our patient had an uneventful post- To our knowledge, this is the first report of a large
operative period and recovered fully. tonsillolith associated with the accessory duct of the
We found only a few reports presenting patients with ipsilateral major salivary gland. It is important to di-
a combination of pathology of salivary glands and agnose this condition prior to the surgery to be able to
tonsilloliths.14,15 However, a direct link between those adequately deal with the accessory duct. Furthermore,
two conditions, as in our case, was never documented. from the aetiopathological point of view, this finding
Giudice et al14 reported the case of a patient who was supports the saliva stasis hypothesis for formation of the
diagnosed with a left tonsillolith and 2 years prior with tonsilloliths and brings them closer to the pathology of
lithiasis of the left Wharton’s duct. They found no an- salivary glands. However, larger studies including a de-
atomical connection between the left tonsil and the left tailed radiological analysis, as in our case, are needed to
submandibular gland. However, the reason for that further investigate this possible aetiology of tonsillo-
could be that, similar to our case, the radiologist did not liths. Additionally, future studies should consider using
actively look for the possibility of an accessory salivary MRI, specifically MR sialography, in clarifying the
duct and, furthermore, the duct remained unobserved nature and course of the accessory salivary duct be-
during surgery. tween the tonsillar calcification and the salivary gland.
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