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Dentomaxillofacial Radiology (2015) 44, 20150090

ª 2015 The Authors. Published by the British Institute of Radiology


birpublications.org/dmfr

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.

Keywords: palatine tonsil; salivary duct; saliva

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.

Case report Discussion

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).

Dentomaxillofac Radiol, 44, 20150090 birpublications.org/dmfr


Tonsillolith and accessory duct of the salivary gland
I Pirkl et al 3 of 4

abscess or tonsillectomy. Laboratory findings were all


in normal range. Lithogenic diathesis was excluded
based on normal ultrasound findings of the salivary
gland, kidneys and gallbladder. During the standard
tonsillectomy surgery and removal of the stone, we
found a flow of blocked saliva and, more surprisingly,
a large accessory salivary duct (Figure 2). Although we
performed the routine pre-operative CT scans, no ac-
cessory duct or fistula formation was reported by the
radiologist. However, after the surgery and intra-
operative visualization of the duct, we collaborated with
radiologists and analysed the pre-operative CT scan in
detail. We found a well-defined, hypodense fistulous
tract measuring 6 mm in width and 36 mm in length,
extending from the lower tonsillar pole towards the left
submandibular gland. This was defined as a large ac-
cessory duct of the left submandibular gland with a di-
rect opening at the lower level of the tonsillar pole
(Figure 4). The inability to diagnose this pre-operatively
can be attributed to the fact that a similar case did not
present itself before, and therefore its possibility was not
Figure 2 Intraoperative intraoral picture showing the opening of the
accessory salivary duct (SD) of the left submandibular gland at the
taken into account. Furthermore, there was a possibility
level of the lower tonsillar pole and tonsillolith (T) in the left tonsil. that the duct observed intraoperatively was not the ac-
cessory but rather the main duct of the submandibular
gland, in which case a submandibulectomy would have
glands.1,4,6 The second theory similarly explains the to be performed. Intraoperative probing of the left or-
mechanism by which these stones are formed by cal- ifice of the submandibular gland was normal, so we
cification of peritonsillar abscess.3,5 However, a dif- decided to leave the submandibular gland intact and
ferent theory was presented by Mishenkin and Shtil9 closely monitor the patient in the post-operative period
hypothesizing that tonsilloliths are a part of the pa- for signs of possible submandibular swelling and/or
thology of salivary glands. They performed a histo-
logical examination of the adjacent tissue of
a tonsillolith and found small salivary gland lobuli
with efferent ducts surrounded by lymphoid tissue.
Their conclusion was that stone formation results
from stasis of saliva in the efferent ducts of the small
accessory salivary glands in the oral cavity, owing to
their mechanical obstruction caused by post-
tonsillectomy scar or chronic inflammation. In our
case, a tonsillolith was formed only by the saliva from
the accessory duct of the large salivary gland. In-
flammation was excluded as an aetiological factor as
the patient was without any inflammation of the
tonsils. This finding put emphasis on saliva stasis and
accessory ducts of salivary glands as a possible cause
of tonsillolith as hypothesized by Mishenkin and
Shtil.9 It is important to note that probably, in most
cases, the minor salivary glands are the aetiological
cause of a tonsillolith and not the major glands, which
can be diagnosed with a comprehensive radiological
assessment (CT, MRI).
The accessory duct of the submandibular gland was
reported in only a few cases up to now.10–13 This anom-
aly was described in most cases as a duplication of the
main Wharton’s duct, with the uniform opening at the
orifice or as a separate opening in the oral cavity.13
Gaur et al11 even reported three ducts with each open-
ing separately at the floor of the mouth. Our patient had Figure 3 A calcified mass (tonsillolith) is removed after tonsillec-
no medical history of chronic tonsillitis, peritonsillar tomy, measuring 31 mm in length.

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Tonsillolith and accessory duct of the salivary gland
4 of 4 I Pirkl et al

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.

References

1. Mesolella M, Cimmino M, Di Martino M, Criscuoli G, Albanese 9. Mishenkin NV, Shtil AL. Calculi of palatine tonsils and causes of
L, Galli V. Tonsillolith. Case report and review of the literature. their formation. [In Russian.] Vestn Otorinolaringol 1965; 27:
Acta Otorhinolaryngol Ital 2004; 24: 302–7. 110–12.
2. Aspestrand F, Kolbenstvedt A. Calcifications of the palatine 10. Rose BH. Bifurcation of the submaxillary duct. Am J Surg 1932;
tonsillary region: CT demonstration. Radiology 1987; 165: 17: 257–8. doi: 10.1016/S0002-9610(32)90492-9
479–80. doi: 10.1148/radiology.165.2.3659369 11. Gaur U, Choudhry R, Anand C, Choudhry S. Submandibular
3. Hiranandani LH. A giant tonsillolith. J Laryngol Otol 1967; 81: gland with multiple ducts. Surg Radiol Anat 1994; 16: 439–40. doi:
819–22. doi: 10.1017/S0022215100067748 10.1007/BF01627668
4. Weller CV. The incidence and pathogenesis of tonsillar con- 12. Gadodia A, Seith A, Neyaz Z, Sharma R, Thakkar A. Magnetic
cretions. Ann Otol Rhinol Laryngol 1924; 33: 79–127. doi: resonance identification of an accessory submandibular duct and
10.1177/000348942403300102 gland: an unusual variant. J Laryngol Otol 2007; 121: e18. doi:
5. Kimura H, Ohashi N, Nakagawa H, Asai M, Koizumi F. Large 10.1017/S0022215107008602
tonsillolith mimicking peritonsillar abscess: a case report. Auris 13. Kuroyanagi N, Kinoshita H, Machida J, Suzuki S, Yamada Y.
Nasus Larynx 1993; 20: 73–8. doi: 10.1016/S0385-8146(12) Accessory duct in the submandibular gland. Asian J Oral Max-
80213-6 illofac Surg 2007; 19: 110–12. doi: 10.1016/S0915-6992(07)80027-6
6. Silvestre-Donat FJ, Pla-Mocholi A, Estelles-Ferriol E. Martinez- 14. Giudice M, Cristofaro MG, Fava MG, Giudice A. An unusual
Mihi V. Giant tonsillolith: report of a case. Med Oral Patol Oral tonsillolithiasis in a patient with chronic obstructive sialoadenitis.
Cir Bucal 2005; 10: 239–42. Dentomaxillofac Radiol 2005; 34: 247–50. doi: 10.1259/dmfr/
7. Jones JW. A tonsillolith. Br Dent J 1996; 180: 128. doi: 10.1038/sj. 19689789
bdj.4808996 15. Suarez-Cunqueiro MM, Dueker J, Seoane-Leston J, Schmelzeisen
8. Caldas MP, Neves EG, Manzi FR, de Almeida SM, Bóscolo FN, R. Tonsilloliths associated with sialolithiasis in the submandibular
Haiter-Neto F. Tonsillolith—report of an unusual case. Br Dent J gland. J Oral Maxillofac Surg 2008; 66: 370–3. doi: 10.1016/j.
2007; 202: 265–7. doi: 10.1038/bdj.2007.175 joms.2006.11.014

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