The gigantiform salivary calculus

The gigantiform salivary calculus

Int. J. Oral Surg. 1982: 11: 135-139 (Key words: calculus. salil'ary .. gland, submandibular .. hi...

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Int. J. Oral Surg. 1982: 11: 135-139 (Key words: calculus. salil'ary .. gland, submandibular .. hi.<;lOlogy .. microradiography,' SEAl)

The gigantiform salivary calculus GbRAN ISACSSON AND NILS-ERIK PERSSON Departments of Oral Pathology and Oral Surgery, School ojDentistry, Karolinska Institute, Stockholm, Sweden

ABSTRACT - The size of salivary calculi may range from small particles to large concrements of several centimetres. One case of a gigantiform salivary calculus located in the Wharton duct of a 48-year-old man is presented. After surgical removal of the 3.6 em long concrement, it was split into 4 parts. The different parts were used for routine histological studies, production of microradiographs of ground sections and for scanning electron microscopy. Decalcified sections disclosed a specimen made of a homogenous central nucleus and a peripherallamellation. The same morphological picture was seen on the microradiographs, where the nucleus showed dense mineralization, and the peripheral part alternating rings of high and low mineral content. Microorganisms, mostly thread-like organisms, were found throughout the entire calculus in specially stained sections. Scanning electron microscopy confirmed the histologic and microradiographic findings. The presence of microorganisms also in the nucleus of the calculus seems to be one important aetiological factor favouring the formation of this gigantiform concrement.

(Received Jar publication 16 June, accepted 20 October 1981).

The aetiology and pathogenesis of salivary calculi have for a long time been debated in the literature. On the basis ofmorphology, different hypotheses concerning the aetiology of salivary calculus have been discussed ll ,13. It has been suggested that fungus, bacteria or foreign bodies are significant factors in the formation of salivary calculi (for references see ISAcssoN'). It has also been proposed that desquamated epithelial cells may form a nucleus, thus acting as a focus for the expansion and development of the calculi~. Using electron microscopy, VAHL et aZ. 15 and HOEHLING et aZ: 7 found bacteria mineralized to a varying degree in the peripheral

parts of salivary calculi, while in the central parts no bacteria were seen. The same observation was made by ANNEROTH et a[.2. The size of salivary calculi varies from small particles described as salivary sand l4 to large concrement formations 4 • The average size of the 2 largest dimensions of salivary calculi lies between 3.3 mm and 17.9 mm l • Larger calculi are very rare and therefore there is reason to report a salivary calculus of unusual size, the extirpated specimen having been studied by routine histology, microradiography and scanning electron microscopy in order to evaluate the character of the concrement.

0300-0785/82/020135-05$02.50/0 © 1982 Munksgaard, Copenhagen

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Fig. 2. Radiographic examination of the floor of the mouth revealing a long radiopaque body extending from the canine to the duct bend at the border of the mylohyoideus muscle. Fig. J. The patient showing an oedematous and swollen floor of the mouth where the Wharton duct is bulging.

Case report A 48-year-old man was referred for treatment because of an infection in the floor of the mouth with symptoms of pain, tenderness, swelling and fever. The acute infection had lasted for 4 weeks. An undiagnosed swelling of the submaxillary area and transient discomfort had been present for 5 years. The acute periods were mostly associated with infections in the ear, nose and throat. Between the acute periods the patient was almost asymptomatic. In the last 2 years he had noticcd a slight enlargement of the extruoral part of the submandibular region. He thought it was caused by his teeth, which had repeatedly been examined and treated. The patient, a restaurant proprietor, smoked about 40 cigarettes a day. With the exception of a slight gastritis the patient was healthy. Clinical examination revealed a red and swollen floor of the mouth on the left side and pus was secreted from the orifice of the left Wharton duct (Fig. I). Ex:traorally, a tender submandibular swelling was visible. No calculus was palpable because of the oedema, but radiographic examination disclosed a long radiopaque body extending from the region of the canine to the duct bend at the border of the mylohyoideus muscle (Fig. 2). The acute infection was treated with phenoxymethyl penicillin (2.4 gjday). The symptoms gradually decreased and after 12 days the patient was asymptomatic. However, the sub-

mandibular gland was still enlarged. The calculus could now easily be palpated. Still protected by penicillin, the patient was operated upon under local anaesthesia with a lingual nerve block. A probe was inserted from the orifice and an incision was made through the mucosa over the calculus. The calculus was exposed, pressed forward and slipped out easily. A 5 cm long iodoform gauze drain was inserted in the duct. The gauze was sutured and the rn,ucosa closed with catgut. After removing the drain two days after surgery, a slight salivary flow could be seen. The wound healed uneventfully. 2 months after surgery, sialography showed a widened duct with small degenerative changes of the gland parenchyma. However, the function of the gland appeared clinically normal. At a I-year followup apparently normal salivary flow rate was seen. The calculus measured 3.6 cm in length and exhibited a rough "papillary" surface (Fig. 3). The specimen was split into 4 parts with a sharp knife. The 2 end parts were examined by scanning electron microscopy after critical point drying and coating

Fig. 3. The extirpated calculus, was yellow in colour, rough in texture and almost 3.6 cm in length.

SALlYARY CALCULUS with a 500 A thick layer of gold. The SEM-examined specimens were then decalcified for 3 h in a 24% EDTA solution at 37°C, dried, gold-coated and examined a second time with the scanning electron microscope. One middle part of the calculus was embedded in methyl methacrylate and used for production of ground sections according to the method described by ANNEROTH et al. I • The other middle part of the calculus was decalcified in 10% ronnie acid, paraffin-embedded, sectioned and stained with haematoxylin eosin and ad modwn BROWN & BRENN', III order to visualize microorganisms. In haematoxylin eosin stained sections it was found that the peripheral parts of the calculus were partly covered with residues from a fibrous connective tissue which occasionally was necrotic. In the outermost area of the calculus a lamellated pattern was seen, but this lamellation decreased towards the centre of the calculus where the structure appeared homogenous. The Brown and Brenn staining disclosed an enormous amount of microorganisms in all parts of the calculus. Thread-like organisms dominated, but also cocci and rods were present (Fig. 4). The microradiograph revealed a calculus built up around a heavily calcified central nucleus, and the calcified homogenous area extended over more than half of the diameter of the concrement. In the more peripheral parts a slightly lamellated pattern appeared with alternating high and low mineral content. Scanning electron microscopy of the undecalcified parts showed that the outermost part of the calculus

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Fig. 5. Detail form the calculus surface showing an

organic film with a thread-like structure, probably of mucinous origin. SEM x 500.

was covered by a sheet of an organic film (Fig. 5). In some areas the surface was denuded where small grainy calcified structures were seen. Close to the surface a typical lamellated pattern was visible where the lamellae were quite homogenous. In the intermediate and in the most central part of the calculus, a fine, grainy structure of calcified sphericles was seen (Fig. 6). No microorganisms were apparent in the central parts of the undecalcified calculus. The decalcified specimen confirn1ed the findings from the Brown & Brenn staining. Cocci- and rod-like organisms were mainly located at the outermost parts of the calculus.

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Fig. 4. Enonnous numbers of microorganisms were

found on all levels of the calculus. Here in the nucleus, thread-like organisms dominated, but also cocci and rods were present. Brown & Brenn, x 620.

Discussion Gigantiform calculus in the Wharton duct is extremely rare4 ,16, probably because even small calcified partiCles may occlude the duct orifice,

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Fig. 6. The intermediate part of the calculus showing

lamellated patterns and a grainy structure which is probably composed of mineralized sphericules. SEM x 1200.

which gives rise to clinical symptoms and induces the patient to ask for treatment. In the present case, in which the patient thought his teeth were responsible for the discomfort, pain and infections did of course lengthen the time before curative treatment. A 5-year duration of symptoms before discovery and treatment is not unusual. ISACSSON & LUNDQUIST" reported a mean duration of symptoms of I year, but in rare cases more than 50 years had elapsed before the calculus was removed. In cases with recurrent episodes of infection the calculus may spontaneously perforate and slip out, so avoiding the need for surgery. In the present case the calculus remained within the duct and grew relatively quickly, perhaps supported by the presence of microorganisms. Thread-like organisms in all parts (also in the centre, i.e. the oldest part) of this gigantiform calculus support the theory of actinomycotic aetiology presented in 1876 by KLEBS'o and later by NAESLUN0 12 • They showed after cultivation that all calculi contained actinomyces. However, Klebs' and Naeslulld's results have not been supported by later investigations (for references see ISACSSON 8 ). ANNEROTH et al. 12 showed that microorganisms were present in

many calculi, but only in the peripheral parts and related to a retrograde secondary infection favoured by salivary stasis. It is obvious, however, that microorganisms may be associated with formation of the central parts of the calculus, i.e. the nucleus. Organic substances from the saliva and from necrotic epithelial and fibrous connective tissue cells are, of course, another organic component. The present calculus was well mineralized and in most parts the mineralized lamellae had flowed together into a homogenous pattern seen on the microradiograph. In the scanning electron microscope, however, these homogenous areas exhibited ultrastructurally a lamellating pattern. This heavy mineralization is probably favoured by a suitable environment for precipitation of calcium phosphate. It is surprising that the salivary gland can produce normal amounts of saliva after heavy infection and stasis. It has been shown in experimental animals that duct ligation gives rise to decrcased flow of saliva and later absence of function of the parencymal cells. However, even after a long period of occlusion the cells may be restored and start to produce saliva again 3 . But in cases ofheavy infection within the gland parenchyma the cells will be destroyed and, of course regeneration is not possible. In the present case the infection was probably localized mainly to the duct. It is necessary, however, to stress the importance of early detection and removal of the calculus in order to avoid infection of the gland, with resulting parenchymal degeneration and subsequent reduction of saliva production.

References L. ANNEROTH. G., ENEROTH, C.-M. & ISACSSON, G.: Morphology of salivary calculus. J. Oral Path. 1975: 4: 257-265. 2. ANNEROTH, G., ENEROTH, C.-M., ISACSSON, G. & LUNDQUIST, P.-G.: Ultrastructure of salivary calculi. Scalld. J. Dent. Res. 1978: 86: /82-192. 3. BHASKAR, S. N., BOLDEN, T. E. & WEINMANN, J. P.: Experimental obstructive adenitis in the mouse. 1. Dent. Res. 1956: 35: 852-862.

SALIVARY CALCULUS 4. BRacHERIOU, C. & BaRGHGRAEF, S. C.: Giant sialith of the submandibular gland. Rev. Stomat%g. ChiI'. Maxillofac. 1972: 73: 517-520. 5. BROWN, J. H. & BRENN, L.: A method for the differential staining of gram-positive and gramnegative bacteria in tissue sections. Bull. Johns Hopkins Hasp. 1931: 48: 69-73. 6. HANSZEL, F.: libel' speichelsteinbildung. Wien. K/in. Wochenschr. 1900: 13: 160-163. 7. HOEHLING, H. J., PFEFFERKORN, F., RADICKE, J. & VAHL, J. : Elektronmikroskopische Untersuchungen zur organischen Matrix und Kristallbildung in menschlichen Speichelstein. DIsch. Zahnartztl. Z. 1969: 24: 663-670. 8. ISACSSON, G.: Salivary calculi. Thesis. Stockholm 1977. 9. ISACSSON, G. & LUNDQUIST, P.-G.: SaIivary calculi as an etiological factor in chronic sialadenitis of the submandibular gland. Laryng. Rhinal. 1982: in press. 10. KlEDS, E.: Beitrage zur Kenntnis der pathogenes des Schistomyceten. Arch. Exp. Pat/wI. 1876: 5: 350-377. II. MATHIS, H.: Zur Frage del' Sialolithiasis. In:

Fortschritte del' Kiefer- und Gesichtschirurgie. Thieme, Stuttgart 1960.

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12. NAESLUND, C.: Studien iiber Speichelsteinbildung. AC/{J Pathol. Microbial. Scand. 1925: 2: 244-276. 13. RAUCH, S.: Die Speicheldriisen des Menschen. Georg Thieme, Stuttgart 1959, p. 434. 14. RAUCH, S., DUCKERT, A., MORGENTHALER, P. & KOESTlN, H.: Beitrag zur Speichelsteingenesc. Sclllveiz Med. Wochenschr. 1960: 90: 460-464. 15. VAHL, J., PFEFFERKORN, G. & HOEHLING, P. G.: Sublichtmikroskopische Untersuchungen am mensch lichen Speichelstein. Disch. Zulll/aer::t!. Z. 1968; 23: 39-44. 16. YOEl, J.: PtJlhology and surgery of the salivary glands. Charles C. Thomas, Springfield, Illinois 1975, p. 860.

Address:

Gown fsacsson. Department of Oral Pathology School of Dell/istry Karolinska I/ls/itllte Box 4064 S-!4!04 Huddi/lge. Sweden