Professional Documents
Culture Documents
Departments of Pathology, Pulmonology and Oral Surgery, Free University, Amsterdam, The Netherlands
ment, i.e, affected lymph nodes in the head and Apparently healthy patients with one or more
neck area, are included. sarcoid granulomas in the oral region, in which
additional examination justified the diagnosis of
Patients with already proven sarcoidosis, who sarcoidosis
also had lesions in the oral region A number of patients have been described in
In some of the patients with already proven which an oral lesion, microscopically diagnosed
sarcoidosis, the oral involvement consisted of as a sarcoid granuloma, led to the detection of
one or more nodules in the oral mucosa, generalized sarcoidosisl.,33,44"7, KERR 30 des-
without preference for any specific site 12 •22,40,45. cribed a 20-year-old man with multiple swollen
A few cases have been reported of involvement lymph nodes in the parotid, submental, sub-
ofthejaw bones 4. 2 1,42. Involvement of glandular mandibular, superficial and deep cervical area.
structures in the oral cavity is mentioned by Roentgenographs of the chest showed enlarge-
KALMAN 29 as well as by NARANG & DIXON 37. A ment of nodes in the upper mediastinum. A
decreased level of a-amylase and increased submental node was examined microscopically.
levels of albumine and lysozyme have been The appearence was that of sarcoidosis.
observed by BEELEY & CHISHOLM2 in parotid
saliva from a patient suffering from sarcoidosis Apparently healthy patients with one or more
with salivary gland involvement. These observ- sarcoid granulomas in the oral region, in which
ations were interpreted as the result of damage the diagnosis ofsarcoidosis could not be proven or
to the gland. Some years earlier the value of remained somewhat debatable
salivary gland function tests in diagnosing SCHROFF 46 has described a 48-year-old female
sarcoidosis and in monitoring the response to with a swelling of the whole thickness of the
possible therapy was described by CHISHOLM et cheek of 4 weeks duration. The lesion was
al. to • biopsied and reported as being of tuberculous
nature. All additional tests, chest films and
Patients with already proven sarcoidosis in which other examinations were negative for tubercu-
an oral biopsy of apparently healthy tissue was losis, which led the author to suggest the
taken diagnosis of sarcoid. Several of such cases have
CAHN et al,' have described a group of 23 been reported thereafter J,9,2J,JI,54,56.
patients with known sarcoidosis in which NITZAN & AZAR 39 have described a 62-year-
palatal biopsies of clinically normal areas were old woman with an indurated submandibular
performed. They were able to demonstrate the mass, measuring about 3 em. The radiograph
presence of sarcoid granulomas in 38% of their revealed a sialolith in the submandibular gland.
cases, TILLMAN'3 described a 66-year-old man The clinical diagnosis was sialolithiasis. The
suffering from sarcoidosis. Oral examination submandibular gland was removed together
showed a normal aspect of the gingiva. with a surrounding lymph node. That lymph
Nevertheless, a gingival biopsy was performed node showed the features of sarcoidosis. No
to rule out amyloidosis. More-or-less as a Kveim test was performed. All other exami-
surprise, the microscopic findings were con- nations were negative for sarcoidosis. The
sistent with sarcoidosis. A biopsy of the labial Mantoux intracutaneous test was negative. The
glands of the lower lip has been reported patient was suffering from migrating arthritic
positive in a number of patients suffering from pain and swellings, xerophthalmia, dyspnea and
sarcoidosis'v-":". NESSAN & JACOWAY even hypertension. Because ofthose symptoms it was
found 58% non-caseating granulomas in bio- felt that this patient, in spite ofnegative findings
psies of the labial glands in a total number of75 elsewhere in the body, suffered from systemic
patients with known sarcoidosis". sarcoidosis. Nitzan and Azar mentioned that
ORAL SARCOIDOSIS 25
Case reports
Case 1. In May 1980, a 23-year-old man was referred
because of multiple nodules on the hard and soft
palate. The nodules were slightly elevated, measured a
few mm and were of a firm elastic consistency. There
was no ulceration (Fig. 1). The patient had his natural
dentition. Possible irritating factors which could have Fig, 2. The chest film showing bilateral enlargement of
played a role in the initiation of the palatal nodules the hilar lymph nodes without pathological changes in
could not be detected, although heavy smoking may the lung fields.
have been of significance. The duration of the
presence of the nodules was unknown to the patient.
This man was known to suffer from sarcoidosis, the
first clinical manifestations being multiple subcu- The patient will be seen at 3-monthly intervals by the
taneous nodules on his legs and arms. The chest film Department of Pulmonology, The nodules of the
had demonstrated bilateral enlargement of the hilar palate have not shown any change during a 6 months
lymph nodes (Fig. 2). follow-up period.
One of the palatal nodules was biopsied, The
histopathology was compatible with a sarcoid granu- Case 2. In June 1977, a 69-year-old woman was
loma (Fig. 3), Healing of the biopsy wound was referred because of a small submucosal nodule on the
uneventful. No treatment has been instituted so far. tip of the tongue of some months duration. On
palpation, a firm, submucosal, somewhat mobile
nodule was felt. The overlying mucosa was intact. The
suffer from sarcoidosis. The third patient, granuloma: characterization of a model system
however, was apparently healthy. A sub- for infectious and foreign body granulomatous
inflammation using soluble mycobacterial, his-
mandibular, rather well-circumscribed swelling
toplasma and schistosoma antigens. Immunol.
was clinically diagnosed as being most likely a 1973: 24: 511-529.
salivary gland tumor. No sialography or 7. CAHN, L. R., EISENBUD, L., BLAKE, M. N. &
cytologic puncture, nor any additional exami- STERN, D.: Biopsies of normal-appearing palates
nation was felt necessary at that time. The true of patients with known sarcoidosis; a preliminary
report. Oral Surg, 1964: 18: 342-345.
nature of the swelling, being an enlarged lymph
8. CAIN, H. & KRAUS, B.: Mehrkcrnige Riesenzellen
node, first became evident at operation, thus in Granulomen. Neu-ordnung der Binnen-
illustrating the limitation of a provisional structur nach Konfluenz von Zellen des
diagnosis by clinical judgement only. Makrophagensystems. Virchows Arch. A. Path.
Anat. und Histol. 1980: 385: 309-333.
There are several reports mentioning the
9. CAMPBELL, 1.: Sarcoidosis or tuberculosis? Br,
presence of sarcoid granulomas in biopsies of Dent. J. 1944: 77: 159-163.
apparently normal oral tissue, especially from 10. CHISHOLM, D. M., LYELL, A., HARoON, T. S.,
the palate and the lower lip, in patients with MASON, D. K. & BEELEY, 1. A.: Salivary gland
proven sarcoidosis. We hesitate, however, to function in sarcoidosis: report of a case. Oral
Surg, 1971: 31: 766-771.
advocate the use of such biopsies when
II. Corrmn, H., HESS, M. W., KELLER, H. D., RODS,
gathering supporting evidence for the diagnosis B. & Tosr, P.: Macrophage kinetics and granu-
of sarcoidosis only, since staging of the disease loma formation. In: Abstracts Int. Siena
is usually based upon the radiographic findings Sarcoidosis Symposium. Siena, Italy, 1979.
12. COVEL, E.: Boeck's sarcoid of mucous mem-
on the chest films. On the other hand, one
brane; report ofa case. Oral Surg, 1954: 7: 1242-
should know that an oral sarcoid granuloma 1244.
can be an early manifestation of sarcoidosis and 13. CRETIEN, J.: Course and treatment of sarcoidosis
that such a patient should at least be seen by the in 350 patients. 3rd European Conf. on
pulmonologist for further evaluation. Sarcoidosis and other granulomatous disorders.
Novi Sad. Jugoslavia, May 1980.
14. DJURIC:, B., MANDl, L. & VEZENDI, S.:
Acknowledgement - We thank Mrs. M. Mooijen for Sarcoidosis in six European cities. In: JONES
her help in preparing the manuscript and Mr. G. J. WILLIAMS, W. & DAVIES, B. H. (eds.): 81h Int.
Oskam for photography of the histologic slides. Con]. on sarcoidosis and other granulomatous
diseases. Alpha Omega Pub\. Ltd. 1980,527-531.
15. EULE, H., ROTH, 1. & WEIDE, W.: Clinical and
functional results of a controlled clinical trial of
References the value of prednisolone therapy in sarcoidosis,
1. AMSDEN, A. F. & BOROS, D. L.: Fe-receptor stages I and II. In: JONES WILLIAMS, W. &
bearing macrophages isolated from hypersensi- DAVIES, B. H. (eds.): Bth Int. Conf. on sarcoidosis
tivity and foreign-body granulomas. Am. J. and other granulomatous diseases. Alpha Omega
Pathol. 1979: 96: 457-473. Publ. Ltd. 1980, 624-628.
2. BEELEY, J. A. & CHISHOLM, D. M.: Sarcoidosis 16. FRITSCH, P.: Quantification et renouvellement
with salivary gland involvement: biochemical des cellules des alveoles et bronchioles des
studies on parotid saliva. J. Lab. Clin. Med. 1976: poumon des rats. Thesis. Rapport CEA-R-4947,
88: 276-281. 1979.
3. BERNIER, L. J. & TlECKE, R. W.: Sarcoidosis. J. 17. GORDON, L. 1., DOUGLAS, S. D. & KAY, N. E.:
Oral Surg. 1951: 9: 256-257. Inhibition of neutrophil migration by sarcoid
4. BETTEN, B. & KOPPANG, H. S.: Sarcoidosis with sera, with partial reversal by trissacharide of N-
mandibular involvement; report of a case. Oral acetyl glucosamine, a lysozyme inhibitor. In:
Surg. 1976: 42: 731-737. JONES WILLIAMS, W. & DAVIES, B. H. (eds.): 8th
5. BHOOLA, K. D.: Changes in salivary enzymes in Int. Conf. on sarcoidosis and other granulomatous
patients with sarcoidosis. Oral Surg, 1965: 20: diseases. Alpha Omega Publ. Ltd. 1980, 84-88.
877-879. 18. GREER, R. O. & SANGER, R. G.: Primary intraoral
6. BOROS, D. L. & WARREN, K. S.: The bentonite sarcoidosis. J. Oral Surg. 1977: 35: 507-509.
28 VAN MAARSSEVEEN, VAN DER WAAL, STAM, VELDHUIZEN AND VAN DER KWAST