British Journal of Oral and Moxillofacial Surgery ( 1996) 34, H-541

0 1996 The British Association of Oral and Maxillofacial Surgeons

Letters to the Editor
POLYMORPHOUS LOW-GRADE
ADENOCARCINOMA OF THE PALATE IN A
16-YEAR-OLD MALE PATIENT
Sir,
Polymorphous low-grade adenocarcinoma (PLGA) of
minor salivary glands is a recently described intraoral neo-
plasm that has also been named lobular carcinoma,
terminal duct carcinoma,2 and low-grade papillary aden-
ocarcinoma.3 Wenig et ~1.~ reviewed the literature and found
that from 125 cases of PLGA, 63% involved the palate,
14% the buccal mucosa, 11% the upper lip, 7% the retromo-
lar region, and 6% other intraoral sites. Extraoral sites
including the major salivary glands are rare, and multiple
synchronous PLGA are very rare.6 PLGA are almost three
times more common in females than in males and generally
present in the sixth decade of life.4 It is seen predominantly
in patients who are between 30 and 70 years old. Among
the patients of known age, the youngest was a 23-old
woman and the oldest was a 94-year-old woman. In this
letter I document what I believe to be the first case of a
PLGA of minor salivary glands of the oral cavity in the
second decade of life.
Case reports
A 16-year-old male patient, presented with firm midline
swelling measuring 3 cm at the junction of the hard and
soft palate of about 4 years duration. No ulceration was
present. The biopsy specimen was reported as PLGA
and wide excision was recommended. There has been no
evidence of recurrence 5 years after operation.
Microscopically, the lesion was submucosal, and dis-
tinctly infiltrative. The predominant architectural pattern
in the central portion of the tumour was one of lobules of
bland neoplastic cells arranged in solid nest (Fig. 1). The
peripheral area showed trabeculae, fascicles, tubules, or
single cells infiltrating surrounding structures (Fig. 2).
Concentric circling by tumour cells around foci of residual
salivary gland acini, around central tumour islands, or
Fig. 1 - Solid nests of isomorphic tumour cells showing nuclei with
bland chromatin and indistinct cell borders. Note zones of
microcystic changes. (H&E, Original magnification x 200).
Fig. 2 - Tnmour infiltration of residual mucous acini.
(H&E, Original magnification x 200).
around nerves or vessels produced a characteristic targetoid
appearance (Fig. 3). A diagnosis of PLGA was made.
PLGA are biologically low-grade neoplasms; recurrence
has been reported to be 17-24%, and the incidence of
regional lymph node metastasis is estimated at 6-9%.4-5
Primary malignant tumours of the salivary glands are
generally regarded as adult neoplasms. A comprehensive
survey of 2410 salivary gland tumors indicated that only
6% of all such neoplasms were diagnosed in the first 20
years of life.* Mucoepidermoid carcinoma represents the
most common malignant subset of pediatric salivary gland
malignant tumours, whereas occurring acinic cell carcinoma
and adenoid cystic carcinoma are rare.g
Arsa J. Minii:
Faculty of Stomatology
Department of Oral Pathology
PO Box 506, Dr SubotiCa 1
11000 Beograd
Yugoslavia
Fig. 3 - Concentric circling of tumour cells around a central focus,
probably an invaded nerve. (H&E, Original magnification x 100).
540
Letters to the Editor 541
References
1.
2.
3.
4.
5.
6.
Freedman PD, Lumerman H. Lobular carcinoma of intra-oral
minor salivary gland origin: report of 12 cases. Oral Surg Oral
Med Oral Path01 1983; 56: 157.
Frierson HF, Mills SE, Garland TA. Terminal duct carcinoma
of minor salivary glands: a nonpapillary subtype of
polymorphous low-grade adenocarcinoma. Am J Clin Path01
1985; 84: 8.
Mills SE, Garland TA, Allen MS. Low-grade papillary
adenocarcinoma of palatal salivary gland origin. Am J Surg
Path01 1984; 8: 367.
Wenig BM, Harpaz N, DelBridge C. Polymorphous low-grade
adenocarcinoma of seromucous glands of the nasopharynx. Am
J Clin Path01 1989; 92: 104.
Vincent SD, Hammond HL, Finkelstein MW. Clinical and
therapeutic features of polymorphous low-grade
adenocarcinoma. Oral Surg Oral Med Oral Path01 1994; 77: 41.
Clayton JR, Pogrel MA, Regezi JA. Simultaneous multifocal
polymorphous low-grade adenocarcinoma: report of two cases.
Oral Surg Oral Med Oral Path01 1995; 80: 71.
Waldron CA, El-Mofty SK, Gnepp DR. Tumors of intraoral
minor salivary glands: a demographic and histologic study of
426 cases. Oral Surg Oral Med Oral Path01 1988; 66: 323.
Eveson JW, Cawson RA. Salivary gland tumours: A review of
2410 cases with particular reference to histological types, site,
age and sex distribution. J Path01 1985; 146: 51.
Jones DC, Bainton R. Adenoid cystic carcinoma of the palate
in a Y-year-old boy. Oral Surg Oral Med Oral Path01 1990; 69:
483.
SUBCUTANEOUS EMPHYSEMA OF THE
PERIORBITAL AREA FOLLOWING
ORTHOGNATHIC SURGERY
Sir,
We thought that a case of a subcutaneous emphysema of
the periorbital region, after orthognatic surgery could be
interesting for all maxillofacial surgeons.
A 37-year-old woman was admitted for correction of a
skeletal Class III malocclusion.
Mandibular and maxillary osteotomy with rigid fixation
and intermaxillary blockage were performed. In the surgical
manoeuvre and more precisely, during the down fracture
stage, the nasal mucosae were interrupted. On the second
postoperative day a sudden swelling of the periorbital soft
tissues was noticed after the patient tried to expel the
accumulation of the endonasal mucor by nose blowing. A
palpatory crepitus typical of the subcutaneous emphysema
was felt. As routine, pharmacologic therapy was performed
because the secondary risk of infection to subcutaneous
emphysema is always a possible complication.
The incident did not have any sequelae and after about
3-4 days a total resolution of the emphysema was observed.
Facial subcutaneous emphysema following orthognatic sur-
gery is an uncommon event but is possible.3-5 Stringer et al.
reported 2 cases of facial subcutaneous emphysema follow-
ing a Le Fort I osteotomy during the postoperative period.
Nannini et al. reported a case of mediastinal emphysema
following a Le Fort I osteotomy which happened 72 h after
the operation. It is now possible to point out several patho-
genetic theories. For example, it is thought that exposition
of the anterior and lateral walls of the rhinopharinx follow-
ing the down-fracture after Le Fort I osteotomy, could
favour the introduction of air in the posterior mediastinum.
The responsibility could be attributed to the use of turbine
or to events connected with postoperative behaviour of the
patient such as coughing, sneezing, and mucous plugging.
We think that another pathogenetic theory could be the
interruption of the nasal mucosae during the down
fracture.
The laceration of the nasal mucosae associated with nose
blowing can cause the passage of the air contained in nasal
cavities to the deep layer of facial structures. So it is import-
ant to avoid the damaging of the nasal mucosae and to
warn the patients about nose blowing postoperatively.
M. Robiony
Assistant Professor
V. Demitri
Resident
F. Costa
Resident
M. Politi
Chief of Department
Department of Maxillo-Facial Surgery
Faculty of Medicine
University of Udine
P.le S. Maria della Misericordia
33100 Udine
Italy
References
1. Demas PN, Braun TW. Infection associated with orbital
subcutaneous emphysema. J Oral Maxillofac Surg 1991; 49:
123991242.
2. Edwards DB, Scheffer RB, Jackler I. Postoperative
pneumomediastinum and pneumothorax following orthognatic
surgery. J Oral Maxillofac Surg 1986; 44: 1377141.
3. McLaughlin, Gilhooly MG. Surgical emphysema complicating
mandibular sagittal split osteotomy. Br J Oral Maxillofac Surg
1984; 22: 269-273.
4. Nannini V, Sachs SA: Mediastinal emphysema following Le
Fort I osteotomy: report of a case. Oral Surg Med Oral Pathot
1986; 62: 508-509.
5. Stringer DE, Dolwick MF, Steed DL. Subcutaneous
emphysema after Le Fort I osteotomy: report of two cases.
J Oral Surg 1979; 37: 115-l 16.