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Adenoid Cystic Carcinoma of Nasal Septum: Report of Two Cases

Authors:
Priya S.R.
Priya S.R.
Devendra A Chaukar
Devendra A Chaukar
Devendra A Chaukar
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Anil KD' Cruz
Anil KD' Cruz
Anil KD' Cruz
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We present two cases of adenoid cystic carcinoma arising from the nasal septum. The first patient was previously untreated. The secondpatient had been operated outside with a presumed diagnosis of a benign lesion. We excised both the lesions with wide margins as isadvisable for this histology. The histopathology examination in the first case showed adenoid cystic carcinoma with no high-risk features. Inthe second case, the histopathology revealed no residual malignancy. Adjuvant treatment was, therefore, not given to either patient. Thepostoperative appearance in both cases was good. They have been disease free on follow-up.
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Adenoid Cystic Carcinoma of Nasal Septum: Report of Two Cases
International Journal of Head and Neck Surgery, September-December 2011;2(3):151-153 151
Adenoid Cystic Carcinoma of Nasal Septum:
Report of Two Cases
1Priya SR, 2Devendra A Chaukar, 3Anil KD' Cruz
1Fellow, Department of Head and Neck Surgery, Tata Memorial Hospital, Mumbai, Maharashtra, India
2Associate Professor, Department of Head and Neck Surgery, Tata Memorial Hospital, Mumbai, Maharashtra, India
3Professor, Department of Head and Neck Surgery, Tata Memorial Hospital, Mumbai, Maharashtra, India
Correspondence: Priya SR, Fellow, Department of Head and Neck Surgery, Tata Memorial Hospital, Mumbai, Maharashtra
India, e-mail: essarpriya@yahoo.com
CASE REPORT
ABSTRACT
We present two cases of adenoid cystic carcinoma arising from the nasal septum. The first patient was previously untreated. The second
patient had been operated outside with a presumed diagnosis of a benign lesion. We excised both the lesions with wide margins as is
advisable for this histology. The histopathology examination in the first case showed adenoid cystic carcinoma with no high-risk features. In
the second case, the histopathology revealed no residual malignancy. Adjuvant treatment was, therefore, not given to either patient. The
postoperative appearance in both cases was good. They have been disease free on follow-up.
Keywords: Adenoid cystic carcinoma, Nasal septum.
10.5005/jp-journals-10001-1073
INTRODUCTION
Adenoid cystic carcinoma (ADCC) arising from the nasal
septum is rare and only a few reports are available in
literature. The nasal cavity and the paranasal sinuses are
seen to have a worse prognosis than other head and neck
sites; however, the nasal cavity tumors have been found to
have a better outcome than sites in the paranasal sinuses.
Surgery with adequate and clear margins followed by
adjuvant radiotherapy when indicated, is the best treatment
modality for adenoid cystic cancers. We report two such
cases that we encountered at our hospital.
CASE REPORTS
Case 1
A 50-year-old man presented with a 6 months history of a
left-sided nasal mass. There was history of occasional blood-
stained discharge. Anterior rhinoscopy revealed a smooth,
nonulcerative mass arising from the cartilaginous nasal
septum, the floor and the lateral wall-being free. There was
no palpable cervical lymphadenopathy. A punch biopsy was
reported as ADCC. A CT scan (Fig. 1A) confirmed that the
bony septum and the paranasal sinuses were clear and that
there were no lymph node metastasis. The chest X-ray was
normal. The tumor was excised by lateral rhinotomy
approach. The columella was divided by a horizontal
incision. The tumor was seen to be arising from the
cartilaginous septum. The entire cartilaginous septum was
removed along with a portion of the bony septum. The
junction of the lateral alar cartilages and the septum was
partly excised for margins.
Grossly the lesion was 1.5 × 1.5 × 1 cm in dimensions.
Histology was adenoid cystic carcinoma of a predominantly
cribriform pattern. There was no invasion of the underlying
cartilage. There was absence of perineural invasion.
Fig. 1A: CT showing lesion on anterior nasal septum
Fig. 1B: Postoperative picture showing minimal deformity
Priya SR et al
152 JAYPEE
of 3 years duration. The histopathology of the mass was
found to be adenoid cystic carcinoma, and he attended our
hospital for further evaluation. On examination, the right
side of his nasal septum showed postoperative changes, with
a doubtful residual growth (Fig. 2A). A review of the slides
from the previous surgery was done, and the histology was
confirmed to be ADCC. A computed tomography (Fig. 2B)
revealed a thickened anterior nasal septum, the rest of the
septum and the lateral wall-being normal. Keeping in view
that the previous excision had been with a presumption of a
benign lesion, revision surgery was decided upon. Excision
was done of the cartilaginous septum along with a cuff of
bony septum (Fig. 2C). The specimen showed only
inflammatory changes on histopathological examination,
with no residual malignancy. Consequently, no adjuvant
treatment was given. The early postoperative period was
uneventful and the appearance was satisfactory.
Both above patients have been disease free for 15 months
till the time of writing this paper.
DISCUSSION
ADCC is the second most common tumor of the nasal cavity
and paranasal sinuses after squamous cell carcinoma.
Sinonasal ADCC accounts for 10 to 25% of all head and
neck ADCCs.1 The nasal cavity is the second most common
site after the maxillary sinus2 for this tumor. However,
ADCC limited to the nasal septum has been reported only
rarely; the number of patients seen being given as six3 and
three4 in the literature.
ADCC can be classified histologically into cribriform,
tubular and solid types. The cribriform is the commonest
and has the best outcome, while the solid tumors are least
common and have the worst prognosis.5 Patients with nasal
cavity disease had better overall and disease-specific survival
as compared with tumors in other sites in the PNS.6 The
clinical stage, particularly the T stage, is the critical factor
in deciding the outcome of salivary gland cancer as per data
collected at Memorial Sloane Kettering Center.7 Other
prognostic factors are size of tumor, margins of resection,
lymph node metastasis, perineural invasion or invasion of
bone vasculature muscle or extraglandular tissue. Salivary
gland tumors of T1 and T2 stage, i.e. less than 4 cm in size,
do well irrespective of histological grade. Radiotherapy is
beneficial for tumors over this size, having little impact over
smaller tumors.8
The recommended treatment of sinonasal ADCC is
complete surgical resection with adequate and tumor-free
margins, followed by radiotherapy. Radiation is not curative,
but in the postoperative setting has been shown to be
effective in improving local control. Patients who undergo
surgery with postoperative radiotherapy as the primary
Fig. 2A: Septum showing doubtful lesion anteriorly
Fig. 2B: CT showing thickened anterior septum
Fig. 2C: Specimen showing lesion in the middle of
cartilaginous septum
The postoperative period was uneventful (Fig. 1B).
Crusting was the only significant complaint, which was
managed by douching. Since it was a low-grade, low-staged
tumor, no adjuvant treatment was given.
Case 2
A 53-year-old man presented with a history of having been
operated upon 2 months before, for a right nasal septal mass
Adenoid Cystic Carcinoma of Nasal Septum: Report of Two Cases
International Journal of Head and Neck Surgery, September-December 2011;2(3):151-153 153
treatment modality have significantly improved overall as
well as disease-specific survival, as compared with those
being treated with other modalities including surgery alone
and radiation alone.5 Aggressive therapy for the primary
tumor does lead to a high and long-term local control rate,
though it does not influence survival, because of metastatic
disease.8
ADCCs typically recur locally, the rate of recurrence
being 100% for solid tumors and 59-89% for lower grade
tumors.9 A recent study found an overall recurrence rate of
65% in sinonasal ADCC, despite surgery and radiotherapy.6
These tumors metastasize frequently (35 to 50%), usually
to lung and bone.10 The 5-year survival rates for low-stage,
low-grade tumors are roughly 85%, but at 10 years, all grades
do equally, with overall survival of less than 50%.11
Both patients in this report presented early. The tumor
was present in a relatively easily accessible area of the
cartilaginous nasal septum. Surgically clear margins were
achieved without significantly disturbed cosmesis. The
pathology of the tumor in the first patient was favorable viz
a T1 lesion, cribriform pattern, absence of perineural
invasion, clear margins. Hence, no further adjuvant
treatment in the form of radiotherapy was found to be
warranted. Surgery in the second case was to ensure wide
margins of residual malignancy, if any. Secondary
reconstruction for nasal dorsum saddling will be planned,
if so required, at a later stage.
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... It has been reported that ACC originating from the nose and maxillary sinus are associated with poor prognosis [4]. For low-grade tumors, five-year survival rates of 85 % have been seen while 10-year survival rates irrespective of the grades of 50 % have been reported [5]. ...
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... It has been reported that ACC originating from the nose and maxillary sinus are associated with poor prognosis [1]. For low-grade tumors, fiveyear survival rates of 85% have been seen while 10-year survival rates irrespective of the grades of 50% have been reported [4]. ...
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Prognostic factors in malignant tumours of the salivary glands
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  • Dec 2009
  • BRIT J ORAL MAX SURG
Salivary gland tumours are a relatively rare group of lesions best managed in specialist centres. We review some of the factors that influence their prognosis. Clinical stage is the most important, with large malignancies having a poor prognosis regardless of histological grade and other features such as perineural invasion. Even high grade neoplasms may do well when they are small. A helpful guide to the management of salivary cancers is the "4 cm" rule. © 2009 The British Association of Oral and Maxillofacial Surgeons.
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Nasal and paranasal sinus carcinoma: Are we making progress? A series of 220 patients and a systematic review
Article
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  • Nov 2002
The authors reviewed treatment results in patients with nasal and paranasal sinus carcinoma from a large retrospective cohort and conducted a systematic literature review. Two hundred twenty patients who were treated between 1975 and 1994 with a minimum follow-up of 4 years were reviewed retrospectively. A systematic review of published articles on patients with malignancies of the nasal and paranasal sinuses during the preceding 40 years was performed. The 5-year survival rate was 40%, and the local control rate was 59%. The 5-year actuarial survival rate was 63%, and the local control rate was 57%. Factors that were associated statistically with a worse prognosis, with results expressed as 5-year actuarial specific survival rates, included the following: 1) histology, with rates of 79% for patients with glandular carcinoma, 78% for patients with adenocarcinoma, 60% for patients with squamous cell carcinoma, and 40% for patients with undifferentiated carcinoma; 2) T classification, with rates of 91%, 64%, 72%, and 49% for patients with T1, T2, T3, and T4 tumors, respectively; 3) localization, with rates of 77% for patients with tumors of the nasal cavity, 62% for patients with tumors of the maxillary sinus, and 48% for patients with tumors of the ethmoid sinus; 4) treatment, with rates of 79% for patients who underwent surgery alone, 66% for patients who were treated with a combination of surgery and radiation, and 57% for patients who were treated exclusively with radiotherapy. Local extension factors that were associated with a worse prognosis included extension to the pterygomaxillary fossa, extension to the frontal and sphenoid sinuses, the erosion of the cribriform plate, and invasion of the dura. In the presence of an intraorbital invasion, enucleation was associated with better survival. In multivariate analysis, tumor histology, extension to the pterygomaxillary fossa, and invasion of the dura remained significant. Systematic review data demonstrated a progressive improvement of results for patients with squamous cell and glandular carcinoma, maxillary and ethmoid sinus primary tumors, and most treatment modalities. Progress in outcome for patients with nasal and paranasal carcinoma has been made during the last 40 years. These data may be used to make baseline comparisons for evaluating newer treatment strategies.
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Adenoid Cystic Carcinoma of the Paranasal Sinuses or Nasal Cavity: A 40-Year Review of 35 Cases
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We retrospectively reviewed 35 cases of adenoid cystic carcinoma that had originated in the minor salivary glands of the paranasal sinuses or nasal cavity. All patients had been seen at two tertiary-care referral centers in western New York State between 1960 and 2000. Twenty patients had been treated with surgery and adjuvant radiotherapy, 10 patients with surgery alone, three with radiotherapy alone, and two with concurrent radiotherapy and chemotherapy. During the study, 22 patients developed recurrent disease--11 locally; three distantly; seven locally and distantly; and one locally, regionally, and distantly. At the conclusion of the study, 14 patients were alive and disease-free, and eight were alive with disease; 10 patients had died with disease, and three had died of other causes with no evidence of disease. Adenoid cystic carcinoma of the paranasal sinuses or nasal cavity is an aggressive neoplasm that results in a high incidence of both local recurrence and distant metastasis, regardless of treatment modality. Most cases are ultimately fatal, although long disease-free intervals have been observed. A combination of surgery and radiotherapy offers these patients the best chance for disease control.
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Sinonasal adenoid cystic carcinoma
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Adenoid cystic carcinoma of the sinonasal tract is a rare cancer that accounts for 10% of all malignancies at this site. The objective of the current study was to evaluate prognostic factors, treatment outcomes, recurrence patterns, and survival rates for sinonasal adenoid cystic carcinoma. A retrospective chart review was performed at an academic tertiary referral center. Between 1990 and 2004, 105 patients were evaluated for adenoid cystic carcinoma of the sinonasal tract at a single institution. Demographics, presentation, anatomic site, Tumor, Lymph Node, Metastasis (TNM) classification, pathology, treatment, recurrences, and survival were evaluated. One hundred five patients with adenoid cystic carcinoma were evaluated, including 58 women and 47 men. Their median age was 50 years, and the mean follow-up was 47 months. The maxillary sinus (47%) and the nasal cavity (30%) were the most common primary tumor sites. The majority of patients presented with T3/T4 (76.7%), N0 (98%), M0 (97%) disease. Eighty-four percent of patients underwent surgery and received postoperative radiation as treatment for their primary disease. The local recurrence rate was 30%, and the distant metastases rate was 38%. The 5-year overall survival and disease specific survival rates were 62.9% and 70.9%, respectively. Adenoid cystic carcinoma of the paranasal sinuses is a rare disease, and the ideal treatment paradigm has yet to be defined. The current data suggested that surgical resection with postoperative radiation therapy offers durable local control and compares favorably with historic data. Although local recurrences develop in a significant percentage of patients, survival from this disease exceeds that of other sinonasal malignancies. Cancer 2007.
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Nasal and paranasal sinus carcinoma: Are we making progress?
Article
BACKGROUND The authors reviewed treatment results in patients with nasal and paranasal sinus carcinoma from a large retrospective cohort and conducted a systematic literature review.METHODS Two hundred twenty patients who were treated between 1975 and 1994 with a minimum follow-up of 4 years were reviewed retrospectively. A systematic review of published articles on patients with malignancies of the nasal and paranasal sinuses during the preceding 40 years was performed.RESULTSThe 5-year survival rate was 40%, and the local control rate was 59%. The 5-year actuarial survival rate was 63%, and the local control rate was 57%. Factors that were associated statistically with a worse prognosis, with results expressed as 5-year actuarial specific survival rates, included the following: 1) histology, with rates of 79% for patients with glandular carcinoma, 78% for patients with adenocarcinoma, 60% for patients with squamous cell carcinoma, and 40% for patients with undifferentiated carcinoma; 2) T classification, with rates of 91%, 64%, 72%, and 49% for patients with T1, T2, T3, and T4 tumors, respectively; 3) localization, with rates of 77% for patients with tumors of the nasal cavity, 62% for patients with tumors of the maxillary sinus, and 48% for patients with tumors of the ethmoid sinus; 4) treatment, with rates of 79% for patients who underwent surgery alone, 66% for patients who were treated with a combination of surgery and radiation, and 57% for patients who were treated exclusively with radiotherapy. Local extension factors that were associated with a worse prognosis included extension to the pterygomaxillary fossa, extension to the frontal and sphenoid sinuses, the erosion of the cribriform plate, and invasion of the dura. In the presence of an intraorbital invasion, enucleation was associated with better survival. In multivariate analysis, tumor histology, extension to the pterygomaxillary fossa, and invasion of the dura remained significant. Systematic review data demonstrated a progressive improvement of results for patients with squamous cell and glandular carcinoma, maxillary and ethmoid sinus primary tumors, and most treatment modalities.CONCLUSIONS Progress in outcome for patients with nasal and paranasal carcinoma has been made during the last 40 years. These data may be used to make baseline comparisons for evaluating newer treatment strategies. Cancer 2001;92:3012–29. © 2001 American Cancer Society.
View
Adenoid Cystic Carcinomas Arising in Salivary Glands. A Correlation of Histologic Features and Clinical Course
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62 cases of adenoid cystic carcinoma (ACC) arising in major and minor salivary glands were studied. Factors which appeared to influence the clinical course included: 1) histologic pattern, 2) presence or absence of tumor on the surgical lines of excision, 3) site, 4) size of primary lesion, 5) presence or absence of tumor in lymph nodes, and 6) degree of cellular atypia. On histologic examination, these neoplasms were classified according to their predominant histologic pattern (tubular, cribriform or solid). Recurrences have been seen in 59% of patients with ACC demonstrating a predominantly “tubular” pattern, as compared to 89% for the “cribriform” lesions and 100% for the “solid” neoplasms. Of patients who eventually died of tumor, those having “tubular” predominant lesions had the longest course (average 9 years before death, in contrast to 8 years for the “cribriform” and 5 years for the “solid” tumors). Our findings suggest that the “tubular” predominant pattern has the best prognosis and represents the best differentiated histologic form of ACC. In contrast, the “solid” pattern is the least differentiated and is associated with the worst prognosis. The predominantly “cribriform” lesions appear to lie between the other two forms both clinically and histologically.
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[Rare tumors of the septum. 8 reported cases (author's transl)]
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  • Sep 1979
The authors present 8 cases of rare tumors of the nasal septum and define a number of characteristic points on the basis of each of them. The following are presented: --a malignant nevus, the rarety of E.N.T. sites of such tumors is emphasized, as well as important role of immunotherapy in their treatment, alongside surgery and radio-therapy. --a keratoacanthoma, difficult in terms of differential diagnosis from a basal cell carcinoma. -- a cavernous hemangioma, excision of which must be wide if recurrence is to be avoided. --one very hemorrhagic mixed tumor. --one case of pseudo tumoral tuberculosis in a woman with no known past history of tuberculous disease. --one very painful glomus tumor, radical treatment of which is surgical, in preference to radiotherapy. --two cylindromas of the septum, one taking the form of a large, angiomatous polyp, which puts in question the name of cystic adenoid carcinoma, suggested for these non-cutaneous tumors.
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[A case report of adenoid cystic carcinoma of the nasal septum]
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  • May 1992
A 50-year-old woman with adenoid cystic carcinoma of the nasal septum is presented. Her chief complaint was recurrent epistaxis. Surgery was originally performed to remove a nasal polyp, but during resection, the surgeon discovered that the mass had arisen from the nasal septum, so only a biopsy was done. The pathological report was adenoid cystic carcinoma, so she was referred and admitted to our hospital. A flat 1.5 x 1.5 cm tumor was recognized on the left side of the nasal septum. Nasal septectomy was performed. The tumor was dissected with nasal septal cartilage, and the defect was covered with a free skin flap from the thigh. The incidence of malignant tumors from the nasal septum is very low, composing from 2.7% to 8.4% of nasal and paranasal malignant tumors. Therefore the incidence of adenoid cystic carcinoma in particular is very low.
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Distant metastases in adenoid cystic carcinoma of salivary origin
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  • Nov 1997
  • AM J SURG
Adenoid cystic carcinoma (ACC) is an aggressive, often indolent tumor, with a high incidence of distant metastasis (DM). Relatively little has been written about the factors that influence distant spread and subsequent survival because it is uncommon and more than a decade of observation may be required to appreciate the prolonged clinical course in some patients. We have retrospectively studied 196 determinate patients who received definitive treatment in our hospital between 1939 and 1986 for ACC in all salivary sites. Inclusion criteria were no prior treatment elsewhere other than excisional biopsy and eligibility for follow-up of at least 10 years. Variables assessed for their impact on distant metastasis included age, gender, site, size, node status, stage, grade, and locoregional treatment failure. Treatment failure occurred in a total of 122 of 196 determinate patients (62%), 74 of whom had DM (38%). This was usually associated with locoregional recurrence (51 patients), but DM was the only indication of failure in 23 whose primary tumor was controlled. Of the 74 patients with known DM, the lung was recorded as the only involved site in 50 patients, lung was involved in addition to other sites in 17, bone metastases alone occured in 5, and the remaining 2 developed disseminated disease. Disease-free intervals varied from 1 month to 19 years (median 36 months) and exceeded 10 years in 9 of 113 patients (8%) with adequate information about treatment failure. Survival with DM was less than 3 years in 54%, but more than 10 yrs in 10% (maximum 16 years). The only significant factors influencing survival were the size of the primary tumor (P <0.0000), local or neck recurrence (P = 0.0006), and the presence of nodal involvement (P = 0.02). The high incidence of DM with locoregional failure confirms the importance of aggressive initial surgery, combined with irradiation, for high-stage tumors or involved surgical margins. Large tumor size and lymph node involvement, rather than microscopic appearance, were predictive of DM. Considering that lung metastases are usually multiple, and prolonged survival without treatment is not unusual, resection of pulmonary metastases may be hard to justify in ACC patients based on the limited experience thus far reported. Chemotherapy for metastatic ACC is probably best withheld until symptoms appear.
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Adenoid Cystic Carcinoma of the Sinonasal Tract: Treatment Results
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  • Jul 2006
Malignancies arising from the sinonasal tract, which includes the nose, paranasal sinuses, and nasopharynx, are uncommon. Although adenoid cystic carcinoma (ACC) is the second most common cancer occurring in the sinonasal tract, only few studies have been reported. This retrospective review was performed to identify the clinical features and treatment outcomes of sinonasal ACC. Thirty-five patients diagnosed and treated for ACC of the sinonasal tract were included in this study. Medical records, radiographs, and pathologic slides were retrospectively reviewed. In two thirds of the patients, the maxillary sinus was the site of origin and cribriform was the most common histologic subtype (61%). Seventy-one percent of the patients had advanced disease (T3, T4) at the time of diagnosis. Five-year overall survival rate was 86% and treatment failure occurred in 18 patients (51%). Five-year local recurrence rate and distant metastasis rate were 30% and 25%, respectively. Adjunctive radiotherapy appeared to reduce local recurrence. Presence of distant metastasis correlated with decreased 5-year survival (P = .001). Five-year survival rate after development of distant metastasis or local recurrence were 17% and 58%, respectively. Based on our findings, we suggest that sinonasal ACC be treated by a combined modality of radical surgery followed by postoperative radiation. The prognosis of sinonasal ACC seems to be determined by the presence of distant metastasis.
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