Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review

Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review

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Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review☆,☆☆ Edward C. Kuan, MD, MBA a , Kevin A. Peng, MD a , Sunita Bhuta, MD b, e , Miguel Fernando Palma Diaz, MD b, e , Zuo-Feng Zhang, MD, PhD c, d , Elliot Abemayor, MD, PhD a, d, e , Maie A. St. John, MD, PhD a, d, e,⁎ a

Department of Head and Neck Surgery, University of California, Los Angeles (UCLA) Medical Center, Los Angeles, CA Department of Pathology and Laboratory Medicine, UCLA Medical Center, Los Angeles, CA c Department of Epidemiology, UCLA School of Public Health, Los Angeles, CA d Jonsson Comprehensive Cancer Center, UCLA Medical Center, Los Angeles, CA e UCLA Head and Neck Cancer Program, UCLA Medical Center, Los Angeles, CA b

ARTI CLE I NFO

A BS TRACT

Article history:

Purpose: Basaloid squamous cell carcinoma (BSCC) is a rare variant of squamous cell

Received 5 January 2015

carcinoma characterized by a highly aggressive clinical course. Though typically found in the larynx, oropharynx, and hypopharynx, we report a rare case of BSCC originating in the maxillary sinus in an otherwise healthy 32-year-old male. Materials and methods: Single case report of a patient with BSCC of the maxillary sinus and retrospective chart review of all cases of BSCC of the maxilla at a single academic institution between January 1, 1986 and December 31, 2013. The MEDLINE database was additionally queried for all case series or reports of BSCC arising in the maxilla, and pertinent clinical data were extracted. Results: The clinical presentation, disease course, and management of a patient with BSCC of the maxilla are presented. In this recent case, the patient presented with persistent alveolar pain and a nonhealing tooth infection. Radiographic studies demonstrated a large necrotic mass in the left maxillary sinus that was biopsy-proven as BSCC. The patient underwent surgical resection followed by postoperative radiation without complications. Conclusions: BSCC of the maxilla is a rare oncologic entity that may progress to late disease stage without obvious clinical signs or symptoms. Optimal treatment involves complete surgical resection followed by postoperative. © 2015 Elsevier Inc. All rights reserved.

1.

Introduction

Basaloid squamous cell carcinoma (BSCC) is a rare, high-grade, aggressive variant of squamous cell carcinoma, most commonly

found in the base of tongue, larynx (supraglottis), and hypopharynx (piriform sinus). First described in the head and neck by Wain in 1986 [1], BSCC has a propensity for regional and distant metastasis, with rates of 64% and 44% traditionally reported in



The authors report no conflicts of interest. This work was presented as a poster at the 2014 American Head and Neck Society/International Federation of Head and Neck Oncologic Societies World Congress Meeting on July 26–30, 2014, in New York City, NY. ⁎ Corresponding author at: UCLA Department of Head and Neck Surgery, 10833 Le Conte Ave, 62-132 CHS, Los Angeles, CA 90095. Tel.: + 1 310 206 6688; fax: + 1 310 825 2810. E-mail address: [email protected] (M.A. St. John). ☆☆

http://dx.doi.org/10.1016/j.amjoto.2015.01.015 0196-0709/© 2015 Elsevier Inc. All rights reserved.

Please cite this article as: Kuan EC, et al, Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.01.015

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the literature [2]. To this day, reports of BSCC occurring in atypical primary sites, such as the maxilla, have been limited to case series or individual reports, with the largest dedicated series limited to 14 patients, of which only three patients had primary involvement of the maxilla (i.e., maxillary sinus). We report the case of a young, healthy male patient presenting with pain over the maxillary teeth and tooth loosening, later found to have a BSCC of the maxillary sinus. We additionally review all cases of BSCC of the maxilla at a single academic institution and search the literature for additional cases of BSCC originating in the maxilla and aim to determine patient factors and outcomes common to these cases.

2.

Materials and methods

This study has been approved by the Institutional Review Board of the University of California, Los Angeles (UCLA).

2.1.

Case report

A 32-year-old healthy male first noticed progressively worsening pain and swelling over his left posterior maxillary region and hard palate. He visited his general dentist who extracted tooth #14 and, at one-week follow-up, noted poor wound healing with persistent pain over the extraction site. He was thus referred to see an oral surgeon who treated him on antibiotics without improvement. The oral surgeon then performed a biopsy of the nonhealing extraction site. When referred to a tertiary academic medical center, the patient was noted to have a 3 cm mass overlying the left hard palate and alveolar ridge. The previously described tooth extraction site had not healed, though no frank fistula was noted. A computed tomography (CT) scan of the face and neck demonstrated a 3.6 × 3.6 × 3.0 cm necrotic mass destroying the left maxillary sinus and extending into the nasal cavity and retroantral region without obvious extension to the skull base (Fig. 1a). Magnetic resonance imaging (MRI) of the neck, face, and orbits confirmed the infiltrative nature of the mass,

broaching the left posterior maxillary wall, involving the infratemporal fossa and encroaching upon the pterygoid plates (Fig. 1b). There was also extension through the hard palate inferiorly. A positron emission tomography (PET) scan revealed avid uptake in the left maxillary sinus but no indication of nodal or distant metastases. Given these findings, the tumor was staged as T4aN0M0. The patient was subsequently presented at a multidisciplinary tumor board conference, where the histology of the patient’s biopsy slides was reviewed. On low power, maxillary bone fragments were surrounded by an infiltrative collection of basaloid cells (Fig. 2). When viewed at high power, abrupt keratinization was noted within islands of basaloid cells characterized by multifocal, marked pleomorphism (Fig. 3). These findings suggested the diagnosis of BSCC. The patient subsequently underwent left subtotal maxillectomy with preservation of the orbital floor. The defect was lined with a split-thickness skin graft, and an immediate surgical obturator was placed. The patient tolerated the procedure well, and pathological review demonstrated negative margins. The tumor specimen measured 4.3 × 4.0 × 3.0 cm and demonstrated no perineural or lymphovascular invasion, though significant bony invasion was found. The patient was subsequently referred to radiation oncology for adjuvant radiation therapy, which he completed without complication.

2.2.

Literature review and data extraction

The MEDLINE database was searched from 1950 to March 1, 2014. The search strategy aimed to identify all reported cases of BSCC of the maxilla. The studies, of which many were case series or case reports, were reviewed and individual case information extracted into a standardized table (Table 1). Duplicate case information was not included. In addition, a query for all documented cases of BSCC of the maxilla between January 1, 1986 and December 31, 2013 was submitted to the UCLA Jonsson Comprehensive Cancer Center Tumor Registry. Specifically, information on age, gender, presenting symptoms,

Fig. 1 – T1-weighted, post-gadolinium MRI of the face (left) and CT of the face (right) demonstrating a large, infiltrative, poorly defined mass within the left maxillary sinus. Please cite this article as: Kuan EC, et al, Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.01.015

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primary sites. Just over half (56%, 5/9) of the patients had a known smoking history, and the same proportion had known alcohol use. Neck nodal disease was noted in only 2 (15%, 2/13) patients, while distant metastases occurred in 6 (43%, 6/14) patients. All cases of distant metastasis invariably involved the lung. Most tumors at presentation were stage IV (79%, 11/14). A majority of patients were treated with surgical resection (86%, 12/14). Only 2 (14%, 2/14) patients were not treated with surgical resection, while 8 (62%, 8/13) patients underwent radiation therapy and, of those, 4 (31%, 4/13) patients also received chemotherapy. The overall prognosis remained poor, with 7 (44%, 7/16) patients dying of the disease within one year.

4. Fig. 2 – Low power view of tumor sample demonstrating bony infiltration by basaloid cells (bone fragment highlighted by white arrow).

primary site, tobacco and alcohol use, neck involvement, distant metastases, TNM staging, therapeutic interventions (surgery, radiation therapy, chemotherapy), and clinical outcome were collected when available.

3.

Results

A total of 14 articles were found in the MEDLINE search. Fulltext review of all 14 articles indicated that 7 articles contained non-overlapping patient case information and were thus included in the analysis [3–8]. This collection yielded a total of 14 reported cases of BSCC of the maxilla in the literature. The UCLA Tumor Registry query returned 4 additional cases of BSCC of the maxilla. Combined with the case presented in this article, a total of 19 cases of BSCC of the maxilla were identified (Table 1). Among the included cases, the median age was 59 (range 28–85). Fourteen of the 18 patients (78%) were male. The maxillary sinus accounted for the vast majority (95%, 18/19) of

Discussion

BSCC of the head and neck is a rare oncologic entity, with just over 1000 cases reported in the Surveillance, Epidemiology, and End Results (SEER) database between 2000 and 2008 [9]. An extensive review of the western literature revealed only an additional 14 cases of maxilla BSCC primaries [3–8], underscoring the distinct rarity of this disease. With the addition of 5 cases from our institution, the current study is, to date, the largest series to report on the characteristics of BSCC of the maxilla. As has been the case with all analyses conducted on BSCC, the current study is limited by the small sample size, precluding meaningful analysis beyond descriptive statistics. Overall, the case series in this current study corroborates traditional teachings regarding BSCC. BSCC has been reported more commonly in elderly men with a history of tobacco and/or alcohol use [2,10]. Distant metastases occurred, on average, more frequently than conventional squamous cell carcinoma (SCC) [9], with the lung being the most frequently involved site [2]. The most common presenting symptoms are subtle at best — nasal obstruction, cheek pain, and cheek swelling. In our patient case, the patient presented with repeated dental infections in the context of loosening maxillary teeth. He pursued dental care appropriately, and his underlying malignancy was uncovered only following tooth extraction. For this reason, most tumors in this region, regardless of histology, present at an advanced stage (e.g., only three patients in our

Fig. 3 – High power view of tumor sample shows abrupt keratinization with peripheral palisading (left, white arrow). Higher power view demonstrates multifocally pleomorphic basaloid cells (right, white arrow), confirming the diagnosis of basaloid squamous cell carcinoma. Please cite this article as: Kuan EC, et al, Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.01.015

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Study

Case Age Gender Symptoms # (years)

Primary Tobacco Alcohol Neck Distant Metastasis Site disease

Wieneke et al., 1999

1

79

F

Sinusitis, headache

L sinuses Yes

2

75

F

3

33

F

Tulunary et al., 2002

4

43

M

Oikawa et al., 2007

5

78

M

Nasal obstruction Nasal obstruction, diplopia Infraorbital swelling, epistaxis Cheek swelling and pain

6

60

M

Ozgursoy et al., 2008

7

28

Yu et al., 2008

8

59

M

Maxillary sinus

4

9

47

M

Maxillary sinus

4

10

69

M

Maxillary sinus

4

11

48

M

Maxillary sinus

4

Cheek pain, diplopia

Stage Surgery Radiation Chemotherapy Clinical Outcome

No

Bone, lung

Yes

No

No

L sinuses

No

None

Yes

No

No

B sinuses

No

Bone, lung

Yes

Yes

Yes

L maxillary sinus R Yes maxillary sinus L Yes maxillary sinus Maxillary sinus

No

TNM

Yes

Yes

No

None

T3N0M0

3

Yes

Yes

Yes

Yes

No

Orbit, skull base, lung

T4bN0M0

4

No

Yes

No

Scalp, pancreas, kidney, adrenal gland, ovaries, lung, bone marrow

Died of disease at 1 year Recurrence in 2 years Died of disease at 1 year Unknown

Disease free 25 months later Died of disease at 6 months Died of disease at 4 months Died of disease at 1 year Died of disease at 1 year Died of disease at 2.5 years Disease free 3.5 years later

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Please cite this article as: Kuan EC, et al, Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.01.015

Table 1 – Documented cases of basaloid squamous cell carcinoma of the maxilla in the literature and from the UCLA Tumor Registry.

51

M

Ishida and 13 Okabe, 2013

85

F

14

60

M

Nasal obstruction

15

77

M

Cheek swelling

16

68

M

Cheek pain

17

46

M

18

55

19 a

32

UCLA

No

None

T4aN0M0

4

Yes

Yes

No

Unknown

R maxillary sinus

No

None

T4bN0M0

4

No

Yes

No

R maxillary sinus L Yes maxillary sinus

No

Dura, liver, lung

T4bN0M0

4

Yes

No

Yes

Yes

Yes

Lung

T4aN2bM1 4

Yes

No

No

L Yes maxillary sinus

Yes

No

None

T2N0M0

2

Yes

Yes

No

Cheek pain

R No maxillary sinus

No

No

None

T2N0M0

2

Yes

No

No

M

Palate mass

L hard palate

Yes

Yes

None

T3N2aM0

4

Yes

Yes

Yes

M

Loose teeth

L No maxillary sinus

No

No

None

T4aN0M0

4

Yes

Yes

No

Alive with disease 10 months later Died of disease at 1.5 years Died of disease within 1 year Disease free 2 years later Disease free 6 months later Disease free 6 months later Disease free 3 months later

Left, L; Right, R; Bilateral, B. The patient reported in the case.

a

Cheek swelling, nasal obstruction Exophthalmos

L No maxillary sinus

No

No

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Please cite this article as: Kuan EC, et al, Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.01.015

Stanciulescu 12 et al., 2012

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series were not stage IV). For otolaryngologists, this signifies the importance of recognizing loosening teeth, dental infections, and nonhealing gingival wounds as a potentially ominous clinical sign. In our series, the prevalence of neck nodal metastases (15%) appears to be lower in maxilla primaries when compared to the head and neck as a whole (>60%) [2]. This finding is not dissimilar to the relatively low prevalence of nodal metastases in conventional SCC of the maxillary sinus (10%) [11]. For this reason, in the case presented, no neck dissection was performed given the predicted low yield at the cost of increased surgical morbidity. Of the four other cases within the UCLA Tumor Registry, three other patients underwent treatment of the neck, with one resulting in 4/18 positive nodes following neck dissection, another with 0/6 positive nodes following neck dissection, and the last (hard palate primary) treated with radiation. Thus, as in the case with conventional SCC of the maxilla, the role of elective neck dissection in the N0 neck remains unclear, but should be considered for advanced stage tumors. It is commonly thought that BSCC of the head and neck carries a poorer prognosis compared to conventional SCC, likely attributable to its tendency for nodal and distant metastasis [2]. In contrast, when tumor stage and site are matched, the clinical outcomes (i.e., disease-specific survival rates) are, in fact, comparable [9,10,12]. A recent study by Fritsch and Lentsch specifically quoted similar disease-specific survival rates for sinonasal primaries [9]. This is not unexpected, as both conventional SCC and BSCC of the maxilla tend to present at a locally advanced stage given the absence of obvious or early symptoms. Multimodality therapy, most commonly surgery followed by radiation therapy, maximizes the chance of cure. In the current series, though follow up periods are rather heterogenous across different sources, nearly half of the reported cases succumbed to the disease within 1 year of initial diagnosis, despite aggressive multimodality therapy. Early detection and timely treatment of these tumors remain a challenge, especially given the rarity of the disease.

5.

Conclusion

BSCC of the maxilla, though exceedingly rare amongst head and neck cancers, remains a difficult disease to treat. Traditionally regarded as more aggressive than SCC, management principles are actually similar between the two entities. The mainstay of

therapy is radical resection (with consideration of elective neck dissection in advanced tumors) followed by adjuvant radiation therapy (and possibly chemotherapy if high-risk features are present), but prognosis nevertheless remains guarded, with oneyear survival rates just over 50%. Advances in early detection of maxilla primaries will certainly have a significant impact on clinical outcomes.

REFERENCES

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Please cite this article as: Kuan EC, et al, Basaloid squamous cell carcinoma of the maxilla: Report of a case and literature review, Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.01.015