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. 1 a ). 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. 1 b). 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.

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.

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.

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

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.

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, 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.

Table 1

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

Study	Case #	Age (years)	Gender	Symptoms	Primary Site	Tobacco	Alcohol	Neck disease	Distant Metastasis	TNM	Stage	Surgery	Radiation	Chemotherapy	Clinical Outcome
Wieneke et al., 1999	1	79	F	Sinusitis, headache	L sinuses	Yes	No	No	Bone, lung			Yes	No	No	Died of disease at 1 year
	2	75	F	Nasal obstruction	L sinuses			No	None			Yes	No	No	Recurrence in 2 years
	3	33	F	Nasal obstruction, diplopia	B sinuses			No	Bone, lung			Yes	Yes	Yes	Died of disease at 1 year
Tulunary et al., 2002	4	43	M	Infraorbital swelling, epistaxis	L maxillary sinus							Yes			Unknown
Oikawa et al., 2007	5	78	M	Cheek swelling and pain	R maxillary sinus	Yes	Yes	No	None	T3N0M0	3	Yes	Yes	Yes	Disease free 25 months later
	6	60	M	Cheek pain, diplopia	L maxillary sinus	Yes	Yes	No	Orbit, skull base, lung	T4bN0M0	4	No	Yes	No	Died of disease at 6 months
Ozgursoy et al., 2008	7	28			Maxillary sinus				Scalp, pancreas, kidney, adrenal gland, ovaries, lung, bone marrow						Died of disease at 4 months
Yu et al., 2008	8	59	M		Maxillary sinus						4				Died of disease at 1 year
	9	47	M		Maxillary sinus						4				Died of disease at 1 year
	10	69	M		Maxillary sinus						4				Died of disease at 2.5 years
	11	48	M		Maxillary sinus						4				Disease free 3.5 years later
Stanciulescu et al., 2012	12	51	M	Cheek swelling, nasal obstruction	L maxillary sinus	No	No	No	None	T4aN0M0	4	Yes	Yes	No	Unknown
Ishida and Okabe, 2013	13	85	F	Exophthalmos	R maxillary sinus			No	None	T4bN0M0	4	No	Yes	No	Alive with disease 10 months later
	14	60	M	Nasal obstruction	R maxillary sinus			No	Dura, liver, lung	T4bN0M0	4	Yes	No	Yes	Died of disease at 1.5 years
UCLA	15	77	M	Cheek swelling	L maxillary sinus	Yes	Yes	Yes	Lung	T4aN2bM1	4	Yes	No	No	Died of disease within 1 year
	16	68	M	Cheek pain	L maxillary sinus	Yes	Yes	No	None	T2N0M0	2	Yes	Yes	No	Disease free 2 years later
	17	46	M	Cheek pain	R maxillary sinus	No	No	No	None	T2N0M0	2	Yes	No	No	Disease free 6 months later
	18	55	M	Palate mass	L hard palate	No	Yes	Yes	None	T3N2aM0	4	Yes	Yes	Yes	Disease free 6 months later
	19 a	32	M	Loose teeth	L maxillary sinus	No	No	No	None	T4aN0M0	4	Yes	Yes	No	Disease free 3 months later

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