Rare case of metastatic adenocarcinoma to the maxillary sinus

  1. Apurwa Prasad 1,
  2. Taha Alrifai 2,
  3. Sumathi Vijaya Rangan 1 and
  4. Jessica Garcia 3
  1. 1 Internal Medicine, AMITA Health Saint Joseph Hospital, Chicago, Illinois, USA
  2. 2 Department of Hematology/Oncology/Cell Therapy, Rush University Medical Center, Chicago, Illinois, USA
  3. 3 Department of Hematology/Oncology, AMITA Health Saint Joseph Hospital, Chicago, Illinois, USA
  1. Correspondence to Dr Sumathi Vijaya Rangan; dr.v.sumathi@gmail.com

Publication history

Accepted:02 Sep 2021
First published:17 Sep 2021
Online issue publication:17 Sep 2021

Case reports

Case reports are not necessarily evidence-based in the same way that the other content on BMJ Best Practice is. They should not be relied on to guide clinical practice. Please check the date of publication.

Abstract

Colorectal carcinoma metastases to the head and neck are exceedingly rare. Patients may present with vague symptoms that may lead to a delay in diagnosis. We report the case of a 51-year-old man with a known history of stage IIIB colorectal adenocarcinoma who presented with right-sided molar tooth bleeding and right-sided palate swelling that led to difficulty speaking, eating and weight-loss of 15 pounds. Imaging studies revealed a 3.1×4.8×3 cm mass that was centred around the posterior aspect of the maxilla. Pathology revealed moderately differentiated intestinal type adenocarcinoma of colonic subtype, immune histochemistry was positive for CDX2, CK20 and MUC2, thus confirming metastatic disease to the maxilla. The patient is undergoing chemoradiation therapy for palliation of his symptoms. Clinicians should be aware of this potentional site of metastatic disease and suspect the diagnosis in a timely manner to avoid delays in making a diagnosis.

Background

Colorectal adenocarcinoma (AC) is the third most common cancer to affect women and men, and the third most common cause for cancer-related mortality in women and in men.1 Metastatic disease is considered to be a major cause of death in patients with colorectal cancer, the most common site for metastasis is the liver, other sites include the lungs, peritoneum, bone and the central nervous system.2–4 Metastasis to the paranasal sinuses are extremely rare. Herein, we highlight a case of metastatic colorectal AC involving the maxillary paranasal sinus in a 51-year-old man. Clinicians should be aware of this potential atypical presentation of metastatic disease. A high clinical index of suspicion is important in order to avoid delays in making a diagnosis.

Case presentation

A 51-year-old Asian man presented to the clinic with bleeding from a right maxillary molar. This bleeding had occurred intermittently for 1 month, mainly when he would floss. The patient sought medical attention and was initially treated with antibiotics for a possible dental abscess. However, he reported a hard swelling in his right palate associated with difficulty speaking and eating followed by a 15-pound weight loss. He denied any pain, redness, discharges, facial numbness, paralysis of facial muscles or trauma to the area. He denied any dental extraction or any procedures to the affected area.

Oral examination yielded a compressible cystic mass with overlying hypervascularity posterior to right third molar along with submucosal fullness of the right hard palate and hyperpigmentation. Additionally, a mildly prominent 1.2 cm right cervical lymph node at level IIa was noted.

The patient had a medical history significant for stage IIIB (T3N1M0) colorectal cancer which was treated with laparoscopic low anterior resection and laparoscopic splenic flexure mobilisation followed by 12 cycles of adjuvant chemotherapy with oxaliplatin, fluorouracil and leucovorin.

Following the completion of adjuvant chemotherapy, the patient’s disease was being monitored by serial carcinoembryonic antigen (CEA) level measurements, which were noted to have an upward trend from 3.3 ng/mL to 96.7 ng/mL prior to his current presentation. Due to the elevated CEA levels, disease recurrence was suspected and CT of the chest, abdomen and pelvis, and whole body positron emission tomography/CT (PET/CT) was performed 2 months prior to the current presentation. However, imaging at that time did not show any evidence of disease recurrence.

During the current presentation, the patient underwent further investigations to evaluate his presentation, including evaluation for disease recurrence.

Investigations

Laboratory investigations at the time of presentation were within normal limits except for an elevated CEA level of 94. 6 ng/mL. This value was close to the CEA levels 2 months earlier, at which time a CT of the chest, abdomen and pelvis, and whole body PET/CT was performed. Imaging at that time did not show any recurrence of disease.

During this presentation, an MRI of the face, neck and mandible with and without intravenous contrast was done and it revealed a large, heterogeneously enhancing mass that measured approximately 3.1×4.8×3 cm and was centred in the posterior aspect of the right maxilla surrounding the roots of maxillary first and second molars with involvement of the hard palate, right greater and lesser palatine foramina, right pterygoid plates and right medial pterygoid muscle. The mass also extended into the right maxillary sinus, right retromolar trigone, right retromaxillary fat and right inferior nasal cavity mucosa. The mass diffusely abutted the anterior aspect of the soft palate.

The patient underwent a biopsy of the mass with removal of the affected tooth. Biopsy results confirmed moderately differentiated intestinal type AC (ITAC) of colonic subtype, staining was positive for CDX2, CK20 and MUC2 (focal) in the tumour cells.

Follow-up imaging using whole body PET/CT in anticipation of palliative radiotherapy now demonstrated the presence of a large, markedly hypermetabolic, mass within the skull base at the floor of the right maxillary sinus extending through to the hard palate (figure 1). In addition, the PET/CT scan showed a hypermetabolic lymph node anterior to the sternocleidomastoid muscle, several mildly hypermetabolic, increasing subcentimetre lung nodules suspicious for lung metastases, and a new small mildly hypermetabolic retroperitoneal lymph node within the upper abdomen to the left of the abdominal aorta.

Figure 1

PET/CT scan showing a large mass within the skull base at the floor of the right maxillary sinus extending through to the hard palate. PET, positron emission tomography.

Differential diagnosis

Due to the patient’s chief complaint of right molar with associated pain, our differential diagnosis initially included trauma, infection (including abscess formation) and primary head and neck malignancies. The patient denied a history of antecedent trauma. He was empirically treated with antibiotics without any significant improvement in his symptoms. The concern for a malignant aetiology became stronger with the patient’s subsequent complaints of feeling a hard mass in the area, and developing a difficulty with speaking, eating and ensuant weight loss. Metastatic colon cancer was suspected given the patient’s known history, as well as the elevation of the CEA level. This was further investigated as detailed above, with final pathology confirming a diagnosis of moderately differentiated ITAC of colonic subtype, with immunohistochemical staining favouring a colon primary.

Treatment

The patient developed severe pain in the right palate and teeth, which made solid food intolerable, and subsequently, he experienced a weight loss of 20 pounds. This led to the placement of a percutaneous endoscopic gastrostomy tube in order to maintain adequate nutrition. Palliative radiotherapy with the addition of capecitabine 825 mg/m2 two times per day, on the days of radiation therapy, was initiated for palliation of his symptoms.

Outcome and follow-up

After 6 months of treatment, there was evidence of disease progression with new liver metastases. The patient was then started on systemic therapy with leucovorin (folinic acid), fluorouracil, irinotecan (FOLFIRI) with bevacizumab. A PET/CT scan was repeated nearly 12 months after the onset of symptoms, and showed disease response. In addition, the CEA level had decreased to 3.9 ng/mL. The patient currently continues to receive systemic therapy with FOLFIRI and bevacizumab and is tolerating treatment well.

Discussion

Sinonasal carcinomas (SNCs) are rare with an estimated worldwide incidence of 1 case per 100 000 persons. Squamous cell carcinoma and AC account for 80% of SNCs.5

Metastases to the paranasal sinuses are exceedingly rare, the most common carcinoma to metastasise to the paranasal sinuses is renal cell carcinoma, other reported primary sites include the lungs, breast, colon, among others.6–11

The most commonly involved paranasal sinuses include the maxillary (50%), ethmoid (19%), frontal (16%), nasal cavity (10%) and sphenoid sinus (5%).11 12 Some of the presenting symptoms reported by Bernstein et al in patients with metastatic tumours to the maxilla, nose and paranasal sinuses include swelling of the cheek, blindness, nasal obstruction, epistaxis, infraorbital pain, exophthalmos and orbital cellulitis. This study highlighted the lack of specific presenting symptoms that would distinguish metastatic tumours to the paranasal sinuses from primary tumours. Hence, clinicians must be aware of this rare metastatic site, and suspect metastases when patients with a history of malignancy presents with the aforementioned signs and symptoms.

To the best of our knowledge, there have been eight cases reported of colorectal carcinoma metastasizing to the paranasal sinuses (table 1).

Table 1

Reported cases of colorectal carcinoma metastasizing to the paranasal sinuses.

Author Age/gender Primary tumour and stage Presenting symptoms Location of metastatic disease Immunohistochemical
Staining findings/histology
CDX2, caudal type homeobox 2; CEA, carcinoembryonic antigen; CK, cytokeratin; N/A, not available; NSE, neuron specific enolase; SIMA, small intestine mucin antigen.
Bin Sabir Husin Athar11 52/female Colon
N/A
Swelling and numbness over the right cheek, right nasal obstruction, epistaxis, anosmia, loosening of upper teeth, headache, weight loss Right maxillary sinus with extension to the ethmoid, frontal, sphenoidal sinuses Positive: CK20,CEA, P53
Negative: CK7, NSE
Akyol18 62/male Colon pT3N1M0 Nodular skin metastatic lesions of the lips, nape, face—swelling of the maxillary area of the face Anterior wall of the maxillary sinus with orbital invasion Positive: CK20
Negative: CK7
Cama19 57/female Colon
IV
Painful slowly increasing swelling of the right alveolar ridge, right otalgia, dysphagia, painful mastication Right alveolar ridge and the posterior wall of the right maxillary sinus, infra temporal fossa and pterygoid muscles Positive, marked: CK20, CEA, CAM 5.2, SIMA, Ber-EP4
Positive, moderate: AE1/AE3, CK20 (fraction of the cells).
Negative: CK7, S-100 protein, vimentin Leu7, synaptophysin, chromogranin
Conill20 77/female Rectum
T3N0
Oedema of the right superior eyelid with proptosis Nasal and paranasal sinuses with extension into right orbit Positive: CK20, p53
Negative: CK7
Hwang21
Patient 1
46/female Colon pT4N0 Headache, vomiting Nasal mass, brain metastases Positive: CDX2, CK20, Vilin
Negative: CK7
Hwang21
Patient 2
64/male Colon pT3N2b Epistaxis Central mass on anterior and posterior nasoendoscopy Positive: Broad spectrum CK (AE1/3), CK20, CDX2
Negative: CK7, P63
Somali22 47/female Colon
IV
Toothache and upper lip numbness Mass involving the sphenoid sinus, left maxillary sinus, left pterygopalatine plate, pterygopalatine, and infra temporal fossa Metastatic adenocarcinoma
Tanaka23 72/female Rectum
N/A
Vision loss in the right eye Posterior ethmoid sinus, sphenoid sinus, optic nerve N/A

Ethics statements

Patient consent for publication

Footnotes

  • Contributors AP and TA contributed equally to this paper. JG and SVR were involved in reviewing of the manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

References

Use of this content is subject to our disclaimer