Dorsal rhinotomy in a dog with a chondro-osseous respiratory epithelial adenomatoid hamartoma: a case report

Camille Andrews Ryan Veterinary Hospital, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA

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Lillian Aronson Ryan Veterinary Hospital, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA

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Molly Church Ryan Veterinary Hospital, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA

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Hunter Piegols Ryan Veterinary Hospital, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA

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Abstract

OBJECTIVE

To describe the clinical presentation, novel surgical approach, and outcome of a dog diagnosed with chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH).

ANIMAL

5-year-old castrated male Yorkshire Terrier.

CLINICAL PRESENTATION, PROGRESSION, AND PROCEDURES

The dog was presented with chronic upper respiratory noise, congestion, facial swelling, ocular discharge, and an abscess on the nasal bridge. Two CT scans were performed 4 months apart. The CT scans yielded similar results: cyst-like nasal masses with severely destructive bilateral rhinitis with extensive polyostotic bony lysis. A dorsal rhinotomy with a turbinectomy and debridement of the nasal cavity were performed. A poorly defined but extensive lesion was found occupying the entirety of the left frontal sinus as well as the nasal cavity.

TREATMENT AND OUTCOME

Histopathology revealed a mass consistent with COREAH. The dog recovered well from surgery, except for self-limiting subcutaneous emphysema, and 3 weeks postoperatively was reportedly doing well, with mild nasal discharge. Stridor, nasal discharge, and sneezing episodes were reported postoperatively; however, these were improved. At 18 months postoperatively, the dog died from uncontrolled seizures while hospitalized for suspected acute hemorrhagic diarrhea syndrome at a different hospital.

CLINICAL RELEVANCE

COREAH should be considered a potential cause of destructive bilateral rhinitis and bony lysis in dogs. Dorsal rhinotomy can be a surgical treatment for dogs with possible COREAH with acceptable outcome, though complete remission of clinical signs may not be achieved. This is the first clinical description of COREAH in a dog.

Abstract

OBJECTIVE

To describe the clinical presentation, novel surgical approach, and outcome of a dog diagnosed with chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH).

ANIMAL

5-year-old castrated male Yorkshire Terrier.

CLINICAL PRESENTATION, PROGRESSION, AND PROCEDURES

The dog was presented with chronic upper respiratory noise, congestion, facial swelling, ocular discharge, and an abscess on the nasal bridge. Two CT scans were performed 4 months apart. The CT scans yielded similar results: cyst-like nasal masses with severely destructive bilateral rhinitis with extensive polyostotic bony lysis. A dorsal rhinotomy with a turbinectomy and debridement of the nasal cavity were performed. A poorly defined but extensive lesion was found occupying the entirety of the left frontal sinus as well as the nasal cavity.

TREATMENT AND OUTCOME

Histopathology revealed a mass consistent with COREAH. The dog recovered well from surgery, except for self-limiting subcutaneous emphysema, and 3 weeks postoperatively was reportedly doing well, with mild nasal discharge. Stridor, nasal discharge, and sneezing episodes were reported postoperatively; however, these were improved. At 18 months postoperatively, the dog died from uncontrolled seizures while hospitalized for suspected acute hemorrhagic diarrhea syndrome at a different hospital.

CLINICAL RELEVANCE

COREAH should be considered a potential cause of destructive bilateral rhinitis and bony lysis in dogs. Dorsal rhinotomy can be a surgical treatment for dogs with possible COREAH with acceptable outcome, though complete remission of clinical signs may not be achieved. This is the first clinical description of COREAH in a dog.

History

A 4-year-old castrated male Yorkshire Terrier was presented for a history of facial swelling and chronic upper airway noise since adoption 3 years prior to presentation. Historically, the dog had mucoid ocular discharge and wheezing, which progressed 4 months before presentation. Additionally, the dog’s owner recognized increasing congestion, facial swelling, and an abscess that had been lanced and drained by the emergency service at the presenting hospital. The dog had been placed on various trials of amoxicillin and clavulanate; neomycin, polymyxin B, and bacitracin; meloxicam; and prednisone, which intermittently improved clinical signs before recurrence.

Diagnostic Findings and Interpretation

Consent was obtained from the owner of the dog prior to all procedures, diagnostics, and care. Initial physical examination findings included interocular swelling, abnormal air flow through both nostrils, pain on facial palpation, decreased retropulsion of both eyes, and severe upper respiratory noise. Thoracic radiographs were taken and revealed no evidence of pulmonary metastasis or bronchopneumonia.

Twenty-six days after initial presentation, the dog was re-presented for a CT scan of the head. Physical examination was similar other than bloody discharge associated with the interocular swelling. The CT scan revealed a rim-enhancing nasal mass causing bilateral destruction of the nasal turbinates (Figure 1). The mass extended through areas of bony lysis with subsequent moderate facial swelling and a draining tract over the nasal soft tissues. The mass extended into the nasopharynx, oral cavity, sphenoidal sinuses, and potentially into the frontal sinuses and retrobulbar spaces bilaterally. The mass contained mineral foci extending linearly from the lacrimal sac region to the left and right nasolacrimal canals and into the caudal nasal passages. Additionally, smoothly margined lysis of the maxillary, palatine, and frontal bones was suggestive of pressure necrosis.

Figure 1
Figure 1

Computed tomography images approximately 1 month after initial presentation showing destructive bilateral rim-enhancing nasal masses, causing bilateral destruction of the nasal turbinates (A) as well as mineral foci, with the arrow pointing to the region with foci, near the region of the lacrimal sac, rostromedial orbit, and left and right nasolacrimal ducts and canal within the caudal nasal passages (B).

Citation: Journal of the American Veterinary Medical Association 262, 3; 10.2460/javma.23.10.0559

The top differential was a chronic nasal polyp and secondary destructive rhinitis, with other infectious, inflammatory, and neoplastic processes possible. Fluid cytology obtained from the draining tract revealed marked suppurative inflammation within viscous proteinaceous fluid consistent with an inflammatory process or possible neoplasia, although no neoplastic cells were identified. A rhinoscopic biopsy was performed and revealed severe chronic suppurative rhinosinusitis. No evidence of neoplasia or infectious agents was found.

Four months later, the dog was re-presented for further evaluation of the upper airway noise, though the draining tract and facial swelling had resolved with no medications prescribed in the interim. A second CT scan was performed to assess for disease progression. The mass had similar destructive bilateral rhinitis with cystic regions and osteolysis of the skull bones, with some regions seeming to have less mass-like tissue. One area of progression noted, however, included erosion of the left ventromedial cribriform. A nasal polyp remained the primary differential with concurrent infection or neoplasia possible as well as conditions like a hamartoma.

Treatment and Outcome

A dorsal rhinotomy and frontal sinusotomy with turbinectomy and nasal cavity debridement using a standard technique was performed.1 Briefly, a midline skin incision was made from dorsal to the orbits to the rostral aspect of the nasal planum using a No. 10 blade. Upon dissection of the subcutaneous tissues, mucoid material protruded from a bone defect (Figure 2). The periosteum on either side of the incision was elevated with a Freer periosteal elevator, and the existing defect in the bone was expanded with Lempert rongeurs. A poorly defined but extensive lesion was found occupying the entirety of the left frontal sinus, and there was diffuse pathology within the nasal cavity involving both the left and right nasal turbinates and nasal septum. The mass was red and friable with indistinct borders. Bony remodeling resulted in a firm obstruction of the right choana, through which a red rubber catheter was unable to be passed. A bone curette, mosquito hemostats, Lempert rongeurs, and suction using a Frazier suction tip were used to remove abnormal tissue within the frontal sinus and nasal cavity. Bony remodeling was removed using a Hall pneumatic air drill. Samples of abnormal tissue were submitted for histopathology and bacterial culture. Moderate hemorrhage occurred during dissection that was managed with gauze packing and diluted epinephrine as well as flushing with cold saline prior to closure. A tie-over bandage was placed immediately following closure. The dog recovered uneventfully from anesthesia. Shortly after recovery, the patient developed significant subcutaneous emphysema; the tie-over bandage was removed and was not replaced. The subcutaneous emphysema resolved on its own after several weeks. The dog was discharged 2 days after surgery.

Figure 2
Figure 2

Intraoperative photographs of the dorsal rhinotomy surgical approach showing mucoid material protruding through a bone defect after incising subcutaneous tissue (A). Further exposure revealed a poorly defined mass occupying the frontal sinus and nasal cavity (B). A turbinectomy with debridement was performed within the nasal cavity before closure (C).

Citation: Journal of the American Veterinary Medical Association 262, 3; 10.2460/javma.23.10.0559

Six fragments of the mass were submitted for histologic evaluation. The mass was covered by respiratory epithelium, which was subtended by a dense collagenous stroma containing irregular plates of chondro-osseous matrix, some demonstrating necrosis (Figure 3). There was marked expansion of the seromucinous glands and collapsed adenomatoid proliferations lined by respiratory epithelium. No infectious agents were found. The mass was diagnosed as severe glandular hyperplasia with chondroid necrosis and bone remodeling consistent with chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH).

Figure 3
Figure 3

Photomicrograph of a chondro-osseous respiratory epithelial adenomatoid hamartoma demonstrating respiratory epithelium covering an irregularly polypoid mass with irregular osseous plate.

Citation: Journal of the American Veterinary Medical Association 262, 3; 10.2460/javma.23.10.0559

Three weeks after surgery, the dog was reported as doing well, with normal appetite and energy levels. Bilateral mucoid discharge and sneezing were noted initially, and the subcutaneous emphysema decreased by the time of the first follow-up appointment. The owner reported that the dog was doing acceptably well at 10 months postoperatively. The dog would have occasional episodes of sneezing with nasal discharge. The dog’s respiratory noise had improved since surgery, though never completely resolved. Eighteen months after the procedure, the dog died secondarily to uncontrolled seizures while hospitalized for suspected acute hemorrhagic diarrhea syndrome at another hospital; this was not suspected to be related to the COREAH.

Comments

COREAH are exceedingly rare tumors, with a literature review identifying 18 human cases and 3 canine histologic descriptions.2,3 The treatment and clinical outcome in dogs has not been previously reported; specifically, the use of a dorsal rhinotomy and sinusotomy for this lesion has not been previously described. COREAH are thought to be a subset of REAH that develop from the pseudostratified respiratory epithelium lining the sinonasal space and nasal cavity.3,4 REAH are rare tumors of the respiratory tract, with < 400 reported cases in people and 2 reported cases in dogs.2,5 Clinical signs of COREAH in dogs are similar to those of other nasal masses and include exophthalmia, epistaxis, difficulty on inspiration, reverse sneezing, and lethargy.2 Computed tomography and MRI are the primary imaging modalities used to identify COREAH lesions in humans; CT and skull radiographs have been reported for identification in dogs.2,3 COREAH in dogs share similar characteristics on CT as those reported in humans, including unilateral and multifocally mineralized soft tissue masses that cause septal deviation; however, one difference in dogs is the presence of osteolysis of skull bones or damage to the nasal turbinates.2 The initial primary differential in this dog was a chronic nasal polyp with secondary destructive rhinitis given the CT findings and prolonged history, with other infectious, inflammatory, and neoplastic processes also considered; given the overall similarities between the 2 CT scans, a malignant process became lower on the differential list due to lack of significant progression. COREAH in dogs are described as polypoid and pale pink to tan with red mottling that may be soft or firm.2 In humans, they are smooth masses, with some reported as being firm.3 Microscopically, COREAH have a chondro-osseous trabecula with respiratory epithelium-lined glandular adenomatoid proliferation and subsequent polypoid growth.2

Currently, there is a paucity of information on the management of COREAH in dogs. Endoscopic tumor resection is described in human medicine.3 In veterinary medicine, the treatment of REAH with endoscopic evaluation via a lateral rhinotomy and coablation was suspected to be curative in a case report.5 In the histologic description of COREAH in 3 dogs, 2 were euthanized without direct medical intervention and 1 was still living at 5 months after excisional biopsy, with no further details provided.2

Until additional studies comparing treatment approaches are performed, a dorsal rhinotomy with a turbinectomy and debridement of the nasal cavity may be considered as a technique for COREAH in dogs given its reported outcome here. A dorsal rhinotomy allowed full visualization and debridement of abnormal tissue, which subsequently allowed submission for biopsy for a definitive diagnosis. After surgery, the dog’s clinical signs improved, with the dog succumbing to likely unrelated disease 18 months postoperatively. Further studies and documentation of cases can provide insight into alternative surgical approaches and better describe the clinical outcome of dogs with COREAH undergoing various treatments.

Acknowledgments

None reported.

Disclosures

The authors have nothing to disclose. No AI-assisted technologies were used in the generation of this manuscript.

Funding

The authors have nothing to disclose.

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