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Recurrent intraluminal eosinophilic tracheal granuloma in a Siberian husky a
a
b
c
Katerina K. Adamama-Moraitou , Nektarios Soubasis , Dimitra Pardali , Dimitra Psalla , d
d
e
Lysimachos G. Papazoglou , Nikitas N. Prassinos , Tilemachos L. Anagnostou & Timoleon a
S. Rallis a
Companion Animal Clinic (Medicine Unit), Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece b
Diagnostic Laboratory, Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece c
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Laboratory of Pathology, School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece d
Companion Animal Clinic (Surgery and Obstetrics Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece e
Companion Animal Clinic (Anesthesiology and Intensive Care Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece Accepted author version posted online: 23 Feb 2015.Published online: 17 Mar 2015.
To cite this article: Katerina K. Adamama-Moraitou, Nektarios Soubasis, Dimitra Pardali, Dimitra Psalla, Lysimachos G. Papazoglou, Nikitas N. Prassinos, Tilemachos L. Anagnostou & Timoleon S. Rallis (2015): Recurrent intraluminal eosinophilic tracheal granuloma in a Siberian husky, Veterinary Quarterly, DOI: 10.1080/01652176.2015.1021493 To link to this article: http://dx.doi.org/10.1080/01652176.2015.1021493
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Veterinary Quarterly, 2015 http://dx.doi.org/10.1080/01652176.2015.1021493
CASE REPORT Recurrent intraluminal eosinophilic tracheal granuloma in a Siberian husky Katerina K. Adamama-Moraitoua, Nektarios Soubasisa, Dimitra Pardalib*, Dimitra Psallac, Lysimachos G. Papazogloud, Nikitas N. Prassinosd, Tilemachos L. Anagnostoue and Timoleon S. Rallisa a
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Companion Animal Clinic (Medicine Unit), Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; bDiagnostic Laboratory, Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; cLaboratory of Pathology, School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; d Companion Animal Clinic (Surgery and Obstetrics Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; eCompanion Animal Clinic (Anesthesiology and Intensive Care Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece (Received 29 September 2014; accepted 17 February 2015) Keywords: dog; canine; tracheal tumor; eosinophilic granuloma; tracheal obstruction; episodic dyspnea
A 5-year-old intact male, Siberian husky, routinely vaccinated and dewormed for intestinal parasites and ectoparasites was referred with a two-week history of recurrent episodes of respiratory distress, stridor, coughing and gagging. Dyspnea lasting for 2 3 minutes occurred upon exercise and/or excitement. Hemoptysis and hypersalivation were noticed just once. The referring veterinarian had prescribed doxycycline (vibramycin, Pfizer, New Jersey USA) at a dose of 5.0 mg/kg body weight (BW) twice daily for a 3-day period, but the dog’s clinical condition failed to improve. Upon admission, episodic inspiratory dyspnea and stridor were detected. Between the episodes, the dog was not exhibiting outward signs. Physical examination revealed a normal body temperature and mucous membrane color. Auscultation of the cervical trachea identified a high-pitched inspiratory sound, and tracheal palpation induced a non-productive cough. A preliminary list of differentials included laryngeal or tracheal disorders leading to airway lumen stenosis (e.g. laryngeal paralysis, or laryngeal or tracheal collapse, foreign body, abscess, granuloma and tumor). The results of a complete blood count (PCV 48%; reference range 37% 55%, hemoglobin concentration 2.7 mmol/L; ref. range 1.9 2.8 mmol/L, WBC 8.4 G/L; ref. range 6.0 16.9 G/L with 6.6 G/L neutrophils, 1.3 G/L lymphocytes, 0.5 G/L eosinophils and 0 G/L monocytes as well as 271 G/L platelets; ref. range 175 500 G/L), routine biochemical profile, as well as urinalysis and arterial blood gas analysis collected under non-stressful conditions were found to be within normal limits. Fecal examination with Baermann and flotation and sedimentation methods for parasitic eggs and serology for Dirofilaria immitis (SNAP Heartworm, IDEXX, USA) were negative.
Lateral radiographs of the neck and thorax demonstrated a soft tissue density located in the cervical trachea, just caudal to the larynx, protruding into the lumen (Figure 1). During endoscopic examination, performed under general anesthesia, the larynx was found to be structurally and functionally normal, while a bilobed, hyperemic, hemorrhagic mass was detected within the cervical trachea occupying approximately 70% of its lumen (Figure 2). The endoscope (Olympus BF-PEZ, Olympus Optical, Hamburg, Germany) was able to bypass the mass. Inspection of the remaining tracheobronchial tree did not reveal any other pathological finding. Squash smears of endoscopically collected tissue specimens using an endoscopic biopsy forceps (Olympus FB-21C-1, Olympus Optical, Hamburg, Germany) that were prepared for cytology revealed normal tracheal mucosa. Surgical excision of the mass was recommended and scheduled two days after admission, due to the potentially life-threatening condition presented by the size and location of the mass, and the inability to reach a diagnosis through cytology. Tracheal resection and anastomosis were performed through a ventral cervical midline incision under general anesthesia. Six tracheal cartilage rings were removed, starting from the second cartilage caudal to the larynx. The mass was localized in the middle of the resected tracheal segment; it was solid, hyperemic, bilobed, hemorrhagic and was firmly attached to four consecutive tracheal rings. It was adhered to the three-fourth of the perimeter of the tracheal wall and protruded into the tracheal lumen, occupying approximately 70% of its space. Anastomosis was carried out under minimal tension using 2/0 polypropylene suture material in a simple, interrupted pattern. The surgical wound was lavaged with normal saline and closed routinely. Cefuroxime (zinacef, GlaxoSmithKline, Greece) was administrated at intra-operative
*Corresponding author. Emails: [email protected]; [email protected] Ó 2015 Taylor & Francis
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Figure 1. Lateral radiograph of the cervical region of a 5-year-old male, Siberian husky dog demonstrating a soft tissue density mass (white arrows) located at the cervical trachea just caudal to the larynx, protruding into its lumen.
period (25 mg/kg BW, as a bolus, iv) and for 10 days postoperatively (zinadol, GlaxoSmithKline, Greece) at the dose of 25 mg/kg BW, twice daily, per os. Having made an uneventful recovery, the dog was discharged from hospital
Figure 2. Endoscopic view of a bilobed, hyperemic, hemorrhagic mass (eosinophilic granuloma) within the cervical trachea protruding into its lumen in a 5-year-old male, Siberian husky dog.
three days after surgery. Imprint cytological smears prepared from the incised tracheal mass showed that eosinophils were the predominant cell type. The resected tracheal rings along with the contiguous tracheal mass were fixed in 10% phosphate-buffered formalin for subsequent histologic examination. The biopsy specimens were processed routinely, and 5 mm sections were cut and stained with hematoxylin and eosin. Histopathology showed that the mass (Figures 3 and 4) consisted of numerous eosinophils, epithelioid macrophages, lymphocytes, plasma cells, fewer neutrophils and mast cells. Multinucleated cells were not observed. The background consisted of many reactive fibroblasts, with surrounding and separate bundles of collagen, and many variable-sized blood vessels lined by reactive endothelium (granulation tissue). Multifocally, collagen fibers were hyalinized but eosinophilic degranulation around collagen bundles (‘flame figures’) were absent. Multifocally, many extravasated erythrocytes were also present. No evidence of parasitic, bacterial or fungal agents was found after serial sections examined with special stainings (Grocott Gomori methenamine silver, periodic acid Schiff and Gram). Examination of the surgical margins showed that there was approximately 2 3 mm of normal tissue surrounding the mass. Mast cell tumor and eosinophilic rich T-cell lymphoma were excluded by histochemical
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Figure 3. Well-demarcated granulomatous nodule attached to the cartilage of the tracheal wall (hematoxylin and eosin staining, bar D 500 mm).
(toluidine blue) and immunohistochemical (IHC against CD3 and CD79a) stainings. Following suture removal 10 days after surgery, prednisone (prezolon, Nycomed, Greece) was administered for a total of 30-day period at a low immunosuppressive dose (0.5 mg/kg BW, twice daily, per os) for 20 days, and tapered gradually over a 10-day period in order to prevent
recurrence of the condition. No anti-parasitic treatment was given except of milbemycin (interceptor, Novartis Animal Health, Switzerland) at a dose of 11.5 mg, once a month, per os. Clinical signs subsided just after surgery. Upon reevaluation 45 days after surgical excision of the mass, the owner reported that all clinical signs had resolved, even
Figure 4. Eosinophils were the predominant population in the mass (hematoxylin and eosin staining, bar D 125 mm). Inset: eosinophils – bilobular cells with eosinophilic granules.
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during exercise. No abnormalities were evident on physical examination. Lateral radiographs of the neck and thorax were unremarkable. Endoscopic re-evaluation 46 days after surgery revealed a small in size (less than 0.7 cm in height and 0.5 cm in width), whitish, mushroom-shaped tracheal mass occluding approximately 5% of the tracheal lumen, located 1 2 cm caudally to the anastomosis (Figure 5). Histopathologic examination of tissue specimens obtained endoscopically demonstrated a recurrence of tracheal eosinophilic granuloma. Due to the dog’s good clinical condition and the size of the mass, conservative management was elected consisting of prednisone (1 mg/kg BW, twice daily, per os) for 30 days. The steroid dosage was tapered gradually over a 40-day period; steroids were given for 70 days in total. Additionally, cefuroxime (zinadol, GlaxoSmithKline, Greece) at the dose of 25 mg/kg BW, twice daily, per os was prescribed for the initial 30 days of steroid administration. At the end of the steroid therapy, a third endoscopy was performed revealing remission of the mass. One year after the third endoscopic evaluation, a fourth endoscopy revealed a normal tracheal lumen. Two years after the last endoscopic examination, follow-up clinical evaluation of the dog showed no evidence of abnormal findings. The aim of this study was to describe the diagnostic approach, management and outcome of an intraluminal tracheal eosinophilic granuloma in a Siberian husky dog which recurred after a combination of surgical excision and steroid administration, but eventually resolved with higher doses of steroids. Tracheal intraluminal neoplastic or hyperplastic masses associated with primary or metastatic tumors (e.g. canine mast cell tumor, T-cell lymphoma), parasitic granulomas, abscesses due to a foreign body, inflammatory mucosal polyps and trauma usually cause nodular lesions in the dog, resulting in tracheal obstruction (Beck et al.
Figure 5. Endoscopic view of a small-sized, intraluminal tracheal mass (eosinophilic granuloma) 45 days after surgical excision of a pre-existing mass. The location of the relapsed mass was approximately 1 2 cm caudal to the anastomosis.
1999; Clifford & Sorenmo 2004; Sherding 2004). Hyperplastic tracheal nodules of unknown origin (De Rick et al. 1977) or related to amyloidosis (Besancon et al. 2004) have also been reported. The magnitude of clinical signs correlates with the severity of tracheal obstruction. Affected dogs may present with a variety of upper airway obstruction signs (Clifford & Sorenmo 2004). Histopathology remains the cornerstone of diagnosis while plain radiography, computerized tomography and endoscopy along with the severity of clinical signs, size of lesion and its location determine the treatment modalities (Clifford & Sorenmo 2004). A list of differentials for the tracheal mass in our case included a tracheal foreign body, tracheal neoplasm, parasitic granuloma (Spirocerca lupi, Onchocerca spp., Oslerus osleri) and eosinophilic granuloma. Histopathologic examination of biopsy specimens taken at tracheoscopy and/or surgical excision was required to confirm the diagnosis. Although the trachea is an unexpected site for canine spirocercosis, S.lupi infection was included among differentials since it is endemic in our region (Haralabidis et al. 1988). A case of obstructive granulomatous tracheitis caused by Onchocerca spp. in a dog has also been described in the past (Papaioannou et al. 2004). Although not seen in our country, the most common nematode to infect the trachea of young dogs is O.osleri (Sherding 2004). Typical lesions are localized in the distal trachea and tracheal bifurcation, forming granulomatous nodules. The most reliable way to diagnose O.osleri infestation is by direct visualization of the parasitic granuloma and larvae during tracheoscopy (Sherding 2004). However, in the present case, a bilobed and almost linear mass was detected in the proximal part of the trachea and no worms were visible. Fecal examination for parasitic infestation and histological and parasitological examination of multiple sections of the tracheal nodule failed to reveal any parasitic agent. Histomorphologically, the lesions seen in this dog closely resembled those described in the skin and oral mucosa of Siberian huskies affected by eosinophilic granulomatous disease (Potter et al. 1980; Scott et al. 2000). Finally, tracheal intussusception was easily excluded at endoscopy (Kim et al. 2012). Canine eosinophilic granuloma is a not very common, benign disorder of unknown etiology that manifests as nodular or plaque-like lesions, predominantly on the skin, tongue or in the oral cavity (Potter et al. 1980; Scott et al. 2000). Other less common locations described are lungs (Confer et al. 1983; Neer et al. 1986; von Rotz et al. 1986; Katajavuori et al. 2012), nasal planum (Da Silva Curiel et al. 1988), external ear canal (Poulet et al. 1991), eyelids (Vercelli et al. 2005), gastrointestinal tract and liver (Brellou et al. 2006). Tracheal eosinophilic granuloma has been rarely described in the literature and only two dogs have been reported: a 10-year-old female, German shepherd (Brovida & Castagnaro 1992) and a 2-year-old male, Siberian husky (Ramirez & Hawkins 1998). Canine tracheal eosinophilic granuloma has been previously reported in just two dogs (Brovida & Castagnaro 1992; Ramirez & Hawkins 1998). The case presented herein is the second to be reported in a Siberian husky.
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Veterinary Quarterly Breed predilection in the latter concerning the oral form of the disease has been described (Madewell et al. 1980; Potter et al. 1980). Several pathogenetic mechanisms have been suggested, including vasculitis, microangiopathy, disorders of fibrinolysis or phagocytic function, an immunologic hypersensitivity reaction to an unidentified antigen or to a grass awn or to a parasite, or heritable predisposition secondary to an unknown injury (Scott et al. 2000; Katajavuori et al. 2012). A possible correlation with bacterial, fungal, parasitic or viral infections appears unlikely (Madewell et al. 1980; Scott et al. 2000). As in the previous cases described, the cause could not be determined; however, it may have resulted from a primary unknown injury with secondary immune-mediated inflammatory reaction. Nevertheless, secondary (reactive) tissue eosinophilia where eosinophils migrate to a specific organ due to a reactive process (helminth infection, allergic disease, drug reaction, chronic inflammatory disorder, neoplastic condition, etc.) could lead to local eosinophil accumulation and tissue remodeling (Valent et al. 2012). A high incidence of eosinophilic granulomatous disease has been noted among Siberian huskies and Cavalier King Charles spaniels. The disease has been attributed to a genetic predisposition (Madewell et al. 1980; Potter et al. 1980; Brodal et al. 1996). However, there is plenty of information to indicate that genetic traits modify the risk and nature of hypersensitivity reactions (Valent et al. 2012; Gotlib 2014). In human medicine, pulmonary eosinophilic granuloma, also known as pulmonary Langerhans cell histiocytosis (PLCH), is part of a complex group of diseases known as Langerhans cell histiocytosis (Hoover et al. 2007). The pathogenesis and etiology of PLCH remain unknown, but it seems to be common among smokers (Friedman et al. 1981), probably due to an inflammatory reaction of the lung tissue to the components of cigarette smoke (Suzuki et al. 2004). Pulmonary Langerhans cell histiocytosis rarely occurs as a solitary nodule (Khoor et al. 2001). Fridlender et al. (2005) described a unique case of PLCH in which the sole manifestation of the disease in a patient was a partially obstructing tracheal mass, which responded well to local excision. Although such a syndrome has not been described in a dog, the present case appears similar to their case. Canine tracheal mast cell tumor and eosinophilic rich T-cell lymphoma of the trachea had to be differentiated from tracheal eosinophilic granuloma due to their similar microscopical appearance following routine histological staining. Advanced histopathological techniques (toluidine blue and IHC against CD3 and CD79a) were able to differentiate those conditions from tracheal eosinophilic granuloma. The poor prognosis of the above-mentioned neoplastic lesions promoted the necessity of their differentiation, since they demand a different post-surgical management (Fox and King 2002). The absence of an increased number of circulating eosinophils was consistent with the case reported by Brovida and Castagnaro (1992). Some cases with marked peripheral eosinophilia could suffer from inflammatory or hypersensitivity disorders (German et al. 2002), but in our
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case, a disorder of this origin was not confirmed. In contrast, Ramirez and Hawkins (1998) reported a case displaying mild eosinophilia where a second eosinophilic lesion was detected in the dorsal part of nasopharynx. Similarly, Lillieh€o€ok and colleagues (2000) mentioned that most cases of eosinophilic lesions in any part of the gastrointestinal tract showed marked eosinophilia. Most canine eosinophilic granulomas have responded to glucocorticoid therapy (Potter et al. 1980; Madewell et al. 1980; Brovida & Castagnaro 1992; Vercelli et al. 2005). Prednisone at 0.5 to 2.2 mg/kg BW orally once daily, or spread over a three- to four-week period, is usually adequate to control clinical manifestations of the granulomas. However, spontaneous remission has occasionally been reported (Madewell et al. 1980; Potter et al. 1980; Ramirez & Hawkins 1998). The case described in this report, similar to the case described by Ramirez and Hawkins (1998) and unlike the case presented by Brovida and Castagnaro (1992), did not completely respond to surgical excision and the low-dose corticosteroid adjunctive treatment prescribed thereafter. The relapsed nodule responded well to increased steroid doses. This emphasizes the necessity for the administration of steroids in immunosuppressive doses at the initial pharmaceutical management. Endoscopic re-evaluation of such cases is crucial because, as in this case, the lesion can recur despite surgical excision and corticosteroid treatment. Also, unlike the case of Ramirez and Hawkins (1998), no recurrence of clinical signs was observed after discontinuation of the second, higher dose of oral anti-inflammatory therapy probably due to the small size of the second granuloma at the time of re-evaluation; thus, it may be suggested that all cases of tracheal eosinophilic granulomas should be concurrently treated with immunosuppressive doses of corticosteroid regardless of clinical signs; treatment should not rely solely on surgical excision. Moreover, response to glucocorticosteroid therapy (consisted of inhibition of eosinophil growth and function) in reactive tissue eosinophilia is not only dose-dependent but correlates well with the number of glucocorticosteroid receptors detected upon eosinophils (Valent et al. 2012). The duration of corticosteroid therapy must be determined according to endoscopic findings. Use of other immunosuppressant agents to avoid possible side effects of corticosteroid therapy, which are not unlikely in large breed dogs, were not elected in this dog due to lack of data regarding their effectiveness in cases of tracheal eosinophilic granuloma. Further studies are needed to prove if such agents are beneficial or not as a replacement therapy. Endoscopic re-evaluation of the tracheal lumen could detect a recurrence of the lesion at its early stages before any clinical signs are apparent. The fact that no clinical evidence of the disease was apparent two years after the last endoscopic examination does not completely rule out the possible presence of a new small-sized tracheal nodule that had no clinical signs. Another endoscopic evaluation at this time point would have been ideal in this patient. Despite the fact that surgical resection of the tracheal rings is considered an invasive option for removal of a granuloma, this was elected in our patient because of its
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critical condition and the failure of cytology to contribute to a definitive diagnosis. Non-invasive alternatives include laser ablation which usually addresses to pedunculated tumors, endotracheal stenting only as a palliative method, etc. (Shapshay 1987; Rebeiz et al. 1990; Freitag & Darwiche 2014). Moreover, we cannot rule out the possibility that if immunosuppressive prednisone was initiated just after the first endoscopy, eventually surgery would have been avoided. In Siberian huskies, canine eosinophilic granulomatous disease has been previously described to affect the skin, oral cavity, gastrointestinal tract, liver, eyelid and tracheal lumen (Madewell et al. 1980; Potter et al. 1980; Scott et al. 2000; Vercelli et al. 2005; Brellou et al. 2006; Lyles et al. 2009). The oral form of the disease prevails and lesions can be located both in lingual or palatine tissue (Ramirez & Hawkins 1998). Histopathologic findings in our case resembled those described by others for the oral cavity and skin of Siberian huskies, supporting the view that this breed is predisposed to the development of eosinophilic granulomas (Johnson 1992; Brellou et al. 2006). In conclusion, it seems that long-term re-examinations should be implemented in cases of canine tracheal eosinophilic granulomatous disease treated with surgery and corticosteroid treatment for early detection of a relapse. Moreover, in the case of recurrence, higher doses of corticosteroid treatment should be attempted to achieve complete resolution.
Disclosure statement No potential conflict of interest was reported by the authors.
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Recurrent intraluminal eosinophilic tracheal granuloma in a Siberian husky a
a
b
c
Katerina K. Adamama-Moraitou , Nektarios Soubasis , Dimitra Pardali , Dimitra Psalla , d
d
e
Lysimachos G. Papazoglou , Nikitas N. Prassinos , Tilemachos L. Anagnostou & Timoleon a
S. Rallis a
Companion Animal Clinic (Medicine Unit), Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece b
Diagnostic Laboratory, Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece c
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Laboratory of Pathology, School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece d
Companion Animal Clinic (Surgery and Obstetrics Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece e
Companion Animal Clinic (Anesthesiology and Intensive Care Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece Accepted author version posted online: 23 Feb 2015.Published online: 17 Mar 2015.
To cite this article: Katerina K. Adamama-Moraitou, Nektarios Soubasis, Dimitra Pardali, Dimitra Psalla, Lysimachos G. Papazoglou, Nikitas N. Prassinos, Tilemachos L. Anagnostou & Timoleon S. Rallis (2015): Recurrent intraluminal eosinophilic tracheal granuloma in a Siberian husky, Veterinary Quarterly, DOI: 10.1080/01652176.2015.1021493 To link to this article: http://dx.doi.org/10.1080/01652176.2015.1021493
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Veterinary Quarterly, 2015 http://dx.doi.org/10.1080/01652176.2015.1021493
CASE REPORT Recurrent intraluminal eosinophilic tracheal granuloma in a Siberian husky Katerina K. Adamama-Moraitoua, Nektarios Soubasisa, Dimitra Pardalib*, Dimitra Psallac, Lysimachos G. Papazogloud, Nikitas N. Prassinosd, Tilemachos L. Anagnostoue and Timoleon S. Rallisa a
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Companion Animal Clinic (Medicine Unit), Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; bDiagnostic Laboratory, Faculty of Health Sciences, School of Veterinary Medicine, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; cLaboratory of Pathology, School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; d Companion Animal Clinic (Surgery and Obstetrics Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece; eCompanion Animal Clinic (Anesthesiology and Intensive Care Unit), School of Veterinary Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, St. Voutira 11 str., 54627, Thessaloniki, Greece (Received 29 September 2014; accepted 17 February 2015) Keywords: dog; canine; tracheal tumor; eosinophilic granuloma; tracheal obstruction; episodic dyspnea
A 5-year-old intact male, Siberian husky, routinely vaccinated and dewormed for intestinal parasites and ectoparasites was referred with a two-week history of recurrent episodes of respiratory distress, stridor, coughing and gagging. Dyspnea lasting for 2 3 minutes occurred upon exercise and/or excitement. Hemoptysis and hypersalivation were noticed just once. The referring veterinarian had prescribed doxycycline (vibramycin, Pfizer, New Jersey USA) at a dose of 5.0 mg/kg body weight (BW) twice daily for a 3-day period, but the dog’s clinical condition failed to improve. Upon admission, episodic inspiratory dyspnea and stridor were detected. Between the episodes, the dog was not exhibiting outward signs. Physical examination revealed a normal body temperature and mucous membrane color. Auscultation of the cervical trachea identified a high-pitched inspiratory sound, and tracheal palpation induced a non-productive cough. A preliminary list of differentials included laryngeal or tracheal disorders leading to airway lumen stenosis (e.g. laryngeal paralysis, or laryngeal or tracheal collapse, foreign body, abscess, granuloma and tumor). The results of a complete blood count (PCV 48%; reference range 37% 55%, hemoglobin concentration 2.7 mmol/L; ref. range 1.9 2.8 mmol/L, WBC 8.4 G/L; ref. range 6.0 16.9 G/L with 6.6 G/L neutrophils, 1.3 G/L lymphocytes, 0.5 G/L eosinophils and 0 G/L monocytes as well as 271 G/L platelets; ref. range 175 500 G/L), routine biochemical profile, as well as urinalysis and arterial blood gas analysis collected under non-stressful conditions were found to be within normal limits. Fecal examination with Baermann and flotation and sedimentation methods for parasitic eggs and serology for Dirofilaria immitis (SNAP Heartworm, IDEXX, USA) were negative.
Lateral radiographs of the neck and thorax demonstrated a soft tissue density located in the cervical trachea, just caudal to the larynx, protruding into the lumen (Figure 1). During endoscopic examination, performed under general anesthesia, the larynx was found to be structurally and functionally normal, while a bilobed, hyperemic, hemorrhagic mass was detected within the cervical trachea occupying approximately 70% of its lumen (Figure 2). The endoscope (Olympus BF-PEZ, Olympus Optical, Hamburg, Germany) was able to bypass the mass. Inspection of the remaining tracheobronchial tree did not reveal any other pathological finding. Squash smears of endoscopically collected tissue specimens using an endoscopic biopsy forceps (Olympus FB-21C-1, Olympus Optical, Hamburg, Germany) that were prepared for cytology revealed normal tracheal mucosa. Surgical excision of the mass was recommended and scheduled two days after admission, due to the potentially life-threatening condition presented by the size and location of the mass, and the inability to reach a diagnosis through cytology. Tracheal resection and anastomosis were performed through a ventral cervical midline incision under general anesthesia. Six tracheal cartilage rings were removed, starting from the second cartilage caudal to the larynx. The mass was localized in the middle of the resected tracheal segment; it was solid, hyperemic, bilobed, hemorrhagic and was firmly attached to four consecutive tracheal rings. It was adhered to the three-fourth of the perimeter of the tracheal wall and protruded into the tracheal lumen, occupying approximately 70% of its space. Anastomosis was carried out under minimal tension using 2/0 polypropylene suture material in a simple, interrupted pattern. The surgical wound was lavaged with normal saline and closed routinely. Cefuroxime (zinacef, GlaxoSmithKline, Greece) was administrated at intra-operative
*Corresponding author. Emails: [email protected]; [email protected] Ó 2015 Taylor & Francis
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Figure 1. Lateral radiograph of the cervical region of a 5-year-old male, Siberian husky dog demonstrating a soft tissue density mass (white arrows) located at the cervical trachea just caudal to the larynx, protruding into its lumen.
period (25 mg/kg BW, as a bolus, iv) and for 10 days postoperatively (zinadol, GlaxoSmithKline, Greece) at the dose of 25 mg/kg BW, twice daily, per os. Having made an uneventful recovery, the dog was discharged from hospital
Figure 2. Endoscopic view of a bilobed, hyperemic, hemorrhagic mass (eosinophilic granuloma) within the cervical trachea protruding into its lumen in a 5-year-old male, Siberian husky dog.
three days after surgery. Imprint cytological smears prepared from the incised tracheal mass showed that eosinophils were the predominant cell type. The resected tracheal rings along with the contiguous tracheal mass were fixed in 10% phosphate-buffered formalin for subsequent histologic examination. The biopsy specimens were processed routinely, and 5 mm sections were cut and stained with hematoxylin and eosin. Histopathology showed that the mass (Figures 3 and 4) consisted of numerous eosinophils, epithelioid macrophages, lymphocytes, plasma cells, fewer neutrophils and mast cells. Multinucleated cells were not observed. The background consisted of many reactive fibroblasts, with surrounding and separate bundles of collagen, and many variable-sized blood vessels lined by reactive endothelium (granulation tissue). Multifocally, collagen fibers were hyalinized but eosinophilic degranulation around collagen bundles (‘flame figures’) were absent. Multifocally, many extravasated erythrocytes were also present. No evidence of parasitic, bacterial or fungal agents was found after serial sections examined with special stainings (Grocott Gomori methenamine silver, periodic acid Schiff and Gram). Examination of the surgical margins showed that there was approximately 2 3 mm of normal tissue surrounding the mass. Mast cell tumor and eosinophilic rich T-cell lymphoma were excluded by histochemical
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Figure 3. Well-demarcated granulomatous nodule attached to the cartilage of the tracheal wall (hematoxylin and eosin staining, bar D 500 mm).
(toluidine blue) and immunohistochemical (IHC against CD3 and CD79a) stainings. Following suture removal 10 days after surgery, prednisone (prezolon, Nycomed, Greece) was administered for a total of 30-day period at a low immunosuppressive dose (0.5 mg/kg BW, twice daily, per os) for 20 days, and tapered gradually over a 10-day period in order to prevent
recurrence of the condition. No anti-parasitic treatment was given except of milbemycin (interceptor, Novartis Animal Health, Switzerland) at a dose of 11.5 mg, once a month, per os. Clinical signs subsided just after surgery. Upon reevaluation 45 days after surgical excision of the mass, the owner reported that all clinical signs had resolved, even
Figure 4. Eosinophils were the predominant population in the mass (hematoxylin and eosin staining, bar D 125 mm). Inset: eosinophils – bilobular cells with eosinophilic granules.
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during exercise. No abnormalities were evident on physical examination. Lateral radiographs of the neck and thorax were unremarkable. Endoscopic re-evaluation 46 days after surgery revealed a small in size (less than 0.7 cm in height and 0.5 cm in width), whitish, mushroom-shaped tracheal mass occluding approximately 5% of the tracheal lumen, located 1 2 cm caudally to the anastomosis (Figure 5). Histopathologic examination of tissue specimens obtained endoscopically demonstrated a recurrence of tracheal eosinophilic granuloma. Due to the dog’s good clinical condition and the size of the mass, conservative management was elected consisting of prednisone (1 mg/kg BW, twice daily, per os) for 30 days. The steroid dosage was tapered gradually over a 40-day period; steroids were given for 70 days in total. Additionally, cefuroxime (zinadol, GlaxoSmithKline, Greece) at the dose of 25 mg/kg BW, twice daily, per os was prescribed for the initial 30 days of steroid administration. At the end of the steroid therapy, a third endoscopy was performed revealing remission of the mass. One year after the third endoscopic evaluation, a fourth endoscopy revealed a normal tracheal lumen. Two years after the last endoscopic examination, follow-up clinical evaluation of the dog showed no evidence of abnormal findings. The aim of this study was to describe the diagnostic approach, management and outcome of an intraluminal tracheal eosinophilic granuloma in a Siberian husky dog which recurred after a combination of surgical excision and steroid administration, but eventually resolved with higher doses of steroids. Tracheal intraluminal neoplastic or hyperplastic masses associated with primary or metastatic tumors (e.g. canine mast cell tumor, T-cell lymphoma), parasitic granulomas, abscesses due to a foreign body, inflammatory mucosal polyps and trauma usually cause nodular lesions in the dog, resulting in tracheal obstruction (Beck et al.
Figure 5. Endoscopic view of a small-sized, intraluminal tracheal mass (eosinophilic granuloma) 45 days after surgical excision of a pre-existing mass. The location of the relapsed mass was approximately 1 2 cm caudal to the anastomosis.
1999; Clifford & Sorenmo 2004; Sherding 2004). Hyperplastic tracheal nodules of unknown origin (De Rick et al. 1977) or related to amyloidosis (Besancon et al. 2004) have also been reported. The magnitude of clinical signs correlates with the severity of tracheal obstruction. Affected dogs may present with a variety of upper airway obstruction signs (Clifford & Sorenmo 2004). Histopathology remains the cornerstone of diagnosis while plain radiography, computerized tomography and endoscopy along with the severity of clinical signs, size of lesion and its location determine the treatment modalities (Clifford & Sorenmo 2004). A list of differentials for the tracheal mass in our case included a tracheal foreign body, tracheal neoplasm, parasitic granuloma (Spirocerca lupi, Onchocerca spp., Oslerus osleri) and eosinophilic granuloma. Histopathologic examination of biopsy specimens taken at tracheoscopy and/or surgical excision was required to confirm the diagnosis. Although the trachea is an unexpected site for canine spirocercosis, S.lupi infection was included among differentials since it is endemic in our region (Haralabidis et al. 1988). A case of obstructive granulomatous tracheitis caused by Onchocerca spp. in a dog has also been described in the past (Papaioannou et al. 2004). Although not seen in our country, the most common nematode to infect the trachea of young dogs is O.osleri (Sherding 2004). Typical lesions are localized in the distal trachea and tracheal bifurcation, forming granulomatous nodules. The most reliable way to diagnose O.osleri infestation is by direct visualization of the parasitic granuloma and larvae during tracheoscopy (Sherding 2004). However, in the present case, a bilobed and almost linear mass was detected in the proximal part of the trachea and no worms were visible. Fecal examination for parasitic infestation and histological and parasitological examination of multiple sections of the tracheal nodule failed to reveal any parasitic agent. Histomorphologically, the lesions seen in this dog closely resembled those described in the skin and oral mucosa of Siberian huskies affected by eosinophilic granulomatous disease (Potter et al. 1980; Scott et al. 2000). Finally, tracheal intussusception was easily excluded at endoscopy (Kim et al. 2012). Canine eosinophilic granuloma is a not very common, benign disorder of unknown etiology that manifests as nodular or plaque-like lesions, predominantly on the skin, tongue or in the oral cavity (Potter et al. 1980; Scott et al. 2000). Other less common locations described are lungs (Confer et al. 1983; Neer et al. 1986; von Rotz et al. 1986; Katajavuori et al. 2012), nasal planum (Da Silva Curiel et al. 1988), external ear canal (Poulet et al. 1991), eyelids (Vercelli et al. 2005), gastrointestinal tract and liver (Brellou et al. 2006). Tracheal eosinophilic granuloma has been rarely described in the literature and only two dogs have been reported: a 10-year-old female, German shepherd (Brovida & Castagnaro 1992) and a 2-year-old male, Siberian husky (Ramirez & Hawkins 1998). Canine tracheal eosinophilic granuloma has been previously reported in just two dogs (Brovida & Castagnaro 1992; Ramirez & Hawkins 1998). The case presented herein is the second to be reported in a Siberian husky.
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Veterinary Quarterly Breed predilection in the latter concerning the oral form of the disease has been described (Madewell et al. 1980; Potter et al. 1980). Several pathogenetic mechanisms have been suggested, including vasculitis, microangiopathy, disorders of fibrinolysis or phagocytic function, an immunologic hypersensitivity reaction to an unidentified antigen or to a grass awn or to a parasite, or heritable predisposition secondary to an unknown injury (Scott et al. 2000; Katajavuori et al. 2012). A possible correlation with bacterial, fungal, parasitic or viral infections appears unlikely (Madewell et al. 1980; Scott et al. 2000). As in the previous cases described, the cause could not be determined; however, it may have resulted from a primary unknown injury with secondary immune-mediated inflammatory reaction. Nevertheless, secondary (reactive) tissue eosinophilia where eosinophils migrate to a specific organ due to a reactive process (helminth infection, allergic disease, drug reaction, chronic inflammatory disorder, neoplastic condition, etc.) could lead to local eosinophil accumulation and tissue remodeling (Valent et al. 2012). A high incidence of eosinophilic granulomatous disease has been noted among Siberian huskies and Cavalier King Charles spaniels. The disease has been attributed to a genetic predisposition (Madewell et al. 1980; Potter et al. 1980; Brodal et al. 1996). However, there is plenty of information to indicate that genetic traits modify the risk and nature of hypersensitivity reactions (Valent et al. 2012; Gotlib 2014). In human medicine, pulmonary eosinophilic granuloma, also known as pulmonary Langerhans cell histiocytosis (PLCH), is part of a complex group of diseases known as Langerhans cell histiocytosis (Hoover et al. 2007). The pathogenesis and etiology of PLCH remain unknown, but it seems to be common among smokers (Friedman et al. 1981), probably due to an inflammatory reaction of the lung tissue to the components of cigarette smoke (Suzuki et al. 2004). Pulmonary Langerhans cell histiocytosis rarely occurs as a solitary nodule (Khoor et al. 2001). Fridlender et al. (2005) described a unique case of PLCH in which the sole manifestation of the disease in a patient was a partially obstructing tracheal mass, which responded well to local excision. Although such a syndrome has not been described in a dog, the present case appears similar to their case. Canine tracheal mast cell tumor and eosinophilic rich T-cell lymphoma of the trachea had to be differentiated from tracheal eosinophilic granuloma due to their similar microscopical appearance following routine histological staining. Advanced histopathological techniques (toluidine blue and IHC against CD3 and CD79a) were able to differentiate those conditions from tracheal eosinophilic granuloma. The poor prognosis of the above-mentioned neoplastic lesions promoted the necessity of their differentiation, since they demand a different post-surgical management (Fox and King 2002). The absence of an increased number of circulating eosinophils was consistent with the case reported by Brovida and Castagnaro (1992). Some cases with marked peripheral eosinophilia could suffer from inflammatory or hypersensitivity disorders (German et al. 2002), but in our
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case, a disorder of this origin was not confirmed. In contrast, Ramirez and Hawkins (1998) reported a case displaying mild eosinophilia where a second eosinophilic lesion was detected in the dorsal part of nasopharynx. Similarly, Lillieh€o€ok and colleagues (2000) mentioned that most cases of eosinophilic lesions in any part of the gastrointestinal tract showed marked eosinophilia. Most canine eosinophilic granulomas have responded to glucocorticoid therapy (Potter et al. 1980; Madewell et al. 1980; Brovida & Castagnaro 1992; Vercelli et al. 2005). Prednisone at 0.5 to 2.2 mg/kg BW orally once daily, or spread over a three- to four-week period, is usually adequate to control clinical manifestations of the granulomas. However, spontaneous remission has occasionally been reported (Madewell et al. 1980; Potter et al. 1980; Ramirez & Hawkins 1998). The case described in this report, similar to the case described by Ramirez and Hawkins (1998) and unlike the case presented by Brovida and Castagnaro (1992), did not completely respond to surgical excision and the low-dose corticosteroid adjunctive treatment prescribed thereafter. The relapsed nodule responded well to increased steroid doses. This emphasizes the necessity for the administration of steroids in immunosuppressive doses at the initial pharmaceutical management. Endoscopic re-evaluation of such cases is crucial because, as in this case, the lesion can recur despite surgical excision and corticosteroid treatment. Also, unlike the case of Ramirez and Hawkins (1998), no recurrence of clinical signs was observed after discontinuation of the second, higher dose of oral anti-inflammatory therapy probably due to the small size of the second granuloma at the time of re-evaluation; thus, it may be suggested that all cases of tracheal eosinophilic granulomas should be concurrently treated with immunosuppressive doses of corticosteroid regardless of clinical signs; treatment should not rely solely on surgical excision. Moreover, response to glucocorticosteroid therapy (consisted of inhibition of eosinophil growth and function) in reactive tissue eosinophilia is not only dose-dependent but correlates well with the number of glucocorticosteroid receptors detected upon eosinophils (Valent et al. 2012). The duration of corticosteroid therapy must be determined according to endoscopic findings. Use of other immunosuppressant agents to avoid possible side effects of corticosteroid therapy, which are not unlikely in large breed dogs, were not elected in this dog due to lack of data regarding their effectiveness in cases of tracheal eosinophilic granuloma. Further studies are needed to prove if such agents are beneficial or not as a replacement therapy. Endoscopic re-evaluation of the tracheal lumen could detect a recurrence of the lesion at its early stages before any clinical signs are apparent. The fact that no clinical evidence of the disease was apparent two years after the last endoscopic examination does not completely rule out the possible presence of a new small-sized tracheal nodule that had no clinical signs. Another endoscopic evaluation at this time point would have been ideal in this patient. Despite the fact that surgical resection of the tracheal rings is considered an invasive option for removal of a granuloma, this was elected in our patient because of its
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critical condition and the failure of cytology to contribute to a definitive diagnosis. Non-invasive alternatives include laser ablation which usually addresses to pedunculated tumors, endotracheal stenting only as a palliative method, etc. (Shapshay 1987; Rebeiz et al. 1990; Freitag & Darwiche 2014). Moreover, we cannot rule out the possibility that if immunosuppressive prednisone was initiated just after the first endoscopy, eventually surgery would have been avoided. In Siberian huskies, canine eosinophilic granulomatous disease has been previously described to affect the skin, oral cavity, gastrointestinal tract, liver, eyelid and tracheal lumen (Madewell et al. 1980; Potter et al. 1980; Scott et al. 2000; Vercelli et al. 2005; Brellou et al. 2006; Lyles et al. 2009). The oral form of the disease prevails and lesions can be located both in lingual or palatine tissue (Ramirez & Hawkins 1998). Histopathologic findings in our case resembled those described by others for the oral cavity and skin of Siberian huskies, supporting the view that this breed is predisposed to the development of eosinophilic granulomas (Johnson 1992; Brellou et al. 2006). In conclusion, it seems that long-term re-examinations should be implemented in cases of canine tracheal eosinophilic granulomatous disease treated with surgery and corticosteroid treatment for early detection of a relapse. Moreover, in the case of recurrence, higher doses of corticosteroid treatment should be attempted to achieve complete resolution.
Disclosure statement No potential conflict of interest was reported by the authors.
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