A Case Report published by Dr. Mamata rani Rout in Journal of Evolution of Medical and Dental Sciences/ Volume 2/ Issue 35/ September 2, 2013.
ABSTRACT: Sinonasal tract is a complex anatomical site with a large list of possible diagnoses. Most common ones being sinonasal polyposis and papillomas. Occasionally there may be mass lesions that are difficult to conclude. This case report describes one such case where it was difficult to conclude.
INTRODUCTION: The sinonasal tract is home to a dizzying array of reactive, benign neoplastic, and malignant entities, and differentiating these can be difficult owing to the limited nature of biopsies from this site that are encountered in E.N.T practice. Most of these consist of inflammatory polyps, papillomas, and squamous cell carcinomas. Other diagnoses such as salivary type tumors, olfactory neuroblastomas, and poorly differentiated carcinomas may occasionally be seen but are familiar to most E.N.T surgeons. The diagnosis becomes more complicated when a biopsy or polypectomy yields a low-grade glandular proliferation. In such circumstances, the diagnosis should include hamartomas.
CASE REPORT: A 29-year-old female patient presented with long-standing nasal obstruction.
Anterior rhinoscopy showed a pinkish grape-like mass which was insensitive to touch and did not
bleed on touch. Diagnostic nasal endoscopy showed a pinkish mass in the left middle meatus which was arising from the maxillary sinus and was extending towards the choana. It was suggestive of sinonasal polyposis.
The patient underwent endoscopic polypectomy with middle meatal antrostomy and the tissue sent for histopathological examination was suggestive of sinonasal polyposis but after one year patient presented with sudden onset of bleeding from the left nasal cavity and nasal obstruction on the same side. On diagnostic nasal endoscopy a pink polypoidal mass, friable, bleeds on touch, covered with yellowish cheesy debris seen completely occupying the left nasal cavity. Computed tomography showed heterogenous opacity with calcified spicules occupying the left maxillary sinus and the osteomeatal complex extending to the left nasal cavity. A provisional diagnosis of fungal rhinosinusitis was made.
The patient underwent endoscopic debridement. Profuse haemorrhage was encountered during
debridement. The lesion showed friable mass with yellowish, cheesy, and foul-smelling debris.
Complete clearance of the mass from the nasal cavity and maxillary sinus achieved.
Histopathological examination showed well-defined fibro-collagenous tissue capsule with
areas of haemorrhage and a large number of vascular spaces of varying sizes lined by endothelial cells. The lumen showed haemorrhage with fibrinoid necrosis at places. Features were suggestive of Vascular Hamartoma.
DISCUSSION: Vascular hamartomas are defined as disorganized and excessive proliferations of
vascular tissue which, due to their limited growth, are considered developmental malformations
rather than true neoplasms. Usually, the use of the term ‘‘hamartoma’’ implies the presence of the
lesion at birth, with subsequent growth paralleling that of the animal but ceasing with maturity.
Vascular hamartomas are classified as capillary telangiectasis, cavernous angiomas, and
arteriovenous or venous malformations. The clinical significance of hamartomas is generally
attributed to their potential to spontaneously bleed. Histochemistry and immunohistochemistry
were used as additional tools to further define vessel structure and the relationship between the
vessels of each hamartoma with the surrounding structures. Hamartomas are usually congenital and simultaneously grow along the rest of the body. Once they reach their adult size they do not extend to involve more tissue unless there is trauma, infections, oedema, inflammation and filling of new vascular channels. They are common in lung, liver, kidney, spleen and intestine but are extremely rare in the upper aerodigestive tract. Dicarlo et al reported a respiratory epithelial adenomatoid hamartoma in the maxillary sinus. Fleming etal reported a case of sinonasal sero mucinous hamartoma. Head and Neck surgeons should be aware of this pathological entity as a differential diagnosis for inverted papilloma and adenocarcinoma, in order to avoid unnecessary aggressive surgery. On the other hand misinterpretation as sinonasal polyposis may result in inadequate treatment therefore complete removal and compulsory histopathological examination of all the masses of nasal cavity should be followed as a strict protocol.
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