Introduction

Fibrous dysplasia of the skull and facial skeleton is not uncommon. It usually manifests with swelling of cheek and proptosis in adolescents₁. We here report a case of isolated fibrous dysplasia of the head of the middle turbinate in an adult female. It is the first description of this kind in the English literature.

Case report

A 34 year old female presented to the out patient dept of Otolaryngology, Head & Neck Surgery, Postgraduate Institute of Medical Education & Research, Chandigarh, India with complaints of recurrent unilateral right sided purulent nasal discharge and obstruction not responding to medical management. Examination revealed enlarged anterior end of the middle turbinate on the right side blocking the ostium of the maxillary sinus. It was not shrinking on application of the vasoconstrictor. A probable diagnosis of the concha bullosa of the middle turbinate was kept and a non contrast computed tomogram (NCCT) of the paranasal sinus was carried out showing ground glass appearance of the affected area (Fig 1,2). Other paranasal sinuses were normal. The patient was subjected to endoscopic resection of the head of the middle turbinate involved under local anesthesia. Histopathology showed it to be fibrous dysplasia. The patient is on regular follow up for the last 10 months and is asymptomatic for any recurrence or further episodes of sinusitis.

Figure 1

Figure 1: Coronal CT of paranasal sinuses showing the lesion of the middle turbinate

Figure 2

Figure 2: Axial CT of the paranasal sinuses showing the lesion.

Discussion

Fibrous dysplasia of the facial skeleton usually involves the ethmoid and maxillary sinuses presenting with proptosis and cheek swelling. It affects the adolescents and presentation in adult age is uncommon₁.

It was first described by Lichenstein in 1938₂. Amongst facial skeleton, it most commonly affects the area of the first molar of maxilla presenting with dental malocclusion, gum, palatal and cheek swelling. Next in the order is the involvement of ethmoid-sphenoid complex presenting with recurrent sinusitis, nasal obstruction and proptosis₃.

It is a developmental anomaly of idiopathic origin affecting the precursor of the bone and affects the osteoclastic differentiation and maturation₄. It can be associated with endocrine disorders₅. It can be monostotic, polyostotic or disseminated with extra skeletal manifestations as seen in Albright syndrome₆.

Isolated involvement of the middle turbinate is rare. The literature mentions three reports of middle turbinate fibrous dysplasia associated with involvement of adjacent ethmoid bone along with either endocrine disorder or Widal syndrome with only one report from the English literature₇,₈. Our case was adult female having the fibrous dysplasia of the head of middle turbinate with rest of the sinuses and bones normal and with no associated disorders.

CT findings of the case were typical for the disease₉ and the management done was surgical resection endoscopically.

Correspondence to

Dr. Ashok K. Gupta Professor Dept. of Otolaryngology and Head & Neck Surgery Postgraduate Institute of Medical Education & Research, Chandigarh-160012,India TEL : 0172-2747585-94 Ext. 6763, Fax : +91-172-2744401,745078 E_mail : akgpgi@yahoo.com