Open AccessA case of Kartagener's syndrome with combined aplasia of frontal and sphenoid sinuses and hypoplasia of maxillary and ethmoid sinuses
Author(s) -
Saloni Sharma,
Bhamidipaty Kanaka Durgaprasad,
P. Vijayalakshmi
Publication year - 2021
Publication title -
journal of family and community medicine/maǧalaẗ ṭib al-usraẗ wa al-muǧtamaʼ
Language(s) - English
Resource type - Journals
eISSN - 2229-340X
pISSN - 1319-1683
DOI - 10.4103/jfcm.jfcm_304_20
Subject(s) - medicine , aplasia , situs inversus , hypoplasia , paranasal sinuses , sinusitis , agenesis , primary ciliary dyskinesia , dextrocardia , bronchiectasis , sinus (botany) , anatomy , frontal sinus , surgery , lung , botany , biology , genus
Kartagener's syndrome (KS), characterized by a triad of bronchiectasis, chronic sinusitis, and situs inversus, is a subset of an autosomal recessive hereditary disorder of primary ciliary dyskinesia. We report the case of a 35-year-old male who presented with a history of intermittent episodes of productive cough, breathlessness, and cold since childhood. High resolution computed tomography of chest revealed bronchiectatic changes, dextrocardia, and right-sided aortic arch. Computed tomography (CT) scan of the abdomen revealed situs inversus. CT of the paranasal sinuses revealed combined aplasia of bilateral frontal and sphenoid sinus with sinusitis. Based on these findings, a diagnosis of KS was made. There was no complaint of infertility, which usually accompanies KS, even though an analysis of his seminal fluid revealed reduced count and reduced motility of sperms. The uniqueness of our case is that our patient was a male aged 35 years; besides, the third decade is an unusual age for presentation of combined aplasia/agenesis of bilateral frontal and sphenoid sinuses with hypoplasia of maxillary and ethmoid sinuses. Even though these findings have been reported in children and young adults, there are very few case reports of such a presentation in adults in literature.