Improved ventilatory function associated with relief of chest pain in a patient with Kartagener's syndrome treated by subcutaneous analgesia

Abstract

Kartagener’s syndrome was defined in 1933 as a distinctive clinico-pathological entity characterized by a triad of signs: situs inversus, bronchiectasis and paranasal sinusitis. The condition is a part of the syndrome of primary ciliary dyskinesia, where 50% of the patients have a situs inversus totalis. In primary ciliary dyskinesia, inflammation is not restricted to the ciliated conducting airways, but also occurs in the alveolar space. Repetitive episodes of pleuritic chest pain are a feature of patients with bronchiectasis and middle lobe syndrome. Pleuritic chest pain can occur in up to 21% of patients with bronchiectasis and 19% of patients with middle lobe syndrome and characteristically occurs in an area overlying a bronchiectatic lobe. This presentation can be confused with other conditions, such as pulmonary embolus. However, the natural history of pleuritic chest pain and its response to analgesia in these conditions is poorly described. We report a patient with Kartagener’s syndrome with intractable pleuritic chest pain, in whom peripheral nerve stimulation with subcutaneous electrodes led to a dramatic therapeutic response. Case report