Abstract
Lipiodol-based lymphangiography is not only a diagnostic tool for visualization of lymphatic disorders such as plastic bronchitis (PB), but also aims a therapeutic effect by embolizing lymph leakages. We performed such percutaneous lymphatic embolization for PB in a Fontan patient with proven absence of right-to-left shunt, and demonstrated important lymphatic abnormalities in the mediastinum. Shortly after the procedure, the patient developed severe convulsive seizures, revealing multiple cerebral embolisms of Lipiodol. Radiological images were impressive, yet the clinical neurological outcome was favorable. Lipiodol-based lymphography in Fontan patients with plastic bronchitis should be avoided as this subgroup is more likely to have developed lympho-pulmonary venous connections which allow systemic emboli.
Highlights
Plastic bronchitis (PB) is a rare but severe complication in patients with Fontan-circulation [1, 2]
Dori and coworkers successfully treated PB with targeted embolization of abnormal lymphatic vessels originating from the thoracic duct with centrifugal flow into the mediastinum [3, 6, 7]
We describe a similar case of a pediatric patient with plastic bronchitis after Fontan-palliation, who underwent percutaneous lymphatic embolization with Lipiodol and developed multiorgan failure due to systemic Lipiodol embolisms
Summary
Plastic bronchitis (PB) is a rare but severe complication in patients with Fontan-circulation [1, 2]. Dori et al showed that most such patients have abnormal centrifugal flow from the mediastinal lymphatics into the airways with secondary formation of bronchial casts [3]. Dori and coworkers successfully treated PB with targeted embolization of abnormal lymphatic vessels originating from the thoracic duct with centrifugal flow into the mediastinum [3, 6, 7]. They recommended inguinal lymphangiography with Lipiodol to identify the lymphatic thoracic problem, and to localize the cysterna chyli, thereby allowing its direct puncture for access to the thoracic duct. Eleven months after the procedure, he is still on prednisone and intermittent t-PA inhalations according to the protocol which was used before the procedure
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