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Answer to Case of the Week: Jun 3-Jun 10, 2010

Teenager with partial complex seizure since age 2 and developmental delay


 




CT demonstrates tram-track calcification in the left parieto-occipital lobe. MR demonstrates leptomeningeal enhancement in the same pattern and mild enlargement of the ipsilateral occipital horn of the ventricle.

Diagnosis: Sturge-Weber-Dimitri-Encephalotrigeminal angiomatosis

Sturge-Weber syndrome (SWS), also known as encephalotrigeminal angiomatosis is a neurocutaneous syndrome with angiomas of the ophthalmic and trigeminal distribution of the 5th nerve (port-wine stain) and of the leptomeninges. 

    This is due to failure of regression of embryonic blood vessels. 

 The cause of this is uncertain, but there are reports of mosaicism in certain patients with SWS    

Inheritance is sporadic. 

It occurs in about 1 in 50,000 patients and is bilateral in 15-20%.     

Symptoms include seizures, focal hemianopsia and hemiparesis,  (sometimes transient), headache and developmental delay.  

Complications include buphthalmos ( large eye) and glaucoma.    

Treatment consists of seizure control with medication and occasionally surgery, glaucoma medication or surgery, laser therapy for port-wine stain, and preventive or abortive therapy for headache.   

  Imaging demonstates the leptomeningeal angioma on radiograph or CT with tram-track calcifications.  

The enhancing leptomeningeal angioma is better seen with MR. 

 Other signs include delayed myelination, loss of brain volume, large  lateral ventricle, decreased cortical veins and enlarged choroid plexus in the ipsilateral hemisphere. 

 Patients may develop calcification of the brain parenchyma adjacent to the angioma. 

 MR spectroscopy shows elevated choline and decreased  NAA.  

Perfusion of the involved hemisphere may be altered.    

 Recognize that patients may have SWS with leptomeningeal angiomas, but no port-wine stain. In addition, patients with port-wine stain do not always have SWS.

 Classification:

Type 1: Leptomeningeal plus facial±glaucoma

Type 2: Facial only±glaucoma

Type 3: Leptomeningeal only 

TAKE HOME MESSAGE:If ipsilateral choroid plexus engorgement may be only finding in first 6 months of life.If both sides enlarged, look for bilateral involvement ( may be subtle)


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    Disclaimer: This information is intended solely for resident review of presented cases which may or may not be pathologically proven. Information is derived from a number of published sources of varying reliability and does not represent original research from the institution. It is not intended to be comprehensive and should therefore not substitute for careful review of the literature.