A 3-year-old girl is brought to the emergency department due to a first-time seizure. The patient was watching television 30 minutes earlier when her left arm began to twitch, which progressed to twitching in all 4 extremities and loss of consciousness that lasted a minute. Medical history is notable for receptive and expressive language delays. Vital signs are normal. Examination shows a sleepy but arousable toddler lying on the bed. An erythematous patch is present on the right forehead and upper eyelid, which her parents say has been present since birth. There is a 5-cm, hyperpigmented macule with an irregular border on the abdomen. Skin examination is otherwise normal. Visual field examination reveals a left homonymous hemianopia. Neurologic examination is otherwise unremarkable. Which of the following is the most likely diagnosis in this patient?
Sturge-Weber syndrome | |
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This patient has new-onset seizure and facial port-wine stain, findings concerning for Sturge-Weber syndrome (SWS). SWS is a neurocutaneous disorder characterized by a capillary malformation (nevus flammeus, or port-wine stain) in the distribution of the first or second branches of the trigeminal nerve and an associated leptomeningeal capillary-venous malformation affecting the brain and eye.
MRI of the brain with contrast detects the intracranial vascular malformation and is diagnostic. Treatment involves controlling seizures and reducing intraocular pressure. Laser therapy may also help reduce the appearance of the port-wine stain.
(Choice A) Infantile hemangioma is a raised, vascular lesion composed of small capillaries that presents at or shortly after birth with a bright red papule/nodule. Hemangiomas initially proliferate in infancy before spontaneous involution occurs. Seizures and neurologic deficits are not seen.
(Choice B) Neurofibromatosis type 1 (NF1) is an autosomal dominant neurocutaneous disorder that can cause intellectual disability and seizures. Multiple, large (>0.5 cm) café au lait macules are typical; this patient's single lesion is seen in up to one-third of the general population and does not support an NF1 diagnosis. Moreover, additional cutaneous manifestations of NF1 include freckling and neurofibromas, not a nevus flammeus.
(Choice C) Neurofibromatosis type 2 (NF2) is an autosomal dominant neurocutaneous disorder associated with bilateral vestibular schwannomas. Cataract is the ophthalmic condition most commonly associated with NF2, and skin manifestations include cutaneous plaques and tumors (not port-wine stains).
(Choice E) Tuberous sclerosis is a neurocutaneous disorder that often presents with seizures and intellectual disability. Typical skin findings include hypopigmented ash-leaf spots, angiofibromas (malar erythematous papules), and shagreen patches (flesh-colored plaques on the back).
Educational objective:
Sturge-Weber syndrome is a neurocutaneous disorder characterized by a capillary malformation (port-wine stain) along the trigeminal nerve distribution and leptomeningeal capillary-venous malformations that affect the brain and eye. Seizures, intellectual disability, and visual field defects can occur.