Grand mal seizures: an unusual and puzzling primary presentation of ruptured hepatic hydatid cyst

Abstract

We report a case of hepatic hydatidosis where the first clinical manifestations, generalized seizures after minor head and abdominal trauma, and delayed anaphylaxis, made the primary diagnosis difficult. Severe anaphylaxis has been reported as initial presentation of quiescent hepatic hydatidosis. In endemic areas, the diagnosis must be carefully ruled out in patients experiencing abrupt anaphylactic shock of uncertain etiology. The occurrence of unexplained vascular collapse after minor abdominal trauma in a patient originating from an endemic area should prompt the diagnosis and urgent treatment should be initiated; firstly emergency management of the anaphylactic shock and later, surgical treatment of the cysts.

Disseminated cerebral hydatid disease (multiple intracranial echinococcosis)

“…We hypothesise that leakage from a hepatic cystic echinococcosis cyst caused systemic embolisation of its contents through a patent foramen ovale and into the cerebral circulation, resulting in multiple cerebral emboli. An alternative explanation is an anaphylactoid reaction following hepatic cyst rupture and systemic antigen circulation, as Meyer et al had described in the case of a 5-year-old child 11. Nevertheless, to our knowledge, this is the first case of intracranial cystic echinococcosis presenting as acute embolic infarct in an adult.…”

An Analysis of Possible Mechanisms of Unexpected Death Occurring in Hydatid Disease (Echinococcosis)

“…Either of these outcomes may be associated with a fatal outcome. Seizures are not however specific for central nervous system disease and have also been caused by hepatic hydatid cyst rupture with anaphylaxis (8). Intracerebral mass effects may be particularly significant if cysts are located within the brainstem (29).…”

“…A variety of clinical manifestations may occur associated with obstruction of the biliary tract, bronchi, renal outflow tract, and cerebrospinal fluid pathways. Antemortem symptoms and signs may therefore include jaundice, urticaria, abdominal pain, biliary colic, cough, dyspnea, hemoptysis, and generalized seizures (6–8). Scolices may be vomited (hydatid emesis) or passed in the feces (hydatid enterica).…”

“…Sudden death is usually due to anaphylaxis (in 20% of cases) following rupture of a cyst and release of highly antigenic hydatid scolices (9). Rupture tends to involve cysts within the liver and less often the heart, and may be precipitated by relatively minor chest or abdominal trauma or by increased pressure within the cyst (3,8,10). Between 1 and 8% of infected individuals may suffer cyst rupture into the peritoneal cavity.…”

Parasitic disorders

“…One study considered the hydatid cyst to be one of the most epileptogenic lesions (Arseni and Marinescu, 1974). Recent studies found partial and generalized seizures, especially in children (Meyer et al, 2006;Abderrahmen et al, 2007). Another retrospective study revealed early postoperative epileptic seizures (first 10 days, usually during the first 48 hours) in 6% of surgical patients.…”