QJM Advance Access originally published online on April 29, 2008
QJM 2008 101(9):745-746; doi:10.1093/qjmed/hcn058
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Severe disseminated hydatid disease successfully treated medically with prolonged administration of albendazole
Department of Internal Medicine, University of Cape Town, Cape Town, South Africa
Department of Medicine, Frere Hospital, Walter Sisulu University, Cape Town, South Africa.
email: ntobekon{at}yahoo.com
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Case presentation |
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A 19-year old gentleman presented to our medical department, referred by a GP with an assessment of minor stroke. The patient had a pneumonectomy for a hydatid cyst of the right lung 4 years previously. After refusing repeat surgery for excision of hepatic hydatid cysts, he was lost to follow-up. On this occasion, he complained of right sided weakness with decreased visual acuity bilaterally for 3 days. The neurological deficit had been improving since the onset (and resolved completely 2 days later). There was a history of a single generalized tonic-clonic seizure about 2 months previously and, also, episodes of syncope associated with impairment of the level of consciousness, but with no abnormal movements noted, followed by confusion on waking. He also described a chronic lingering headache, dizziness not related to posture, as well as progressive deterioration of his academic performance over the past year.
Clinically, he was lucid, apyrexial and with no peripheral stigmata of HIV infection. CNS examination revealed a motor deficit on the right side with 4/5 power in the right arm and leg, proximally and distally. Power on his left side was normal. There was no meningism. No abnormalities of tone, reflexes or sensation were noted, and there were no features to suggest raised intra-cranial pressure.
A chest radiograph revealed bilateral circular, nodular pulmonary infiltrates, as well as calcified rounded structures in his liver. Lab results revealed a normal biochemistry, liver function tests and full blood count (notably with a normal eosinophil count). The result of a HIV test was negative. Serology for Ecchinococcus was positive for both IgM and IgG. CT imaging of the chest and abdomen revealed multiple hydatid cysts in lungs, mediastinum and the presence of both cysts and calcified foci in the liver. CT scan of the brain showed multiple hydatid cysts, compressing most of the brain parenchyma, with mass effect and compression of the lateral horns of the ventricles. No hydrocephalus or extracranial collections were noted (Figure 1).
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After neurosurgical consultation, it was decided that only medical therapy could potentially benefit the patient. He was treated with 6 months of oral albendazole. A repeat CT scan at the end of 6 months revealed complete resolution of hydatid cysts in his body, most remarkably, in his brain. His initial symptoms had all resolved, and his academic performance was notably improving. No further seizures have been noted at 2 year follow-up (Figure 2).
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Discussion |
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Ecchinococcosis is caused by infection with the metacestode stage of the tapeworm Echinococcus. Humans do not play a role in the biological cycle of E.granulosus, and happen to be accidental hosts.1 Symptoms depend on organs affected, cyst size, organ compression, obstruction to blood and lymphatic flow, as well as cyst rupture. Additionally, CNS involvement may present with seizures, abnormal mentation, features of raised intra-cranial pressure and spinal cord compression. Brain involvement only occurs in <2% of patients infected with E.granulosus.2 Diagnosis rests on a combination of serology and radiological imaging.3 Treatment may be medical or surgical.4 Benzimidazole carbamate compounds (albendazole and mebendazole), have, for a long time, remained the cornerstone of medical therapy for echinococcosis.1 We note that increased toxicity should not be a major concern with prolonged courses of albendazole therapy.4,5 Our case report demonstrates that, in cases of disseminated disease with multiple organ involvement, or involvement of organs like the brain, where surgical intervention is clearly hazardous, a prolonged course of albendazole therapy is appropriate, and may prove very beneficial to the patient.
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References |
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1. McManus DP, Zheng W, Li J, Bartley PB. Echinococcosis. The Lancet (2003) 362:1295–1304.
2. Aydin MD, Ertek M, Yazgi H. Multiple infected cerebral hydatid cysts treated with Albendazole. Turk J Med Sci (2003) 33:393–395.
3. Tuzun M, Altino RN, Arda IS, Hekimoglu B. Cerebral hydatid disease: CT and MR findings. Clin Imaging (2002) 26:353–357.[CrossRef][Web of Science][Medline]
4. Rigano R, Ioppolo S, Ortona E, et al. Long term serological evaluation of patients with cystic echinococcosis treated with bendimidazole carbamates. Clin Exp Immunol (2002) 129:485–492.[CrossRef][Web of Science][Medline]
5. Franchi C, DiVicco B, Teggi A. Long term evaluation of patients with hydatidosis treated with benzimidazole carbamates. Clin Infect Dis (1999) 29:304–309.[Web of Science][Medline]
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