A case of Osler-Weber-Rendu disease with brain abscess; the mechanism of the formation of brain abscess and its treatment in Osler-Weber-Rendu disease

A 54 year-old man, who had a hereditary hemorrhagic telangiectasia (Osler-Weber-Rendu disease; O-W-R) accompanied by pulmonary arteriovenous fistulas (PAVFs) and congestive heart failure, developed seizure, right hemiparesis and dysphasia. A brain CT scan revealed a cystic lesion with perifocal edema in left frontoparietal lobe. A contrast enhanced CT scan showed a ring-like enhancement. Dynamic CT scans disclosed that the ring in the cortical side was enhanced more thickly than that in the ventricular side. Considering the severity of the cardio-pulmonary condition, and the deep location of the abscess, we performed an echo-guided aspiration and drainage of the abscess under local anesthesia. No bacteria were demonstrated in the culture of the contents of the abscess. After the surgery, the right hemiparesis and dysphasia were much improved and a CT scan showed the marked reduction of the abscess. However, around eight days after the surgery, the patient showed severe pleural effusion due to progressive heart failure and died on the 11th postoperative day. Autopsy disclosed a shrunken brain abscess, multiple cerebral infarction, multiple PAVFs and severe constrictive pericarditis which was regarded as the cause of death in the patient. In this report, we presented the therapeutic advantage of echo-guided surgery for the treatment of brain abscess in a high-risk patient. We also discussed the mechanism of the formation of brain abscess in patients of O-W-R disease by reviewing published cases.     

The rational use of antibiotics in the treatment of brain abscess

The Working Party was instituted to investigate the rationale of therapeutic antibiotic usage in patients with brain abscess and to make recommendations for current practice. A systematic review of English language publications on brain abscess over the last 25 years was carried out using electronic databases and secondary sources, and data were evaluated. Few publications were identified where the microbiological procedures were adequately described and many authors continue to report sterile pus in a proportion of cases. The vast majority of reports were retrospective neurosurgical assessments in which details of laboratory procedures and antibiotic regimens were missing. There are no published reports of controlled clinical trials or comparative therapeutic studies. The recommendations made by the Working Party are based on relevant published information and the expertise of Working Party members. Recommendations vary according to the location of the abscess which reflects the likely source of the infection and therefore the bacterial types most likely to be present in aspirated pus. Bacteria with multiple resistance to antimicrobial agents do not feature significantly in cases of brain abscess.     

Surgical treatment of brain abscess

The author reviews the methods of surgical treatment of brain abscess. All methods are grouped into the following six categories: tube drainage, marsupialization, the migration method of Kahn, tapping only, aspiration, and excision. At present, only two methods are used: aspiration and excision. Antibiotics and computed tomographic (CT) scanning play important roles in the surgical treatment of a brain abscess. The use of CT scanning is invaluable in the management of brain abscess for early and exact localization, assessment of the adequacy of operation, and sequential follow-up.     

A case of amebic brain abscess

Amebic brain abscess is a rare and usually fatal complication of Entamoeba histolytica infections. We successfully treated a patient with this infection accompanied by brain, liver and pulmonary abscesses. Treatment consisted of administration of metronidazole through a nasogastric tube, and drainage of the brain abscess. A 51-year-old male patient presented with a two-week history of fever, severe back pain, and chest pain. Liver and pulmonary abscesses were drained upon admission. Biopsy of the colon showed infection by Entamoeba histolytica. After oral administration of metronicazole, the patient became somnolent, and neurological examination showed left side weakness. CT scan and MRI of the brain disclosed an abnormal lesion in the right basal ganglia. He was then transferred to our department, where stereotactic aspiration with drainage of the brain abscess was performed. The abscess was filled with reddish purulent material. Treatment with metronidazole (1,200 mg daily) for 18 days resulted in almost complete resolution of the intracerebral lesion and survival of the patient without any neurological deficits. His general condition improved dramatically, and he was discharged from our hospital 2 months later. Only 14 cases with amebic brain abscess associated with Entamoeba histolytica infection who recovered after treatment have been reported. We suggest amebic brain abscess should be taken into consideration for patients with brain abscess with a history of dysenteric illness especially since early diagnosis and aggressive management is likely to result in a cure.     

Nocardial brain abscess: case report

A 25-year-old man, who was slightly immunosuppressed, presented headache and right motor weakness due to multiple brain abscesses disseminated from lung abscess. They were diagnosed, by bacteriological examination, as nocardial brain abscesses (nocardia asteroides) 4 weeks after the first operation. In spite of delay in the diagnosis, he was relieved by operations (three times) and chemotherapy including high doses of Sulfamethoxazole-Trimethoprim. He was eventually discharged. We stress the necessity of early diagnosis and the efficacy of Sulfamethoxazole-Trimethoprim for nocardial brain abscess.     

A case of Rhodococcus equi brain abscess

We treated a patient with acquired immunodeficiency syndrome for a brain abscess caused by Rhodococcus equi, an actinomycete that usually infects the lung in immunosuppressed hosts. Rhodococcus equi brain abscess is an extremely rare lesion that has never been reported in a patient with acquired immunodeficiency syndrome. The infection was cured by lengthy therapy with multiple antibiotics after aspiration of the lesion to identify the infective organism and determine its sensitivity to antibiotics.     

Aspergillus brain abscess. A case report

The history and findings in a patient with erythroleukaemia who developed a fungal brain abscess during the agranulocytic phase of induction treatment is reported. The radiological features of fungal infection are reported, with emphasis on the importance of clinical judgement in making the diagnosis. The autopsy findings further illustrate the increasing importance of this previously very rare condition.     

A case of hemorrhage into a brain abscess

A rare case of hemorrhage into a brain abscess in a 23-year-old man is reported. The patient complained of headache and low-grade fever on February 26, 1986. Two days later, he developed right hemiparesis and right hemisensory disturbance with mild consciousness disturbance and was admitted to a local hospital. Seven days after the onset, he suddenly became semicomatose, developed anisocoria and was consequently transferred to the University Hospital. On admission, his temperature was 37.5 degrees C and neurological examination revealed semicoma, anisocoria and right hemiparesis without nuchal rigidity. Enhanced CT scan showed a high density area within an irregular ring enhancement at the left basal ganglia. At that time, malignant glioma was diagnosed and an emergency operation was performed by left frontotemporal craniectomy. During the operation blood clot was found in the posterior part of the basal ganglia. After operation, a histological examination was made and a brain abscess was diagnosed. Gram staining revealed gram-positive bacillus. By aspiration of the abscess and chemotherapy, recovery was gradually made. He was discharged with motor dysphasia and mild right hemiparesis three months later. Differentiation between abscess and malignant glioma and the cause of the hemorrhage are discussed.     

The value of different methods of treatment of brain abscess in the CT era

Summary 67 cases of brain abscess were analyzed retrospectively. As 2 comatose patients died on admission before any treatment was started, the results are based on 65 treated patients. Different methods of treatment included: total removal in 36 patients, drainage in 14, aspiration in 6 and conservative treatment in 9. Management mortality was 18,5% and was almost not dependent on the method of treatment (except aspiration) being lowest in the drainage group. The mortality was significantly higher in patients with serious impairment of consciousness on admission.Follow-up examination after 1 to 11 years was performed in 47 out of 53 discharged patients and revealed in 25 of them (53%) full recovery. 10 additional cases (21%) are independent. The best early and long term results were obtained in patients by drainage and medical treatment.Chronic epilepsy developed in 34% of patients with supratentorial lesions. The risk of epilepsy was lowest in the group of patients treated by drainage.The authors present the opinion that removal of brain abscess is necessary only in exceptional cases.     

Aspiration and drainage for a gas-producing brain abscess causing delayed bleeding from the abscess capsule--a case report]

A 65-year-old man was admitted with the complaint of gait disturbance. CT scan revealed a low density lesion in the right parietal lobe. MRI was carried out 3 days after admission, the lesion showing low intensity on T1 WI and T2 WI. Gd-DTPA enhanced T1 WI showing abnormal enhancement surround it. CT scan then revealed a gas bubble in the lesion 12 days after admission, so we diagnosed it as gas-producing brain abscess and aspiration, drainage and irrigation with antibiotics were performed. Although, the brain abscess was reduced in size after the operation, the lesion expanded again 2 weeks after the operation. MRI was performed and the lesion showed iso-intensity on T1 WI and high intensity on T2 WI. Emergent aspiration and drainage were performed and uncoagulated old-hematoma-like matter such as chronic subdural hematoma was removed. As significant neovascularization with inflammatory cells had been detected in the capsule of the brain abscess, we suspected that aspiration and drainage surgery for brain abscess may cause delayed bleeding from the capsule of the abscess. We conclude that attention should be drawn to such a complication.     

Aspects of modern brain abscess diagnosis and treatment

Summary A clinical analysis of 115 patients with brain abscesses, who were operated in our department, provide valuable information about the diagnosis and treatment of such abscesses. Even in this antibiotic era the brain abscess has not lost its significance. The diagnosis of this lesion is one of the most difficult problems in neurology, and modern approaches with highly specialized instruments are therefore of especial importance; brain abscesses today are best detected by computer tomography. Various methods of treatment are described. The preferred procedure is a combined treatment consisting of puncture of the abscess, repeated aspiration of pus, instillation of a suitable antibiotic into the abscess cavity and complete removal of the abscess capsule at a later date. The importance of this treatment scheme is demonstrated by our own observations. Several unsolved problems including the prevention and treatment of rupture into the ventricle, the occurrence of multiple abscesses and the clinical treatment of fresh phlegmonous inflammations are discussed.

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Zusammenfassung Eine klinische Analyse unserer 115 operierten Hirnabszesse ergibt für die Diagnostik und Therapie der Abszesse wichtige Anhaltspunkte. Der Hirnabszeß hat auch in der antibiotischen Ära nicht an Bedeutung verloren. Seine Diagnose gehört zu den schwierigsten Kapiteln der Neurologie. Daher haben die modernen apparativen und instrumentellen Zusatzuntersuchungen um so größere Bedeutung. Die sicherste Methode zum Nachweis eines Hirnabszesses ist jedoch gegenwärtig die Computer Tomographie. Die verschiedenen Behandlungsverfahren werden dargestellt, wobei sich aber als die heute anzustrebende Therapie eine kombinierte Behandlung durch Punktion, mehrmalige Aspiration des Eiters, Einführen eines geeigneten Antibiotikums in die Abszeßhöhle und spätere Totalexstirpation des gesamten Narbengebietes erweist. Dies läßt sich auch anhand der eigenen Beobachtungen demonstrieren. Einige ungelöste Probleme — die Verhütung und Behandlung des Ventrikeleinbruchs, das Auftreten multipler Abszesse und das Vorgehen bei frischen, noch nicht eingeschmolzenen Entzündungen — werden besonders herausgestellt.