Cystic Fibrosis
With Brain Abscess
Patricia K. Duffner, MD, Michael E. Cohen, MD
\s=b\ A 21-year-old patient with cystic fibrosis developed bilateral brain abscesses due to anaerobic Streptococcus. This rare association presents an interesting etio-
logic study. (Arch Neurol 36:27-28, 1979)
rarely encounters sepsis f\ne ^ metastatic infection in
or
patients cystic fibrosis, despite the fre¬ quent pulmonary infections suffered by these patients. The reasons for this with
not well understood. This report describes a 21-year-old man with cystic fibrosis who developed bilateral brain abscesses due to anaerobic are
Streptococcus. REPORT OF A CASE
21-year-old man was found to have cystic fibrosis at 21 months of age. He had been hospitalized many times for treat¬ ment of recurrent pneumonia and bronchiectasis. Sputum cultures had shown Staphylococcus aureus and mucoid and nonmucoid Pseudomonas aeruginosa. The patient had been relatively well until the day prior to admission, when he had a grand mal convulsion. He was taken to a local hospital where additional seizures were controlled with intravenously admin¬ A
The following day the patient was trans¬ ferred to Buffalo Children's Hospital where he was oriented but lethargic. He was afebrile. Blood pressure was 110/60 mm Hg. Pulse was 60 beats per minute. There were decreased breath sounds at the right base and extensive osteoarthropathy. No focal neurologic findings were present.
Results of funduscopic examination were normal. A roentgenogram of the chest disclosed air trapping, peribronchial thick¬ ening, and prominent hilar shadows char¬ acteristic of cystic fibrosis. Staphylococcus aureus grew from a sputum culture as well as mucoid and nonmucoid aeruginosa. Blood cultures were sterile. On the second hospital day the patient was found to have papilledema. A mild right facial paresis; hyperreflexia, right greater than left; and a right Babinsky sign were present. He Brain scan abscesses.
complained of a left-sided headache. That day he developed right adversive seizures.
A nuclide brain scan showed increased in both frontoparietal areas. The scan was compatible with bilateral brain abscess (Figure). Arteriography indicated a mass in the left parietal region with a shift to the right. A computerized axial to¬ mography (CT) scan the following day suggested diffuse increased intracranial pressure of a nonfocal nature. Despite contrast enhancement, space-occupying le¬ sions were not seen. Operation was defer¬ red because of discrepancy in neuroradiologic studies. The patient was given oxacillin, gentamicin, and chloramphenicol intravenously in full dosage. His condition gradually deteriorated, with decreasing responsiveness and pulse rate and increas¬ ing blood pressure.
uptake
showing bilateral abnormality compatible
istered anticonvulsants. There was no recent history of head trauma, drug inges¬ tion, or fever. He was not receiving anti¬ biotics. There was no past history of seizures. A lumbar puncture showed an opening pressure of 140 mm H,0, a protein level of 20 mg/dl, 2 RBCs, and 2 WBCs; his blood glucose level was 107 mg/dl, and a CSF glucose value was 53 mg/dl.
Accepted for publication April 11, 1978. From the Department of Neurology, Children's Hospital of Buffalo, State University of New
York at Buffalo, School of Medicine.
Reprint requests to Department of Child Neurology, Buffalo Children's Hospital, 219 Bryant St, Buffalo, NY 14222 (Dr Cohen).
Downloaded From: http://archneur.jamanetwork.com/ by a University of Manitoba User on 06/19/2015
with clinical
diagnosis
of brain
Five days following admission the patient developed signs of uncal hernia-
tion. Bilateral burr holes done. At depth of 3 cm, 20 ml of thick, greenish pus was aspirated from the left parietal region. Aspiration from a needle inserted through the burr hole on the right produced 5 ml of pus. Peptostreptococcus intermedius, an anaerobic Streptococcus sensitive to peni¬ cillin and chloramphenicol, grew from cultures. The patient was given sodium penicillin G and chloramphenicol intraven¬ ously in high dosage and oxacillin and gentamicin were discontinued. The patient improved but two weeks later he again developed severe headache and signs of increasing intracranial pressure. Nuclide brain scan again showed uptake bilaterally with the left frontoparietal lesion in¬ creased in size. Both sides were aspirated and 17 ml of purulent fluid was removed. Cultures were sterile. Chloramphenicol therapy was discontinued after three weeks. The patient was treated with peni¬ cillin intravenously for a total of 24 days and then with penicillin V potassium orally for 17 days until discharge. Six months later he showed no neurologic deficits but continued to have frequent seizures. were
a
COMMENT
An interesting aspect of this case that the CT scan, with enhance¬ ment, was normal despite the pres¬ ence of bilateral brain abscesses. These abscesses were easily identified on the same day by an isotope brain
was
scan.
The source for our patient's brain abscesses was most likely chronic pulmonary infection with bronchiectasis. Pleuropulmonary disease is the most common source for metastatic brain abscesses in adults and is asso¬ ciated with more than one abscess in about 40% of the cases.1 Although chronic sinusitis as a source cannot be completely discounted, the bilaterality of the brain abscesses is more sugges¬ tive of bronchiectatic lung disease. The patient's abscesses were caused by an anaerobic Streptococcus, intermedius. Anaerobic Streptococcus has been considered to be an uncom¬ mon organism in brain abscess, but the reported incidence is increasing. A recent review showed a 21% incidence of anaerobic Streptococcus isolated from brain abscesses prior to 1970 but up to 50% since then. This may be due to improved laboratory techniques. Peptostreptococcus has been isolated from brain abscesses secondary to otic, paranasal sinus, oral, and pulmo¬ nary infection, and to congenital heart disease, head trauma, and post-
operatively.Why patients with cystic fibrosis do not have a higher incidence of infec¬ tious metastatic disease than the
general population is poorly under¬ stood. The high titers of locally produced I g A3 as well as the serum
hypergammaglobulinemia (especially IgA and IgG)4 found in these patients may help explain the lack of extension of infection. High specific antibody titers to S aureus,
aeruginosa, and Haemophilus influenzae are common in cystic fibrosis.37 Another explanation for the de¬
creased incidence of metastatic infec¬ tious disease in cystic fibrosis is that many patients are maintained on broad spectrum antibiotics. Although antibiotic levels may be inadequate to eradicate the pathogens in the tena¬ cious mucus of the respiratory tree, they may be sufficient to prevent hematogenous spread of infection. This case is of interest because of the rarity of metastatic infections in cystic fibrosis. Staphylococcus aureus grew from the patient's sputum culture as well as mucoid and nonmucoid aeruginosa, but these organ¬ isms were not isolated from his brain abscesses. The immunologie factors suggested above may have prevented the spread of the total colonized flora of his lung to the brain. Alternatively, the existence of different organisms in the brain and lung raises the possi¬ bility that there was no commonality and that the association was coinci¬ dental.
References 1. Gates EM, Kernohan JW, Craig WM: Metastatic brain abscess. Medicine 29:71-98, 1950. 2. Brewer NS, MacCarty CS, Wellman WE: Brain abscess: A review of recent experience. Ann Intern Med 82:571-576, 1975. 3. Martinez-Tello FJ, Braun DG, Blanc WA: Immunoglobin production in bronchial mucosa and bronchial lymph nodes, particularly in cystic
fibrosis of the pancreas. J Immunol 10:989-1003, 1968. 4. Schwartz RH: Serum immunoglobin levels in cystic fibrosis. Am J Dis Child 11:408-411, 1966. 5. Neter E: Pseudomonas aeruginosa infection and humoral antibody response of patients with cystic fibrosis. J Infect Dis 130(suppl):132-133, 1974.
Downloaded From: http://archneur.jamanetwork.com/ by a University of Manitoba User on 06/19/2015
6. Diaz F, Mosovich LL, Neter E: Serogroups of Pseudomonas aeruginosa and the immune response of patients with cystic fibrosis. J Infect Dis 121:261-274, 1970. 7. Holbert SP, DiSant' Agnese PA, Kotch FR: Staphylococcal antibodies in cystic fibrosis of the pancreas. Pediatrics 26:792-799, 1960.
With Brain Abscess
Patricia K. Duffner, MD, Michael E. Cohen, MD
\s=b\ A 21-year-old patient with cystic fibrosis developed bilateral brain abscesses due to anaerobic Streptococcus. This rare association presents an interesting etio-
logic study. (Arch Neurol 36:27-28, 1979)
rarely encounters sepsis f\ne ^ metastatic infection in
or
patients cystic fibrosis, despite the fre¬ quent pulmonary infections suffered by these patients. The reasons for this with
not well understood. This report describes a 21-year-old man with cystic fibrosis who developed bilateral brain abscesses due to anaerobic are
Streptococcus. REPORT OF A CASE
21-year-old man was found to have cystic fibrosis at 21 months of age. He had been hospitalized many times for treat¬ ment of recurrent pneumonia and bronchiectasis. Sputum cultures had shown Staphylococcus aureus and mucoid and nonmucoid Pseudomonas aeruginosa. The patient had been relatively well until the day prior to admission, when he had a grand mal convulsion. He was taken to a local hospital where additional seizures were controlled with intravenously admin¬ A
The following day the patient was trans¬ ferred to Buffalo Children's Hospital where he was oriented but lethargic. He was afebrile. Blood pressure was 110/60 mm Hg. Pulse was 60 beats per minute. There were decreased breath sounds at the right base and extensive osteoarthropathy. No focal neurologic findings were present.
Results of funduscopic examination were normal. A roentgenogram of the chest disclosed air trapping, peribronchial thick¬ ening, and prominent hilar shadows char¬ acteristic of cystic fibrosis. Staphylococcus aureus grew from a sputum culture as well as mucoid and nonmucoid aeruginosa. Blood cultures were sterile. On the second hospital day the patient was found to have papilledema. A mild right facial paresis; hyperreflexia, right greater than left; and a right Babinsky sign were present. He Brain scan abscesses.
complained of a left-sided headache. That day he developed right adversive seizures.
A nuclide brain scan showed increased in both frontoparietal areas. The scan was compatible with bilateral brain abscess (Figure). Arteriography indicated a mass in the left parietal region with a shift to the right. A computerized axial to¬ mography (CT) scan the following day suggested diffuse increased intracranial pressure of a nonfocal nature. Despite contrast enhancement, space-occupying le¬ sions were not seen. Operation was defer¬ red because of discrepancy in neuroradiologic studies. The patient was given oxacillin, gentamicin, and chloramphenicol intravenously in full dosage. His condition gradually deteriorated, with decreasing responsiveness and pulse rate and increas¬ ing blood pressure.
uptake
showing bilateral abnormality compatible
istered anticonvulsants. There was no recent history of head trauma, drug inges¬ tion, or fever. He was not receiving anti¬ biotics. There was no past history of seizures. A lumbar puncture showed an opening pressure of 140 mm H,0, a protein level of 20 mg/dl, 2 RBCs, and 2 WBCs; his blood glucose level was 107 mg/dl, and a CSF glucose value was 53 mg/dl.
Accepted for publication April 11, 1978. From the Department of Neurology, Children's Hospital of Buffalo, State University of New
York at Buffalo, School of Medicine.
Reprint requests to Department of Child Neurology, Buffalo Children's Hospital, 219 Bryant St, Buffalo, NY 14222 (Dr Cohen).
Downloaded From: http://archneur.jamanetwork.com/ by a University of Manitoba User on 06/19/2015
with clinical
diagnosis
of brain
Five days following admission the patient developed signs of uncal hernia-
tion. Bilateral burr holes done. At depth of 3 cm, 20 ml of thick, greenish pus was aspirated from the left parietal region. Aspiration from a needle inserted through the burr hole on the right produced 5 ml of pus. Peptostreptococcus intermedius, an anaerobic Streptococcus sensitive to peni¬ cillin and chloramphenicol, grew from cultures. The patient was given sodium penicillin G and chloramphenicol intraven¬ ously in high dosage and oxacillin and gentamicin were discontinued. The patient improved but two weeks later he again developed severe headache and signs of increasing intracranial pressure. Nuclide brain scan again showed uptake bilaterally with the left frontoparietal lesion in¬ creased in size. Both sides were aspirated and 17 ml of purulent fluid was removed. Cultures were sterile. Chloramphenicol therapy was discontinued after three weeks. The patient was treated with peni¬ cillin intravenously for a total of 24 days and then with penicillin V potassium orally for 17 days until discharge. Six months later he showed no neurologic deficits but continued to have frequent seizures. were
a
COMMENT
An interesting aspect of this case that the CT scan, with enhance¬ ment, was normal despite the pres¬ ence of bilateral brain abscesses. These abscesses were easily identified on the same day by an isotope brain
was
scan.
The source for our patient's brain abscesses was most likely chronic pulmonary infection with bronchiectasis. Pleuropulmonary disease is the most common source for metastatic brain abscesses in adults and is asso¬ ciated with more than one abscess in about 40% of the cases.1 Although chronic sinusitis as a source cannot be completely discounted, the bilaterality of the brain abscesses is more sugges¬ tive of bronchiectatic lung disease. The patient's abscesses were caused by an anaerobic Streptococcus, intermedius. Anaerobic Streptococcus has been considered to be an uncom¬ mon organism in brain abscess, but the reported incidence is increasing. A recent review showed a 21% incidence of anaerobic Streptococcus isolated from brain abscesses prior to 1970 but up to 50% since then. This may be due to improved laboratory techniques. Peptostreptococcus has been isolated from brain abscesses secondary to otic, paranasal sinus, oral, and pulmo¬ nary infection, and to congenital heart disease, head trauma, and post-
operatively.Why patients with cystic fibrosis do not have a higher incidence of infec¬ tious metastatic disease than the
general population is poorly under¬ stood. The high titers of locally produced I g A3 as well as the serum
hypergammaglobulinemia (especially IgA and IgG)4 found in these patients may help explain the lack of extension of infection. High specific antibody titers to S aureus,
aeruginosa, and Haemophilus influenzae are common in cystic fibrosis.37 Another explanation for the de¬
creased incidence of metastatic infec¬ tious disease in cystic fibrosis is that many patients are maintained on broad spectrum antibiotics. Although antibiotic levels may be inadequate to eradicate the pathogens in the tena¬ cious mucus of the respiratory tree, they may be sufficient to prevent hematogenous spread of infection. This case is of interest because of the rarity of metastatic infections in cystic fibrosis. Staphylococcus aureus grew from the patient's sputum culture as well as mucoid and nonmucoid aeruginosa, but these organ¬ isms were not isolated from his brain abscesses. The immunologie factors suggested above may have prevented the spread of the total colonized flora of his lung to the brain. Alternatively, the existence of different organisms in the brain and lung raises the possi¬ bility that there was no commonality and that the association was coinci¬ dental.
References 1. Gates EM, Kernohan JW, Craig WM: Metastatic brain abscess. Medicine 29:71-98, 1950. 2. Brewer NS, MacCarty CS, Wellman WE: Brain abscess: A review of recent experience. Ann Intern Med 82:571-576, 1975. 3. Martinez-Tello FJ, Braun DG, Blanc WA: Immunoglobin production in bronchial mucosa and bronchial lymph nodes, particularly in cystic
fibrosis of the pancreas. J Immunol 10:989-1003, 1968. 4. Schwartz RH: Serum immunoglobin levels in cystic fibrosis. Am J Dis Child 11:408-411, 1966. 5. Neter E: Pseudomonas aeruginosa infection and humoral antibody response of patients with cystic fibrosis. J Infect Dis 130(suppl):132-133, 1974.
Downloaded From: http://archneur.jamanetwork.com/ by a University of Manitoba User on 06/19/2015
6. Diaz F, Mosovich LL, Neter E: Serogroups of Pseudomonas aeruginosa and the immune response of patients with cystic fibrosis. J Infect Dis 121:261-274, 1970. 7. Holbert SP, DiSant' Agnese PA, Kotch FR: Staphylococcal antibodies in cystic fibrosis of the pancreas. Pediatrics 26:792-799, 1960.
