Brain Abscess Caused by Blastomyces dermatitidis in a Child With Cystic Fibrosis

Brain Abscess Caused by Blastomyces dermatitidis in a Child With Cystic Fibrosis

selected reports Brain Abscess Caused by B~sromycesdermauud~ina Child With Cystic Fibrosis* William M. Gershan, M.D .; Lee S. Rusakow, M .D.; Kelly ...

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selected reports Brain Abscess Caused by B~sromycesdermauud~ina

Child With Cystic Fibrosis*

William M. Gershan, M.D .; Lee S. Rusakow, M .D.; Kelly j. Henrickson, M.D .; and Mark L . Splaingard, M.D .

An 8-year-old girl with moderately severe cystic fibrosis and right upper lobe bronchiectasis developed a cerebellar abscess caused by Blastomyces dermatitidis. To our knowledge, this is the youngest child with cystic fibrosis and a brain abscess, and the first documented case caused by a fungus. (Chest 1994; 106:601-03)

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rain a bscess is rare in patients with cystic fibrosis (C F) . Previously d escribed cas es have occurred in adults or adolescents a nd were ca used b y ba ct eria th at a re part of th e normal or al flora. 1-7 W e report th e case of an 8-year-o ld girl with m od erat ely se ve re C F who d ev el op ed se ve re headaches with vomiting and was found to have a cerebellar a bscess ca used by th e fungus Blastomyces dermatitidis. CASE R EPOHT

An 8-year-old girl with CF, moderat ely severe obstructive lung disease, and chronic right upp er lobe bronchiectasis noted on previous chest radiographs (Fig 1) and computed tomographic (CT) scan was admitted to the hospital for a right up per lobe lobectomy. At the tim e of hospital admission, she comp lained of mild, intermittent front al heada ches of 3 weeks' duration without fevers, emesis, or dizziness. History included a central venous access port placed for intravenous antibiotic administration and a gastrostomy tub e insert ed for failur e to thr ive. She was hospitalized three times during the 7 month s prior to the plann ed lobectomy for respiratory exacerbations associated with Pseudomonas aeruginosa colonization. Treatment during those hospitalizations includ ed antipseudomonal an tibiotics. The patient's first course of systemic steroids for bronchospasm was 3 months prior to hospital admission when she was placed on a regimen of predn isone, 0.5 mg/ kg/ day , for 5 weeks. While receiving int ravenous ant ipseudom onal antibiotics prior to surge ry, her headaches worsened and she began vomiting. Results of her neurologic and fundu scopic examinations were norm al. A head CT scan demonstrated a 4X3-cm left cerebellar mass that enhanced with contrast (Fig 2). The mass was surg ically resected and found to be a discrete abscess cavity. A fungal potassium hydroxide (KOH) stain of the lesion revealed budding yeast and the patient was started on a regimen of intrav enous amphotericin B. Cultures of the lesion grew B dermatitidis and serologic tests via immunodiffusion were positive for Blastomyces. Blood and urine fungal cultures were negative and liver fun ction studies, a bone scan, abd ominal CT scan, and an ophthalmologic examination were all normal. On furth er qu estioning, we learned that our patient had *From the Departm ent of Pediatrics, The Medical College of Wisconsin, Children's Hospital of Wisconsin, Milwaukee.

FIGURE I. Right upp er lobe bronchiectasis (arrow) noted on chest radiog raph .

FIGURE 2. Head CT scan de monstrating left posterior cerebellar abscess (arrow). CHEST 1 106 / 2 1 AUGUST, 1994

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Table I-Reported Cases of Patients With Cystic Fibrosis and Brain Absc ess Source

+ Patient

No.

Duffner and Cohen' 1 Fischer et al2 2 3 4 Sherma n et al3 5

Age, yr

Abscess Culture

Associated Findings

Outcome

21

Proteus int ermediu s

Bronchiectasis

Seizure disorder

17 24 19

Proteus mirabi lis; Peptostreptococcus Str ept ococcus viridans Streptococcus species

Cor pulmonale Clubbing Bronchiectasis

Died 3 wk postoperatively No neurologic deficit No neurologic deficit

20

Sterile

Bronchiectasis; opacified sinuses

No neurologic deficit

22

Not reported

Cor pulmonale

No neurologic deficit

19

Sterile

Clubbing

No neurologic deficit

22

P aerugin osa; Escherichia coli; Bacteroides [ragilis

Bronchiectasis; opacified sinuses; diabetes mellitus

Dicd 10 d postoperativel y

Not reported Not reported

Not reported Not reported

Not reported Not reported

B derma titid is

Bronchiectasis

No neurologic deficit

Ayres and Kinsella" 6

Rabkin and Blaser '' 7 Kline6 8

Saez- Llorens et aF

9 10 Present case 11

::::;15 ::::;15 8

fr eq uent ly vacationed with her fa mily to northern Wisconsin , an area tha t is en de mic for B dermatitidis. She received a total of 1.2 g of am photericin B (54 rug /kg) and did we ll without neurologic seq uela e. Follow-up bra in scans revealed resolution of the abscess cavi ty. Th e pat ient was readmitted to the hospital 2 mo nths aft er her abscess resect ion for th e right upper lobe lobectom y. Lun g tissue speci mens fro m the resected lobe revealed acu te and chronic bronch itis and fibr osis with intra-alveolar hem orrhage. Fu ngal KOH stains we re nega tive an d a washing of th e speci men gr ew P aeruginosa, but no fung i. Th e patient remains neurologicall y norma l 12 month s afte r abscess resection. DIS CUSSION

Onl y ten cases of brain abscess in cystic fibrosis have been previously reported (Table 1).1-7 Each of these previous cases involved eit her olde r adolescents or adults or did not specify the pati ent's age. Rabkin and Blaser-' suggested that the "absence of pediatric cases suggests some morbid developm en ts in long-standing disease necessary before susceptibility to brain abscess is established." This case would dispute that claim as our patient was only 8 years old. Other studies have suggested a relationship between sinus disease in CF and developm ent of brain abscess.3,6,8 Our pat ient , however , had no previous clinical symptoms to sugge st chronic sinus inflammation . This present case also differs from ea rlie r reports in th e orga nism that caused the abscess. In five cases, either aerobic or anae robic bacteria wer e culture d from the abscess cavit y I,2,6 while cultures were either sterile3,5 or not rep ort ed f-? in the oth ers. Blastomy ces dermatitidis, an unusual cause of intracranial abscess in children,9-13 was recovered from our pati ent. Central ner vous system blastom ycosis is usuall y caused by hematogenous spread from a pulm onary source.J! Although we cann ot det ermine wheth er th e primary focus of infection in our patient was her lung, th e orga nism never gre w on several sputum cul602

tures tha t were sent in the period prior to the diagnosis of her brain abscess. Her right upper lobe was severel y bronchiect atic and colonized with P aeruginosa for man y years and th e possibilit y of a recent cocolonization with B dermat ituli s exists. It is also possible th at the amphotericin B erad icated th e organism from her lungs by the time that the right upper lobe lobect om y was done . We believe tha t our pati ent con tracted blasto m ycosis while vacationing in northern Wisconsin, an area that is ende mic for B dermatitidis. 9,12,14 Since her famil y frequ ent ly visited this area, it is impossible to determine exactly whe n she was initially exposed to the organism. We are unc ertain whe ther the oral stero ids played a role in her acquisition of this infection . This case demonstra tes that unusual infections cause d by uncommon orga nisms may complicate the course of CF in children . R EFERENCES

2 3 4 5 6 7

8

Duffn er PK, Cohen ME. Cystic fibrosis with br ain abscess. Arch Ne urol 1979; 36:27-8 Fischer EG , Shwachman H, Wepsic JG. Brain abscess and cystic fibrosis. J Pediatr 1979; 95:385-88 Sherm an JM , Orenstein DM, Stern RC. Brain abscess and cystic fibrosis. J Pedi atr 1980; 96:952 Ayres J, Kinsella H. Multiple cerebra l abscessesin an adult with cystic fibrosis. Br J Dis Ch est 1982; 76:99-101 Rabkin CS, Blaser MJ. Brain abscess: a complication of cystic fibrosis in adults. Ann Neurol 1984; 15:608-10 Kline MW . Brain abscess in a pati ent with cystic fibrosis. Pediatr Inf ect Dis 1985; 4:72-3 Saez-Llorens XJ, Umana MA, Odi o CM, McCracken GH , Nelson JD . Brain abscess in infant s and child ren . Pediatr Infect Dis 1989; 8:449-58 Fairbanks DN , Milmo e GJ. Th e diagnosis and management of sinusitis in child ren: complication s and sequelae: an otolar yngologist's perspect ive. Pedi atr Infect Dis 1985; 4(suppI6):S75-9 Brain Abscess Caused by B dermatitidis in CF (Gershan et aQ

9 Sarosi GA, Davies SF. Blastomycosis: state of the art. Am Rev Respir Dis 1979; 120:911-38 10 Gonyea EF . Th e spectrum of pr imar y blastomycotic meningitis: a review of cent ral nervous system blastom ycosis. Ann Neuro11978; 3:26-39 11 Roos KL, Bryan JP, Maggio WW, Jan e JA, Scheid WM. Intr acranial blastomyc oma. Medicine 1987; 66:224-35 12 Bell RM, Starshak RJ, Sty JR, Harb JM. Solitary intracranial blastomycotic abscess. Wis Med J 1983; 82:23-5 13 Laskey WK, Sarosi GA. Blastomycosis in childr en. Pediatrics 1980; 65:111-14 14 Bradsher RW. Blastomycosis. Clin Infect Dis 1992; 14(suppl 1):S82-90

Hepatic Vein Obstruction Due to Swan-Ganz Catheter Placement* Michael S. Davis, M .D., F.C.C.P.

Complications from Swan-Ganz catheters during insertion, long-term placement, or removal have been known since its development. I describe the unusual presentation of a pacing Swan-Ganz catheter mispositioned into the hepatic vein producing vascular obstruction, yet with adequate cardiac pacing.(Chest 1994; 106:603-05) the introduction of the flow-directed balloonSince tipped catheter (Swan-Canz) for continuous monitor-

ing of hemodynamic function and cardiac pacing, a number of authors have reported complications associated with the routine use of this catheter.lf Arrhythmias, infection, perforation of the pulmonary artery, as well as vascular occlusion and pulmonar y infarction have occurred. In addition, the catheter has injured the pulmonic and tricuspid valves and has been reported to knot within the ventricle. Recently, I had the opportunity to evaluate a patient with a Swan-Canz pacing catheter with an unu sual presentation that I have not seen reported. C ASE R EPORT

This 73-year-old woman pr esented in October 1992 with a complaint of chest pain . Paramedics were called to her home and *From the Pulmonary Section , Department of Medicine, Doctor s Hospit al of Mant eca, Mant eca, Calif. Reprin t request:c/ o Elaine Aberer, Staff Development Director , 1205 East North Street , Mant eca, Call] 95336

noted the pati ent to have a BP of 68/42 mm Hg, and an irr egular pulse rate of 26 beats/min. A card iac rhythm (lead 2) suggested a third-degree heart block with junctional escape rhythm . Th e patient was transported to the local eme rge ncy department wher e one of the staff ph ysicians inserted a pacing Swan-Ganz catheter (Baxter model 93-200 H-7F, Edwards Swan-Ganz Pacing TO Catheter with AMC Thromboshield) with successful capture at a rat e of 80 beats/m in; vital signs improved (Fig 1). Cardiac enzy mes subsequently becam e positive. Th e patient had a histor y of high BP, type II diabe tes mellitus, and peripheral vascular disease. She had a myoc ard ial infarction in June 1991. Coron ar y angiograms at that tim e revealed 40 percent narrowing of the right carotid art ery , 30 percent narrow ing of the trunk of the circumflex, and 50 percent narrowing of th e proxim al ant erior descending branch. Th e ejection fraction at that tim e was estimated to be 40 percent. An anterior apical wall aneurysm was also identifi ed on the echocard iogra m. A chest radi ograph obta ined at the tim e of this hospital adm ission revealed normal heart size and a light diffuse bilatera l lower lobe infiltrate. A Swan-C an z catheter was noted to loop in the right ventricle and terminate in what was reported as consolidated right lower lobe. Th e att ending ph ysician presumed the Swan-Ganz catheter was prop erly positioned becau se of appropr iate pacer capture. Th e nur ses reported either the "inability to 'wedge ' the cath eter " or "abnormal pulmonary artery pressures on monitoring" and appropriate hemodynamics were thus not obtained. Cardiology consultation confirm ed the diagnosi s of an acute inferior wall myocardial infarction with complications of complete heart block and congestive heart failure. Th ey recommended observation for 7 days prior to the decision to insert a permanen t pacem aker. On the fourth hospital day, acute respiratory failur e developed. Arteri al blood gases ind icat ed a pH of 7.41, PC02 of 23.9 mm Hg , P02 of 48.9 mm Hg, HCOs of 50 mmol /L, and Sa02 of 86 percent (FI0 2 of 45 percent ). Th e patient had a WBC count of 19,400 cells per ml with 75 percent polymorphonuclear cells, 9 percent bands, 9 percent lymphocytes, and 6 percent monocytes. Liver enzyme levels were mark edl y eleva ted with respect to those at hospit al admission (Ta ble 1). A new chest radio grap h revealed the heart size to be normal. Persistent bilat eral basilar infiltrates were seen. Th e Swan-G anz catheter appear ed looped in the right ventricle with the tip of the catheter below the diaphragm and presumed in the right hepatic vein (Fig 2). Echocardiogram revealed an ejection fraction of 10 to 20 percent. In view of the inappropriate placem ent of the Swan-G an z catheter and suspected hepatic vein obstruction, the decision was made to repla ce the catheter. Th e patient was 100 perc ent dependent on the pac em aker as indicated by a tr ial of turning off the pacem aker resulting in the patient's heart rat e dropping to 30 beat s/m in with an occasional junction al escape rhythm and systolic BP dropping to 40 mm Hg. A new pacing Swan-G anz catheter was insert ed befor e rem oval of the old ca theter with the use of fluoroscopy to ensure appropriate plac em ent and to det ect possible entangleme nt with the initial Swan-Ganz catheter. Th is was perform ed successfully with appropr iate pacemaker capture. Hemod ynamics were now appropriately recorded (Table 1).

nv

FIGURE l. Electroca rdiographic tracing subseq uent to insertion (at the time of hospital admission) of Swan-Can z catheter with ventric ular pace lead s showing successful capture.

CHEST 1 106/ 21 AUGUST, 1994

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