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Haemophilus aphrophilus as a cause of chronic suppurative pulmonary infection and intra-abdominal abscesses
Journal of Infection (I987) I5, I6I-I63
CASE REPORT
Haemophilus aphrophilus pulmonary
as a c a u s e o f c h r o n i c s u p p u r a t i v e infection and intra-abdominal abscesses
K a t h r y n Kiddy a n d Jane W e b b e r l e y
Department of Microbiology, Selly Oak Hospital, Birmingham B29 6JD, U.K. Accepted for publication 6 March I987 Summary A chronic Haemophilus aphrophilus pulmonary infection associated with the formation of intra-abdominal abscesses in a previously healthy 38-year-old man is described. Other reports have related the isolation of H. aphrophilus to underlying malignant disease or, when the cause of endocarditis, to pre-existing valvular disease. The underlying condition, if any, in this case remains unclear.
Introduction
Haemophilus aphrophilus is rarely implicated as the cause of disease in human beings. Although its isolation in cases of endocarditis and brain abscess is well described, reports of infections in other sites seldom appear. We report here a case of chronic, suppurative pulmonary infection due to H. aphrophilus associated with the formulation of intra-abdominal abscesses in a 38-year-old immunocompetent man.
Case r e p o r t A previously healthy, 38-year-old caucasian ambulance man presented in January I984 with right-sided, non-pleuritic chest pain and a cough that produced purulent sputum. He had not smoked for I year. A chest X-ray showed elevation of the right hemi-diaphragm with adjacent streaky opacity and apparent shrinkage of the right lower lobe. T h e patient refused further investigation but improved on treatment with ampicillin and was allowed home. Sputum cultures at that time were negative. When seen as an outpatient in April of the same year, X-ray appearances had improved and further surveillance was deemed unnecessary. In September I984, however, the patient was admitted to hospital complaining of loss of appetite, breathlessness, and a productive cough associated with left-sided, non-pleuritic chest pain of I weeks' duration. He had had recurrent, mild chest infections over the preceding months and had received several courses of antibiotics from his general practitioner although sputum cultures had not been performed. On admission, he was noted to be pale, sunken-eyed and dyspnoeic at rest. His fingers were clubbed and there was clinical evidence of bilateral pleural effusions, later confirmed by chest X-ray. His temperature was 39"0 °C, haemoglobin concentration IO'7 g/dl and white blood cell (WBC) oi63-4453/87/o5oi6I +03 $02.00/0
~ I987 The British Society for the Study of Infection
I62
K. KIDDY AND J. WEBBERLEY
count 24"6X I09/1 (91 °/o neutrophils). Intravenous ampicillin and erythromycin initially led to rapid improvemet but, 3 days later, he developed a swinging pyrexia, the W B C count had risen to 29"4 × IO9/1 and the erythrocyte sedimentation rate (ESR) was 121 1Tlm/h. T h e haemoglobin concentration had fallen to 9"9 g/dl, a blood-film showing microcytosis and h y p o c h r o m i a ; tests of liver function showed mild disorder. S p u t u m cultures taken on admission were negative. Ultrasound examination of the abdomen revealed several large, multilocular, cystic lesions around the liver, the right kidney and pancreas. T h e appearances were t h o u g h t to be consistent with either a large cystadenoma of the pancreas or hydatid disease. One week later, serological tests for hydatid disease being negative, a laparotomy was performed. At operation, a large multilocular abscess was found around the liver w i t h several smaller abscesses in the tail of the pancreas. T h e larger of these were drained; smaller ones were left undisturbed. Neither appearances at operation nor subsequent histological examination revealed any evidence of malignant disease. Cultures of pus all yielded a heavy growth of H. aphrophilus susceptible to ampicillin. Identity of the organisms was confirmed in the U.K. National Collection of T y p e Cultures. T h e patient made a good post-operative recovery and was allowed h o m e 3 weeks later in N o v e m b e r I984. Over the ensuing months, he gradually improved, returning to full time work and gaining weight, although frequent recurrences of a cough that produced blood-stained s p u t u m required n u m e r o u s courses of ampicillin. A heavy, pure growth of H. aphrophilus was obtained from s p u t u m in January I986 when the E S R had fallen to 34 m m / h , t h e haemoglobin concentration was z3"3 g / d l and the W B C count was 9"2 x zo9/1. T h e patient remained reluctant to submit to further investigation but, when he did eventually agree to bronchoscopy in February I986, no obvious abnormality was found. Culture of bronchoscopic aspirate yielded a heavy, pure growth of H. aphrophilus. In view of the evidence of continuing infection with frequent exacerbations and unchanging antibiotic susceptibility of the causative organism, the patient has since been receiving amoxycillin on a long-term basis. He has remained well. Discussion
H. aphrophilus, first described by Khairat in I940,1 is a small, Gram-negative cocco-bacillus which appears to form part of the normal oropharyngeal microflora. Kraut and colleagues, 2 using selective techniques, isolated the organism from gingival and intra-dental material in 35 % of 45 healthy adults studied. Consistent with an oropharyngeal source is an apparent association between H. aphrophilus infection and a past history of dental disease or manipulation. ~'4 Reports of infection following animal bites and accidental inoculation with toothpicks would support this. 3 Since,the initial isolation of the organism from the blood of a patient with endocarditis, H. aphrophilus has been recorded as the cause of infection at various sites. In total, we have found I43 cases of H. aphrophilus infection described in the world literature, a-13 Of these, 5z (35"7%) patients had endocarditis and I9 (I3"3 %) had brain abscesses while only 9 (6"3 %) had p u l m o n a r y infections, notably lung abscesses or empyema. I n only five
H a e m o p h i l u s a p h r o p h i l u s infection
I63
i n s t a n c e s (3"5 % ) w a s H. aphrophilus i s o l a t e d f r o m i n t r a - a b d o m i n a l sites w h e r e it w a s a s s o c i a t e d w i t h p e r i t o n i t i s o r s u r g i c a l w o u n d d r a i n a g e r e s u l t i n g f r o m a c u t e a p p e n d i c i t i s , c h o l e c y s t i t i s o r p e r f o r a t e d d u o d e n a l ulcer. F o r m a t i o n o f multiple intra-abdominal abscesses has not previously been described. I n m o s t cases r e p o r t e d , o n e o r m o r e o t h e r o r g a n i s m s w e r e i s o l a t e d in a d d i t i o n to H. aphrophilus, a l t h o u g h p a r t i c u l a r a s s o c i a t i o n h a s n o t b e e n m a d e . I n o u r case, H. aphrophilus w a s i s o l a t e d o n e a c h o c c a s i o n in p u r e g r o w t h . O f t h e I 3 4 cases f o r w h i c h details w e r e a v a i l a b l e , IO2 (76"I % ) p a t i e n t s h a d some pre-disposing or associated condition. Bieger and colleagues z noted a c o n n e c t i o n b e t w e e n t h e i s o l a t i o n o f H. aphrophilus a n d m a l i g n a n t d i s e a s e o r c a n c e r c h e m o t h e r a p y in 26 % c a s e s ; in m a n y cases o f e n d o c a r d i t i s , p r e e x i s t i n g lesions o f h e a r t v a l v e s w e r e m e n t i o n e d . I n o u r case, t h e u n d e r l y i n g c o n d i t i o n , if a n y , r e m a i n s u n c l e a r . (We thank D r I. G r e e n for permission to report this case and M r s S. Thornhill for secretarial help as well as staff of the U . K . National Collection of T y p e Cultures for confirming the identity of the organism.)
i. 2. 3. 4. 5. 6. 7. 8. 9. IO. I I. I2. I3.
References Khairat O. Endocarditis due to a new species of Haemophilus. Bacteriol I94O; 50: 497505. Kraut SM, Attenbery HR, Finegold SM, Sutter VL. Detection ofHaemophilus aphrophilus in the human oral flora with a selective medium, ff Infect Dis I972; I 2 6 : I 8 9 - I 9 2 . Bieger RC, Brewer NS, Washington JA. Haemophilus aphrophilus: A microbiologic and clinical review and report of 42 cases. Medicine I978; 57: 345-355Root TE, Silva EA, Edwards LD, Topp JH. Hemophilus aphrophilus endocarditis with a probable primary dental focus of infection. Chest I98I ; 80: IO9-Iro. Varghese R, Melo JC, Barnwell, P, Chun CH, Raft MJ. Endocarditis due to Hemophilus aphrophilus. Report of a case with possible transmission from dog to man. Chest r977; 72: 68o-682. Crawford SA, Evans JA, Crawford GE. Necrotising fascitis associated with Haernophilus aphrophilus. Report of a case. Arch Intern ivied I978; I38: I7X4-I715. Garner JG. Isolation of Actinobacillus actinomycetemcomitans and Haemophilus aphrophilus at Auckland Hospital. N Z Med ff I979; 89: 384-386. Huck W, Britt MR. Haemophilus aphrophilus cholecystitis. Am ff Clin Path I978; 69: 361-363. Mesko ZG, Bauza J, Vinas C. Hemophilus aphrophilus with cerebral embolism. ,7 Pediatr I976; 89; IO3I-IO32. Sugarman B, Pesanti EL. Haemophilus aphrophilus. Arch Neurol I979; 36: 859. Waiters JP, Ferguson RK, Michaud PA, Knobel P, Fontolliet Ct-I. Glomerulonephritis associated with Haemophilus aphrophilus endocarditis. Clin Nephrol I978 ; 9: 73-76. White CB, Lampe RM, Copeland RL, Morrison RE. Soft tissue infection associated with Haemophilus aphrophilus. Pediatrics r98I ; 67: 434-435. Arneborn P, Lindquist BL, Sj6berg L. Severe pulmonary infection by Haemophilus aphrophilus in a non-compromised child. Scand.7 Infect Dis r985 ; I7 : 327-329 •
CASE REPORT
Haemophilus aphrophilus pulmonary
as a c a u s e o f c h r o n i c s u p p u r a t i v e infection and intra-abdominal abscesses
K a t h r y n Kiddy a n d Jane W e b b e r l e y
Department of Microbiology, Selly Oak Hospital, Birmingham B29 6JD, U.K. Accepted for publication 6 March I987 Summary A chronic Haemophilus aphrophilus pulmonary infection associated with the formation of intra-abdominal abscesses in a previously healthy 38-year-old man is described. Other reports have related the isolation of H. aphrophilus to underlying malignant disease or, when the cause of endocarditis, to pre-existing valvular disease. The underlying condition, if any, in this case remains unclear.
Introduction
Haemophilus aphrophilus is rarely implicated as the cause of disease in human beings. Although its isolation in cases of endocarditis and brain abscess is well described, reports of infections in other sites seldom appear. We report here a case of chronic, suppurative pulmonary infection due to H. aphrophilus associated with the formulation of intra-abdominal abscesses in a 38-year-old immunocompetent man.
Case r e p o r t A previously healthy, 38-year-old caucasian ambulance man presented in January I984 with right-sided, non-pleuritic chest pain and a cough that produced purulent sputum. He had not smoked for I year. A chest X-ray showed elevation of the right hemi-diaphragm with adjacent streaky opacity and apparent shrinkage of the right lower lobe. T h e patient refused further investigation but improved on treatment with ampicillin and was allowed home. Sputum cultures at that time were negative. When seen as an outpatient in April of the same year, X-ray appearances had improved and further surveillance was deemed unnecessary. In September I984, however, the patient was admitted to hospital complaining of loss of appetite, breathlessness, and a productive cough associated with left-sided, non-pleuritic chest pain of I weeks' duration. He had had recurrent, mild chest infections over the preceding months and had received several courses of antibiotics from his general practitioner although sputum cultures had not been performed. On admission, he was noted to be pale, sunken-eyed and dyspnoeic at rest. His fingers were clubbed and there was clinical evidence of bilateral pleural effusions, later confirmed by chest X-ray. His temperature was 39"0 °C, haemoglobin concentration IO'7 g/dl and white blood cell (WBC) oi63-4453/87/o5oi6I +03 $02.00/0
~ I987 The British Society for the Study of Infection
I62
K. KIDDY AND J. WEBBERLEY
count 24"6X I09/1 (91 °/o neutrophils). Intravenous ampicillin and erythromycin initially led to rapid improvemet but, 3 days later, he developed a swinging pyrexia, the W B C count had risen to 29"4 × IO9/1 and the erythrocyte sedimentation rate (ESR) was 121 1Tlm/h. T h e haemoglobin concentration had fallen to 9"9 g/dl, a blood-film showing microcytosis and h y p o c h r o m i a ; tests of liver function showed mild disorder. S p u t u m cultures taken on admission were negative. Ultrasound examination of the abdomen revealed several large, multilocular, cystic lesions around the liver, the right kidney and pancreas. T h e appearances were t h o u g h t to be consistent with either a large cystadenoma of the pancreas or hydatid disease. One week later, serological tests for hydatid disease being negative, a laparotomy was performed. At operation, a large multilocular abscess was found around the liver w i t h several smaller abscesses in the tail of the pancreas. T h e larger of these were drained; smaller ones were left undisturbed. Neither appearances at operation nor subsequent histological examination revealed any evidence of malignant disease. Cultures of pus all yielded a heavy growth of H. aphrophilus susceptible to ampicillin. Identity of the organisms was confirmed in the U.K. National Collection of T y p e Cultures. T h e patient made a good post-operative recovery and was allowed h o m e 3 weeks later in N o v e m b e r I984. Over the ensuing months, he gradually improved, returning to full time work and gaining weight, although frequent recurrences of a cough that produced blood-stained s p u t u m required n u m e r o u s courses of ampicillin. A heavy, pure growth of H. aphrophilus was obtained from s p u t u m in January I986 when the E S R had fallen to 34 m m / h , t h e haemoglobin concentration was z3"3 g / d l and the W B C count was 9"2 x zo9/1. T h e patient remained reluctant to submit to further investigation but, when he did eventually agree to bronchoscopy in February I986, no obvious abnormality was found. Culture of bronchoscopic aspirate yielded a heavy, pure growth of H. aphrophilus. In view of the evidence of continuing infection with frequent exacerbations and unchanging antibiotic susceptibility of the causative organism, the patient has since been receiving amoxycillin on a long-term basis. He has remained well. Discussion
H. aphrophilus, first described by Khairat in I940,1 is a small, Gram-negative cocco-bacillus which appears to form part of the normal oropharyngeal microflora. Kraut and colleagues, 2 using selective techniques, isolated the organism from gingival and intra-dental material in 35 % of 45 healthy adults studied. Consistent with an oropharyngeal source is an apparent association between H. aphrophilus infection and a past history of dental disease or manipulation. ~'4 Reports of infection following animal bites and accidental inoculation with toothpicks would support this. 3 Since,the initial isolation of the organism from the blood of a patient with endocarditis, H. aphrophilus has been recorded as the cause of infection at various sites. In total, we have found I43 cases of H. aphrophilus infection described in the world literature, a-13 Of these, 5z (35"7%) patients had endocarditis and I9 (I3"3 %) had brain abscesses while only 9 (6"3 %) had p u l m o n a r y infections, notably lung abscesses or empyema. I n only five
H a e m o p h i l u s a p h r o p h i l u s infection
I63
i n s t a n c e s (3"5 % ) w a s H. aphrophilus i s o l a t e d f r o m i n t r a - a b d o m i n a l sites w h e r e it w a s a s s o c i a t e d w i t h p e r i t o n i t i s o r s u r g i c a l w o u n d d r a i n a g e r e s u l t i n g f r o m a c u t e a p p e n d i c i t i s , c h o l e c y s t i t i s o r p e r f o r a t e d d u o d e n a l ulcer. F o r m a t i o n o f multiple intra-abdominal abscesses has not previously been described. I n m o s t cases r e p o r t e d , o n e o r m o r e o t h e r o r g a n i s m s w e r e i s o l a t e d in a d d i t i o n to H. aphrophilus, a l t h o u g h p a r t i c u l a r a s s o c i a t i o n h a s n o t b e e n m a d e . I n o u r case, H. aphrophilus w a s i s o l a t e d o n e a c h o c c a s i o n in p u r e g r o w t h . O f t h e I 3 4 cases f o r w h i c h details w e r e a v a i l a b l e , IO2 (76"I % ) p a t i e n t s h a d some pre-disposing or associated condition. Bieger and colleagues z noted a c o n n e c t i o n b e t w e e n t h e i s o l a t i o n o f H. aphrophilus a n d m a l i g n a n t d i s e a s e o r c a n c e r c h e m o t h e r a p y in 26 % c a s e s ; in m a n y cases o f e n d o c a r d i t i s , p r e e x i s t i n g lesions o f h e a r t v a l v e s w e r e m e n t i o n e d . I n o u r case, t h e u n d e r l y i n g c o n d i t i o n , if a n y , r e m a i n s u n c l e a r . (We thank D r I. G r e e n for permission to report this case and M r s S. Thornhill for secretarial help as well as staff of the U . K . National Collection of T y p e Cultures for confirming the identity of the organism.)
i. 2. 3. 4. 5. 6. 7. 8. 9. IO. I I. I2. I3.
References Khairat O. Endocarditis due to a new species of Haemophilus. Bacteriol I94O; 50: 497505. Kraut SM, Attenbery HR, Finegold SM, Sutter VL. Detection ofHaemophilus aphrophilus in the human oral flora with a selective medium, ff Infect Dis I972; I 2 6 : I 8 9 - I 9 2 . Bieger RC, Brewer NS, Washington JA. Haemophilus aphrophilus: A microbiologic and clinical review and report of 42 cases. Medicine I978; 57: 345-355Root TE, Silva EA, Edwards LD, Topp JH. Hemophilus aphrophilus endocarditis with a probable primary dental focus of infection. Chest I98I ; 80: IO9-Iro. Varghese R, Melo JC, Barnwell, P, Chun CH, Raft MJ. Endocarditis due to Hemophilus aphrophilus. Report of a case with possible transmission from dog to man. Chest r977; 72: 68o-682. Crawford SA, Evans JA, Crawford GE. Necrotising fascitis associated with Haernophilus aphrophilus. Report of a case. Arch Intern ivied I978; I38: I7X4-I715. Garner JG. Isolation of Actinobacillus actinomycetemcomitans and Haemophilus aphrophilus at Auckland Hospital. N Z Med ff I979; 89: 384-386. Huck W, Britt MR. Haemophilus aphrophilus cholecystitis. Am ff Clin Path I978; 69: 361-363. Mesko ZG, Bauza J, Vinas C. Hemophilus aphrophilus with cerebral embolism. ,7 Pediatr I976; 89; IO3I-IO32. Sugarman B, Pesanti EL. Haemophilus aphrophilus. Arch Neurol I979; 36: 859. Waiters JP, Ferguson RK, Michaud PA, Knobel P, Fontolliet Ct-I. Glomerulonephritis associated with Haemophilus aphrophilus endocarditis. Clin Nephrol I978 ; 9: 73-76. White CB, Lampe RM, Copeland RL, Morrison RE. Soft tissue infection associated with Haemophilus aphrophilus. Pediatrics r98I ; 67: 434-435. Arneborn P, Lindquist BL, Sj6berg L. Severe pulmonary infection by Haemophilus aphrophilus in a non-compromised child. Scand.7 Infect Dis r985 ; I7 : 327-329 •