BRIEF CLINICAL OBSERVATIONS

rest. Twenty minutes into each tennis match, he would experience hypersalivation associated with fatigue but without associated chest pressure or shortness of breath. The hypersalivation episodes progressed in frequency and severity to where, after a brisk walk to work each morning, the patient stood with his mouth open, allowing copious saliva to pour out. He reported normal swallowing, lacrimation, and facial sweating. On the day of admission, after rushing into work, the patient experienced hypersalivation associated with substernal chest pressure. An electrocardiogram in the emergency room revealed ST elevation inferiorly and ST depressions anterolaterally. The patient ruled in for a non-Q-wave myocardial infarction with a peak creatine phosphokinase level of 794 U with 9% MB fraction. After an uncomplicated postinfarction course, coronary angiography revealed 100% occlusion of an aorta-to-right coronary artery saphenous graft and a tandem graft to the posterior descending artery. Other disease included 100% occlusion of the midsegment of the native right coronary and left anterior descending coronary arteries, and 70% narrowing of the proximal first obtuse marginal. The saphenous vein bypass to the left anterior descending artery was widely patent. He subsequently underwent a second successful multi-vessel revascularization surgery. At 5 months after surgery, he has experienced no further episodes of excessive salivation with exertion. The Ramsay Hunt syndrome, or herpetic geniculate ganglionitis, presents typically with facial palsy associated with herpetic vesicles in the external auditory canal [2]. Cases have been described, however, in which fibers of the 5th, 8th, 9th, and 10th cranial nerves have also been in202

volved [3]. Our patient had involvement of both the fifth and ninth cranial nerves as manifested by herpetic lesions on the forehead and pharynx, respectively, as well as the seventh cranial nerve as manifested by facial palsy. Herpetic inflammatory lesions likely involved the trigeminal ganglion, the geniculate ganglion, and the superior and inferior ganglia of the ninth cranial nerve. Salivation is controlled through parasympathetic innervation to the parotid, submandibular, and sublingual glands. Parasympathetic innervation to these glands may have been injured, as the axons cross either the geniculate ganglion or the superior and inferior ganglia. The mechanism by which our patient manifested excessive salivation is uncertain. However, we propose denervation hypersensitivity as a possible mechanism for excessive salivation as an anginal equivalent. Interruption of preganglionic fibers can result in increased sensitivity of postganglionic neurons and the tissues they supply to circulating hormones. In animal models, increased denervation sensitivity has been observed in structures supplied by cholinergic fibers, including the salivary glands [4]. In cats, Simeone and Maes [5] demonstrated that submaxillary gland secretion in response to intravenous acetylcholine is increased unilaterally in a gland denervated over a period of 50 to 90 days as compared with the contralateral freshly denervated gland. In our patient, herpetic infection may have resulted in partial denervation of the right-sided salivary glands and subsequent hypersensitivity to parasympathetic stimuli. Therefore, whenever the patient became ischemic, he salivated. In summary, we describe a patient with hypersalivation as an anginal equivalent. The mechanism we suggest is based upon his

August 1991 The American Journal of Medicine

Volume 91

prior episode of herpetic ganglionitis. Partial denervation of the right-sided salivary glands has likely resulted in hypersensitivity to the typical parasympathetic stimuli seen in inferoposterior ischemia. PHILLIP J. c0ZZI.M.D. JAMES V. TALANO~M.D. Northwestern

Universitv

Medical School Chicago, Illinois ”

1. Webb S. Adgey A, Pantridge J. Autonomic disturbance at onset of acute myocardial infarction, BMJ 1972; 3: 89-92. 2. Linden RJ. Function of cardiac receptors. Circulation 1973; 48: 463-80. 3. Engstrom H, Gunnar W. Herpes zoster of the seventh, eighth, ninth and tenth cranial nerves. Arch Neural Psychiatry 1949; 62: 638-52. 4. Cannon WB. The law of denervation. Am J Med Sci 1939; 198: 738-50. 5. Someone FA. Maes JP. Sensitization of the submaxillary gland by sympathetic denervation. Am J Physiol 1939; 125: 674-9. Submitted

November 29, 1990, and accepted in revised form February 19. 1991

PASTEURELLAMU1TDCIDA: ZOONOTICCAUSEOF PERITONITISIN A PATIENT UNDERGOINGPERITONEAL DIALYSIS Pasteurella multocida, which resides in the upper respiratory tract of a variety of animal hosts, especially dogs and cats, is a common cause of human infection subsequent to an animal bite [l]. In the United States, there are an estimated 1 to 1.5 million cases of pet bites per year [l] and a recent survey found that 31% and 38% of households have cats or dogs, respectively [2]. In New York City alone, there are an estimated 1 million dogs or cats as household pets [3]. The risk of exposure to P. multocida in the home is therefore considerable. The reported clinical spectrum of human infection caused by P. multocida is diverse [1,3]; unusual presentations include either spontaneous peritonitis in patients with cirrhotic liver disease [4] or peritonitis in the setting of renal failure in a patient under-

BRIEF CLINICAL

going peritoneal dialysis at home 151. In the latter instance, occult or direct P. multocida contamination of the peritoneal catheter or dialysis tubing by a household pet resulted in peritonitis [5]. We present the second case of P. multocida peritonitis occurring in a patient undergoing ambulatory peritoneal dialysis in which the organism was introduced into the peritoneal cavity by a penetrating cat bite to the dialysis tubing. We further suggest guidelines for the prevention of this unusual infectious complication of home peritoneal dialysis. The patient was a 54-year-old man with end-stage renal disease secondary to hypertension. His wife performed continuous cycling peritoneal dialysis at home using an automated cycling device. Approximately 6 months after beginning home dialysis, the patient presented to the emergency room at the Mount Sinai Hospital with chills, nausea, vomiting, abdominal pain, and cloudy peritoneal dialysis fluid. On physical examination, the patient was febrile to 37.9”C orally, and had diffuse abdominal tenderness. Admission laboratory data showed a white blood cell count of 20,700/mm3, with 20% bands and 69% segmented neutrophils. The cell count in the peritoneal dialysate was 1,800/mm3 with 88% polymorphonuclear leukocytes. Gram stain of the dialysate showed small gram-negative coccobacilli. The patient received intravenous vancomycin and gentamicin in the emergency room. His symptoms improved with continuous peritoneal dialysis, and within 72 hours, his fever, abdominal pain, and leukocytosis resolved. He was discharged and treated for 2 weeks with cefaxolin on an outpatient basis. When the initial peritoneal dialysis fluid culture grew P. multocida, the patient’s wife was asked whether there were any pets in the house. She report-

ed that they owned two cats and that one of the cats had indeed been in the patient’s bedroom and bitten the dialysis tubing on the evening prior to admission. She had noted leakage of dialysis fluid from the tubing at the puncture site. The initial peritoneal dialysis fluid inoculated to 5% sheep blood, chocolate, and MacConkey agars showed small grayish-white colonies on blood and chocolate agars only. Growth was accompanied by a characteristic “mousey” odor and an absence of hemolysis on sheep blood agar. Gramstained smear of the growth comprising the colonies showed small gram-negative coccobacilli and occasional longer bacillary forms. The isolate was identified as P. multocida by its positive reaction for cat&se, oxidase, nitrate reduction and indole production, and anaerogenic fermentation of glucose and sucrose, but not lactose, when inoculated to Kligler’s iron agar. Acidification of the slant took place within 24 hours, followed by acidification of the butt in 48 hours. The isolate tested for antibiotic susceptibility by the Bauer-Kirby disc-agar diffusion method was shown to be inhibited by ampicillin, penicillin, cefoxitin, cefoperazone, chloramphenicol, and tetracycline, and was moderately susceptible to gentamicin and resistant to vancomycin. Several facets of human P. multocida infection are emphasized by the present case. Initially, the most frequent antecedent event predisposing to P. multocida infection is an animal bite usually inflicted by a domestic animal. Although our patient was not, per se, directly bitten by his cat, the infecting organism was introduced into his body by a penetrating cat bite of his peritoneal dialysis tubing. Although the number of P. multocida cells was not quantifiable, one can only speculate that a modest August

1991

The American

OBSERVATIONS

number of these cells introduced into the peritoneal cavity through this access route were sufficient to induce severe peritonitis. Interestingly, in the case of “cat bite peritonitis” described by Paul and Rostrand [5], a similar route of acquisition was postulated, as a consequence of the patient’s dialysis tubing being penetrated by either a cat bite or cat scratch. In both patients, however, purulent peritonitis developed within 24 hours after violation of the dialysis tubing. In spontaneous P. multocida peritonitis in patients with hepatic cirrhosis, by way of contrast, the portal of entry is thought to be through the gastrointestinal tract because of the usual concomitant occurrence of periappendiceal abscesses and appendicitis [6,7]. The in vitro antibiotic susceptibility pattern of P. multocida isolates generally shows susceptibility to tetracycline, ampicillin, penicillin, second- and third-generation cephalosporins, and chloramphenicol. Aminoglycoside susceptibility is variable and resistance to vancomycin and clindamycin has been described [8]. Interestingly, however, both our patient and the patient described by Paul and Rostrand [5] responded to intravenous gentamicin and vancomycin. Perhaps peritoneal concentration of these agents in excess of tested concentrations may account for the observed therapeutic success. The recent advent of automatic cyclers for home peritoneal dialysis has created a setting for exposure of the patient’s peritoneal catheter and dialysis tubing to animal bites and scratches. Although only 846 of 12,242 home peritoneal dialysis patients in the U.S. in 1987 were on automated cyclers, this segment of the home dialysis population continues to increase [9]. Since approximately one third of our patients undergoing peritoneal dialysis own cats or Journal

of Medicine

Volume

91

203

BRIEF CLINICAL

OBSERVATIONS

dogs, they are advised to avoid performing exchanges in the presence of pets as part of their home peritoneal dialysis training. As a consequence of our own experience and the earlier one reported by Paul and Rostrand [5], we are instructing our patients to avoid performing peritoneal exchanges in the presence of pets. Further, in the event of a penetrating bite or scratch to the tubing, we advise immediate medical attention and initiation of empiric therapy for the prevention of P. multocida or other pet-induced peritonitis.

discharges from infected avian species, producing either a respiratory or systemic illness [1,2]. The diagnosis is usually based on an acute febrile disease with pulmonary symptoms and signs, and a fourfold increase in the titer of antibody against the organism. Hematologic abnormalities in psittacosis are uncommon, mainly in the form of cold agglutininemia and hemolytic anemia [3]. Severe pancytopenia at presentation of psittacosis is distinctly uncommon [l], and its occurrence as a result of reactive hemoROGERD. LONDON, M.D. phagocytosis has not been previMount Sinai School of Medicine ously reported. We believe this is EDWARDJ. BoTToNE,P~.D. the first report documenting such Mount Sinai Hospital New York, New York an association. A 54-year-old textile factory 1. Weber DJ, Wolfson JS. Swartz MN, Hooper DC. Pasteurella multocida infections. Report of 34 cases worker was hospitalized because and review of the literature. Medicine (Baltimore) of fever, headache, general mal1984; 63: 133-54. aise, cough with whitish sputum, 2.Veterinary service market for companion anipurpura over the limbs, and promals. Report of the American Veterinary Medical Association. Schaumburg. Illinois. August 1988 gressive deterioration of mental 3. Furie RA, Cohen RP. Hartman BJ. Roberts RB. function for 10 days. She had PasteureNa mulfocida infection: report in urban setbeen keeping a pet bird (thrush) ting and review of spectrum of human disease. NY at home for about 1 year. Her State J Med 1980; 80: 1597-1602. 4. Vakil N. Adiyody J. Treser G, Lue Y. PasteureNa past health was good. Examinamultooda septicemia and peritonitis in a patient tion revealed a temperature of with cirrhosis: case report and review of the litera39°C. She was conscious but ture. Am J Gastroenterol 1985; 80: 565-8. 5. Paul RV. Rostrand SG. Cat-bite peritonitis: Pasmentally dull with impairment of teurella multocida peritonitis following feline conrecent memory and higher inteltamination of peritoneal dialysis tubing. Am J Krdney lectual function. There was no Dis 1987; 10: 318-9. neck rigidity or papilledema. 6. Controni G, Stephens V. Appendicitis with peritonitis due to PasteureNa multocida. Am J Med TechFine crepitations were detected nol 1968; 34: 467-9. over the lower lobe of her left 7. Raffi F. David A, Mouzard A, Le-Neel JC, Baron D, lung. There was no lymphadeCourtieu AL. Pasteurella multocida appendiceal nopathy or hepatosplenomegaly. peritonitis: report of three cases and review of the literature. Pediatr Infect Dis 1986; 5: 695-8. The peripheral blood count 8. Stevens DL, Higbee JW, Oberhoffer TR. Everett showed pancytopenia: hemogloEE. Antibiotic susceptibilities of human isolates of bin level 9.2 g/dL, white blood Pasteurella multocida. Antimicrob Agents Checell count 1.7 X log/L, and platemother 1979; 16: 322-4. 9. US Renal Data Systems, USRDS 1989, Annual let count 7 X log/L. The red Data Report. NIH. NIDDK. Bethesda, Maryland. Aublood cells were normochromic gust 1989, USRDS; 23. and normocytic and the leukoSubmitted January 4, 1991, and accepted in cyte differential count was norrevised form February 19, 1991 mal. The erythrocyte sedimentation rate was 139 mm/hour. Results of liver function tests were PSIllACOSIS-ASSOCIATED abnormal: aspartate transamiHEMOPHAGOCYTIC nase 84 III/L (normal, 0 to 40 SYNDROME IU/L) and y-glutamyl transpeptidase 120 IU/L (normal, 4 to 40 Chlamydia psittaci infection is III/L). The lactate dehydrogenase level was also elevated to 912 contracted through exposure to 204

August

1991 The American

Journal

of Medicine

Volume

91

IU/L (normal, 211 to 423 IU/L). The coagulation profile was normal. A chest radiograph revealed extensive patchy consolidation involving both lung fields. Marrow aspiration was performed, showing normocellular marrow with adequate megakaryocytes, normoblastic erythropoiesis, and active granulopoiesis. There was an increase in bland-looking histiocytes (Figure l), with many demonstrating phagocytosis of red blood cells, erythroblasts, leukocytes, and platelets. These histiocytes accounted for 5% of all nucleated cells. There was no malignant infiltration. A diagnosis of reactive hemophagocytic syndrome was made. The patient was treated with erythromycin. Carbenicillin and amikacin were later added in view of her progressive neutropenia, with the nadir white blood cell count reaching 0.9 X log/L. Her condition improved gradually with antibiotic therapy, and the fever subsided within 48 hours of commencement of erythromycin administration. Extensive microbiologic studies including sputum and blood cultures for bacteria and serologic tests for Epstein-Barr virus, cytomegalovirus, herpes simplex virus, varicella zoster virus, Mycoplasma, Coxiella, and Legionella were all negative. However, antibodies against C. psittaci as detected by complement fixation method showed a more than fourfold increase in titer (from a titer of less than 10 to a titer of 40) within 2 weeks. Her blood counts returned to normal after 2 weeks: hemoglobin level 11.7 g/dL, white blood cell count 4.89 X log/L, and platelet count 167 X log/L. She has remained well after 2 years. Reactive hemophagocytic syndrome is a clinicopathologic entity characterized by systemic proliferation of benign hemophagocytic histiocytes, resulting in fever, hepatosplenomegaly, and