, BRIEFCLINICALOBSERVATIONS

LEPROMATOUSLEPROSYAND SEROPOSITIVITYFOR HTLV-I Leprosy and HTLV-I (human Tcell lymphotropic virus type I) infection are both endemic conditions in the Caribbean. It is surprising that their association has never been mentioned. We report what we believe to be the first such case. In 1976, lepromatous leprosy was diagnosed in a 34-year-old woman living in the French West Indies. Between 1976 and 1984, she was treated with sulfone, rifampicin, and clofazimine, and finally recovered from leprosy. However, an increase in the erythrocyte sedimentation rate (ESR) was still present. In 1988, she was found in good health, with a healed nose bending and some decreased vibratory sensitivity of both legs. No Hansen’s bacilli were found; the ESR was 40/80 mm/hour. In 1989, no change was observed on physical examination and the ESR was 40 mm/hour. Because of the previous history of leprosy in this patient from the Caribbean, we considered a retrovirus infection. Results of serologic tests were negative for HIV-l and HIV-2 (human immunodeficiency virus types 1 and 2), but were positive for HTLV-I (by enzyme-linked immunosorbent assay and Western blot). Study of the circulating lymphocyte phenotype showed a CD4 level of 850/mm3, with a CD4/CD8 ratio of 2.25. HTLV-I is the causative agent of HTLV-I-associated paraplegia and of a certain kind of T-cell leukemia [l]. HTLV-I is known to suppress T-cell immunity. A likely linkage between HTLV-I and some infections due to opportunistic agents such as leish-

mania in Kenya or trypanosoma in Venezuela [2] has been shown. In simians, SIV-1 (simian immunodeficiency virus type 1) retrovirus infection seems to enhance the occurrence of lepromatous leprosy [3]. In humans, three cases of active leprosy were found in association with HIV-l infection [4,5]. No case associated with HTLV-I has previously been reported. Both these infections have been proved to induce immunodeficiency; which one led to the other remains unknown.

which can present as a primary septicemia or a localized wound infection [l-3]. Although this bacterium often demonstrates an exceptional capability to invade soft tissues, skeletal muscles are rarely involved [3-51. We recently observed a case of massive generalized rhabdomyolysis complicating V. vulnificus sepsis, a unique presentation not previously described. A 51-year-old woman presented with a 24-hour history of severe lower leg pains, chills, vomiting, and a diffuse nonpruritic J.-R. HARLE P. DISDIER rash; she denied recent insect G. KAPLANSKI bites, toxin exposure, intraveC. TAMALET nous drug abuse, alcohol ingesC. WEILLER-MERLI Hopital de la Timone tion, or previous medications. J.-J. BONERANDI She worked in a sushi restaurant, Hopital Sainte Marguerite P.-J. WEILLER but she could not recall recent Hopital de la Timone shellfish consumption or visits to Marseille, France the beach. 1. Blattner WA. Human T lymphotropic viruses and Her temperature was 38.8”C; diseases of long latency. Ann Intern Med 1989: 111: blood pressure 100/60 mm Hg; 4-6. pulse 100 beats/ minute; respira2. Reeves WC, Saxinger C. Brenes MM, et al. Human T-cell lymphotropic virus type I (HTLV-I) seroepidetions 36/minute. She had marked miology and risk factors in metropolitan Panama. periorbital edema and ecchymoAm J Epidemiol 1988; 127: 532-9. sis, and a diffuse urticarial rash 3. Gormus BJ, Murphey-Corb M, Martin LN. et al. that promptly evolved into disInteractions between simian immunodeficiency VIrus and Mycobacterium leprae in experimentally increte, generalized, purpuric, maoculated rhesus monkeys. J Infect Dis 1989; 160: culopapular lesions. The pa405-13. tient’s peripheral pulses were 4. Lamfers EJP, Bastiaans AH, Mravunac M, Rampen FHJ. Leprosy in the acquired immunodeficiency only detectable via Doppler sosyndrome. Ann Intern Med 1987; 107: 11 l-2. nography, despite strong bila5. Janssen F. Wallach D. Khuong MA, et al. Associateral carotid and femoral pulses. tion de maladie de Hansen et d’infection par le virus The lower extremities had de I’immunodeficience humaine. Presse Med 1988; 17: 1652-3. marked muscle rigidity, tense edema, and tenderness, a process Submitted May 7. 1990, and accepted May 31. 1990 that later ascended to the upper extremities with excruciating pain, ultimately resulting in MASSIVERHABDOMYOLYSIS:A board-like stiffness and paralysis RAREPRESENTATIONOF PRIMARY W/?/O VULAWCUS of all four limbs. The white blood cell count was SEPTICEMIA 2,000/mm3 with 36% segmented In recent years, attention has forms and 24% bands; the crebeen focused on the seriousness atine kinase level was 9,260 U/L. and often fatal consequences of The bleeding time, prothrombin Vibrio vulnificus infections, time, and partial thromboplastin

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time were prolonged and the platelet count was 22,000/mm3. Toxicology screening showed no drugs or alcohol. The patient’s condition rapidly deteriorated as she developed anuria, metabolic acidosis, profuse rectovaginal bleeding, respiratory failure, and coma. Despite broad-spectrum antibiotics and aggressive supportive measures, the patient died 9 hours after her admission. All three blood cultures grew V. vulnificus. A limited autopsy showed micronodular cirrhosis of the liver. This patient’s clinical manifestations were those of overwhelming primary V. vulnificus septicemia accompanied by generalized rhabdomyolysis, disseminated intravascular coagulation, and renal failure. Kelly and McCormick [6] have reported a case with rhabdomyolysis localized to the left calf, myonecrosis, necrotizing vasculitis, and direct bacterial infiltration of the muscle tissues. A clinical review by Klontz et al [2] reported one of 37 cases of primary V. vulnificus septicemia with myonecrosis limited to one of the limbs. Two other cases [7,8] of localized myonecrosis have been described. To our knowledge, no other case of primary V. vulnificus septicemia has been reported with the extensive, diffuse rhabdomyolysis noted in our patient. In the absence of other predisposing causes of rhabdomyolysis [9], we suspect the bacteria gained intravascular access via the digestive tract, spreading hematogenously and seeding the muscles of her limbs. Tissue damage was probably the result of cytotoxins and concomitant necrotizing vasculitis [l-6] associated with V. vulnificus infections. Massive muscle edema led to high pressures within the muscle compartments, obstruction of blood flow, loss of peripheral pulses, and aggravation of the ongoing myonecrosis. Acute

The rare primary intracranial meningeal lymphoma (PIML) usually manifests as a solitary extra-axial mass that resembles an intracranial meningioma. An unusual presentation characterized a recent patient: a 21-year-old woman who had a subacute meningeal syndrome presenting with behavioral change and headache. No mass lesion was found by initial computed tomographic (CT) and magnetic resonance imaging (MRI) scans of the head. Brain biopsy revealed a primary meningeal lymphoma. This case is only the second such instance of PIML with a subacute meningeal syndrome. Primary central nervous sysANTONIO FERNANDEZ,M.D. FEDERICOR. JUSTINIANI, M.D. tem (CNS) lymphoma accounts Mount Sinai Medical Center Miami Beach, F~OIT$ for 1.5% of primary brain tumors and is usually intraparenchymal. University of Miami School of Except in very rare instances, the Medicine histology of primary intracranial Miami, Florida malignant lymphomas is of the 1. Morris JG Jr. Vibrio vulnificus-a new monster of non-Hodgkin’s type. This neothe deep? Ann Intern Med 1988; 109: 261-3. plasm may also present in the 2. Klontz KC, Lieb S, Schreiber M, Janowski HT. Baldy LM, Gunn RA. Syndromes of Vibrio vulnificus spinal epidural space. In rare ininfections. Clinical and epidemiologic features in stances, it is limited to the cranial Florida cases, 1981-1987. Ann Intern Med 1988; meninges. In these cases, CT 109: 318-23. 3. Case records of the Massachusetts General Hosscans have usually revealed intrapital. Weekly clinicopathological exercises (case 41cranial lesions suggesting lym1989). N Engl J Med 1989; 321: 1029-38. phoma, meningioma, or other ex4. Howard RJ. Pessa ME, Brennaman BH, Ramphas tra-axial neoplasm. This is a case R. Necrotizing soft-tissue infections caused by marine vibrios. Surgery 1985; 98: 126-30. with a highly unusual presenta5. Hoffman TJ. Nelson B, Darouiche R, Rosen T. tion for PIML in a patient who vibrio vulnificus septicemia. Arch Intern Med 1988; could not be shown to have ac148: 1825-7. quired immunodeficiency syn6. Kelly MT, McCormick WF. Acute bacterial myositis caused by Vibrio vulnificus. JAMA 1981; 246: drome (AIDS). 72-3. A 21-year-old woman was ad7. Roland FP. Leg gangrene and endotoxin shock mitted to the psychiatry service due to Vibrio parahaemolybcus-an infection acin April 1988 after displaying quired in New England coastal waters. N Engl J Med 1970; 282: 1306. catatonic posturing and poor per6. Fernandez CR, Pankey GA. Tissue invasion by unsonal hygiene. Her evaluation at named marine vibrios. JAMA 1975; 233: 1173-6. that time included an MRI scan 9. Gabow PA, Kaehny WD, Kelleher SP. The spectrum of rhabdomyolysis. Medicine (Baltimore) of the brain, which revealed mod1982; 61: 141-52. erate enlargement of the lateral 10. Bowdre JH, Hull JH, Cocchetto DM. Antibiotic ventricles attributed as likely to efficacy against Vibrio vulnificusin the mouse: supe reflect congenital hydrocephalus. riority of tetracycline. J Pharmacol Exp Ther 1983; 225: 595-8. She improved with supportive care and nutritional support and Submitted January 29, 1990, and accepted in revised form May 31, 1990 was discharged. The woman

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renal failure was probably the result of rhabdomyolysis [9]. This patient, a waitress in a sushi restaurant, with underlying liver disease, was at high risk for the development of this infection [l-3]. In view of the popularity and wide commercial distribution of oysters and shellfish for raw consumption in the United States, physicians should strongly consider primary V. vulnificus septicemia when a patient with underlying liver disease presents with a prominent skin rash, generalized rhabdomyolysis, and sepsis. Early recognition and treatment with intravenous tetracycline [2,3,5,10] may potentially influence the often fatal outcome of this devastating illness.

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Massive rhabdomyolysis: a rare presentation of primary Vibrio vulnificus septicemia.

, BRIEFCLINICALOBSERVATIONS LEPROMATOUSLEPROSYAND SEROPOSITIVITYFOR HTLV-I Leprosy and HTLV-I (human Tcell lymphotropic virus type I) infection are b...
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