, 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
October
1990
The American
Journal
of Medicine
Volume
89
535
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
October
1990
The American
Journal
of Medicine
Volume
89
535
