Unilateral internuclear ophthalmoplegia and recovery with thiamine in Wernicke syndrome.

Internuclear ophthalmoplegia is usually caused by multiple sclerosis, tumors, or vascular lesions of the brain stem. We report a patient with Wernicke syndrome who presented with a right-sided internuclear ophthalmoplegia. He recovered completely with intravenous thiamine (vitamin B1). There were no lesions in the magnetic resonance image (MRI) of the brain, suggesting a derangement at the cellular level as the cause.

Angiotropic large cell lymphoma presenting as fever of unknown origin.

BACKGROUND: Fever has never before been described as the chief complaint and major finding in angiotropic large cell lymphoma (ALCL). ALCL is a rare and usually fatal intravascular tumor characterized by a widespread proliferation of malignant mononuclear cells within vessels of small caliber, causing their blockage. The majority present as high-grade, B-cell lymphomas with a predilection for the central nervous system and the skin. CASE REPORT: We report a 61-year-old woman who presented with a fever of unknown origin (FUO) that lasted 9 weeks from onset to death. To our knowledge, this is the first case of ALCL to present solely as a FUO, and the second case of ALCL to be diagnosed by muscle biopsy. CONCLUSION: We suggest that this rare malignancy (ALCL) be considered in the differential diagnosis of FUO.

Esophageal papillomatosis from human papilloma virus proven by polymerase chain reaction.

Human papilloma viruses (HPVs) are known to infect the genitourinary tract, the skin, the anal canal, and the upper respiratory tract. Esophageal papillomas and especially HPV-induced squamous papillomas of the esophagus are rare. The authors report a case of extensive HPV-induced esophageal polyposis, which was probably sexually transmitted. The 53-year-old female patient presented with chronic diarrhea and had occult blood in the stool. She underwent esophagogastroduodenoscopy, at which time multiple esophageal polyps were observed and biopsy specimens obtained. Histologic evaluation was consistent with benign papillomas. Polymerase chain reaction and DNA hybridization of the biopsied tissue specimens confirmed the diagnosis of HPV infection. Because of our observation and because of HPV's relationship to cervical and esophageal cancer, further evaluation of HPV as the cause of esophageal papillomatosis and as a risk factor for esophageal cancer is warranted.

Acalculous cholecystitis and septicemia caused by non-O1 Vibrio cholerae: first reported case and review of biliary infections with Vibrio cholerae.

The first case of septicemic acute acalculous cholecystitis caused by non-O1 Vibrio cholerae is described in a healthy traveler, and biliary tract infections from V. cholerae are reviewed. Immediately after a vacation in Cancun, Mexico, a 55-year-old man developed acute cholecystitis. Blood and bile cultures grew non-O1 V. cholerae. At surgery, the gallbladder was acalculous, inflamed, distended, and nearly ruptured. Pathogenetic factors may have included diarrhea prophylaxis with bismuth subsalicylate, distension of the gallbladder from illness-induced fasting, and bacterial toxins in the gallbladder. The patient received i.v. cephapirin, followed by oral cephradine for a total of 10 days, and he made a quick and complete recovery. V. cholerae should be considered in the differential diagnosis of persons from endemic areas who present with cholecystitis or acute jaundice.

Blastomycotic cranial osteomyelitis.

This is the second case report of a temporal bone osteomyelitis caused by Blastomyces dermatitidis, which presented as a chronic serous otitis media. The presenting serous otitis media was refractory to conventional medical and surgical management and progressed to a temporal bone osteomyelitis prior to diagnosis. B. dermatitidis is a rare fungal pathogen that causes a systemic pyogranulomatous disease that primarily manifests itself in the skin, bones, pulmonary, and genitourinary systems. If left untreated it is associated with a high rate of mortality. The otologic presentation of this rare disease is emphasized, while the clinical and therapeutic features are reviewed.

Leprosy in six isolated residents of northern Louisiana. Time-clustered cases in an essentially nonendemic area.

Northern Louisiana has been essentially free of indigenous leprosy, and now it is not. Six new cases of leprosy have been diagnosed: three in 1986, the other three in 1985, 1983, and 1982, respectively. The patients had been lifelong residents of six scattered rural parishes. Leprosy had never been reported from five of them. No patient had had contact with human leprosy. The patients were white; four were women; the mean +/- SD age at onset was 60.3 +/- 16.4 years (age range, 31 to 80 years); and the mean +/- SD interval to diagnosis was 1.2 +/- 1.4 years. One patient had Hodgkin's disease at the age of 25 years and leprosy at the age of 31 years; another patient had cervical carcinoma. All rural northern Louisiana residents coexist with armadillos (Dasypus novemcinctus), some of which are infected with Mycobacterium leprae, the significance of which is unknown. Hypothetically, exposure to an unknown human case, reactivation of "asymptomatic" leprosy through immunosenescence or immunosuppression, or infection from an environmental source might have occurred. Because the patients lacked contact, travel, residence, and exposure risk factors, the origin of leprosy in the new indigenous cases is noteworthy and is not understood.

Wegener granulomatosis and trimethoprim-sulfamethoxazole. Complete remission after a twenty-year course.

Wegener granulomatosis was diagnosed in a 42-year-old woman in 1965. Although a regimen of azathioprine and prednisone was helpful, the disease progressed. Cyclophosphamide was added to this regimen in 1969. On three separate occasions her disease relapsed when cyclophosphamide therapy was discontinued. In 1984, she developed cyclophosphamide-resistant disease and drug toxicity. We were able to discontinue cyclophosphamide therapy after a trimethoprim-sulfamethoxazole regimen that was begun in February 1985 led to rapid improvement, a fall in the erythrocyte sedimentation rate, and a complete remission. Her 22-year survival is the longest one reported. Because patients with Wegener granulomatosis sometimes respond to trimethoprim-sulfamethoxazole, this therapy deserves careful study and implies that Wegener granulomatosis is an as yet unidentified infection.

Inguinal abscess caused by Rhizopus rhizopodiformis: successful treatment with surgery and amphotericin B.

Rhizopus rhizopodiformis has seldom been isolated from human mucormycosis. We report the first subcutaneous abscess to be caused by this fungus. It occurred in a diabetic man and presented as an inguinal mass, suggestive of a hernia, superficial to his cadaveric renal transplant. The fungus was readily isolated from pus inoculated onto blood and chocolate agars after a short incubation. The patient was cured by surgical drainage and treatment with 2.0 g of intravenous amphotericin B. Complete identification of such isolates is recommended.

Five-year follow-up of a man with subcutaneous mycetomas caused by Microsporum audouinii.

A black man with subcutaneous mycetomas caused by Microsporum audouinii was treated by a combination of griseofulvin, 18.5 g of amphotericin B, excisional surgery, and later, ketoconazole, resulting in a satisfactory arrest or cure of the clinical illness. Complications of therapy included residual impaired renal function and a change in hair color from black to a rust brown color. The continued use of the term mycetoma to describe such lesions is justified.

Mycetoma caused by microsporum audouinii. First reported case.

A 25-year-old black man had generalized tinea corporis for 19 years and had associated subcutaneous mycotic granuloma of the scalp and nape of the neck for 17 years. The dermatophyte from the skin was identified as Microsporum audouinii and responded to oral griseofulvin treatment. Biopsy specimens of the subcutaneous nodules revealed yellowish granules containing masses of irregularly septated hyaline hyphae with frequent vesicles. This fungus differed only in gross colonial morphology from the M. audouinii isolated from the skin and has also been identified as M. audouinii. The subcutaneous nodules were treated and controlled by amphotericin B and are still under treatment.

The hypereosinophilic syndrome: analysis of fourteen cases with review of the literature.

Several closely related disease entities make up the idiopathic hypereosinophilic syndrome (HES). The syndrome is manifest by persistent and prolonged eosinophilia with organ damage. A group of 14 patients had hematologic, cardiac, and neurologic abnormalities attributable to this disease. Patient survival and response to chemotherapy was significantly better in this group than in previously reported patients. The etiology of HES remains unknown, as does the mechanism of tissue damage.