Anti-transfer RNA antibodies in two patients with pulmonary fibrosis, Raynaud's phenomenon and polyarthritis.

Patient W.S. (a 61-year-old woman) and patient T.M. (a 41-year-old man) developed recurrent fevers, polyarthritis, Raynaud's phenomenon and interstitial pulmonary fibrosis without apparent polymyositis. From HeLa cell extracts, sera from both patients immunoprecipitated all species of intact and deproteinised tRNAs. To identify the antibody binding site more precisely, tRNAs transcribed in vitro from cloned Escherichia coli tRNA genes and various mutants were prepared and used as antigens for immunoprecipitation. When the TpsiC loop, or the D loop were deleted, such mutants were not bound by both sera, suggesting that the D and TpsiC loops were required for antibody binding. Abrogation of tRNA binding occurred when 18G of tRNATrp was replaced with 18A to break the tertiary L-shape structure of tRNA. These results strongly suggest that sera from W.S. and T.M. recognise the tertiary conformation of L-shaped tRNA which is constructed with both D and TpsiC loops. These autoantibodies may also serve as a marker for a new subset of patients with connective tissue diseases that is distinct from anti-aminoacyl-tRNA synthetase syndrome.

Progressive systemic sclerosis complicated with primary cerebral malignant lymphoma.

We describe a 68-year-old man who developed primary cerebral malignant lymphoma 5 years after the onset of progressive systemic sclerosis (PSS). There was no association with Sjögren's syndrome. This is the first report of PSS complicated with primary cerebral malignant lymphoma.

Turner's syndrome associated with Hashimoto's thyroiditis and sarcoidosis: a case report.

A 32-yr-old Japanese female who was suffering from Turner's syndrome associated with Hashimoto's thyroiditis and sarcoidosis is reported. It is known that Hashimoto's thyroiditis is not infrequently complicated with Turner's syndrome on the basis of an abnormal X chromosome. In this case, sarcoidosis occurred 4 months after Hashimoto's thyroiditis. This extremely interesting coincidence of Hashimoto's thyroiditis and sarcoidosis in Turner's syndrome has not been reported to date to our knowledge.

Direct identification of lung cancer cells producing granulocyte colony stimulating factor with monoclonal anti-G-CSF antibodies.

This is a case report of an 82-year-old man with squamous cell lung cancer which produced G-CSF. The leukocyte cell count was elevated to 94,400/mm3. The serum G-CSF concentration was also elevated to 351 pg/ml. This is the first report to demonstrate, by application of immunohistochemical staining techniques directly to tumor tissue obtained at autopsy, squamous carcinoma cells which produce G-CSF. The tumor cells were stained with monoclonal anti-G-CSF antibodies.

Hemoglobin Bethesda causing polycythemia in a Japanese family.

A family with Hemoglobin Bethesda is reported. A 23-year-old man was hospitalized for the evaluation of polycythemia. Analysis of hemoglobin using high pressure liquid chromatography showed the presence of hemoglobinopathy. Separation of globin into alpha and beta chains revealed approximately 50% of the beta chain to be abnormal. Analysis of the DNA sequence of the beta chain gene identified Hb Bethesda. The family study disclosed that his father and sister also had the same hemoglobinopathy. This case is the first report of Hb Bethesda in Japan.

[A case of shigellosis due to Shigella dysenteriae 1 which was thought to be infected in domestic area].

A 43-year-old man was admitted to our hospital because of bloody diarrhea and abdominal pain on January 10, 1989. On laboratory examination S. dysenteriae 1 was isolated from his stool, therefore he was diagnosed as shigellosis. After the administration of ofloxacin was started, S. dysenteriae 1 was immediately eradicated. But his diarrhea persisted until the 19th day from the onset of the illness and his abdominal pain persisted until the 21st day. He had never been overseas and he had never eaten any imported uncooked food recently. Only 14 cases of shigellosis due to S. dysenteriae 1 were found in the last ten years in Japan. All of them were overseas travellers. Although we were not able to determine the source of infection in our case, we inferred that he was primarily infected with S. dysenteriae 1 in Japan.

[Clinical studies on the treatment of Campylobacter enteritis--emergence of quinolone-resistant Campylobacter jejuni after treatment with new quinolones].

In recent years, new quinolones such as ofloxacin (OFLX) and tosufloxacin (TFLX) have been frequently used in the treatment of bacterial enteritis caused by unknown organisms. The agent of first choice for the treatment of Campylobacter enteritis is one of the macrolides, but new quinolones are often administered accidentally to adult patients with Campylobacter enteritis. We have detected quinolone-resistant strains of Campylobacter jejuni (C. jejuni) after the treatment of some patients with new quinolones, and accordingly we reviewed the treatment of Campylobacter enteritis. We experienced 178 adult patients with Campylobacter enteritis from January 1989 to November 1991 at our hospital. From them, we selected 52 patients (32 males and 20 females) in whom stool culture were performed both before and after treatment. The initially administered antimicrobial agent was a macrolide (rokitamycin, RKM) in 6 cases, a new quinolone in 22 cases, and kanamycin (KM) in 24 cases. The new quinolone used was OFLX in 17 cases, TFLX in 3 cases, and norfloxacin (NFLX) in 2 cases. Fifty-one of the 52 C. jejuni strains isolated before treatment were susceptible to OFLX and erythromycin (EM) according to antimicrobial disc susceptibility tests. C. jejuni was eradicated in all patients treated with RKM or KM. However, treatment failed to achieve bacteriological cure in 8/22 (36.4%) patients given new quinolones. In these patients, the strains of C. jejuni isolated before treatment were susceptible to OFLX, but the strains isolated after treatment were all resistant to OFLX according to disc susceptibility tests.(ABSTRACT TRUNCATED AT 250 WORDS)

[A fatal case of acute enteritis caused by Salmonella Weltevreden after travel to Indonesia].

A 67-year-old male was admitted to our hospital because of watery diarrhea and pre-shock status at 10:30 am on March 20, 1995. He had travelled to Bali Island in Indonesia from March 13 to March 18, 1995. On admission, his systolic blood pressure was 60 mmHg and body temperature was 35.2 degrees C. His skin was very dry. Laboratory tests showed that s-Cr was 6.3 mg/dl and CPK was 5620 IU/l. A massive fluid transfusion was given immediately and then his blood pressure rose to 158/92 about two hours after admission. However, he developed a high grade fever and systemic cyanosis in the evening of the first hospital day and died at 0:20 am on March 21st. Salmonella Weltevreden was detected in the fecal and blood cultures obtained on admission. We considered that his acute renal failure was attributable to rhabdomyolysis due to dehydration and that the cause of death was probably septic shock. The patient had a previous history of cholecystectomy ten years ago and also suffered from hypertension, but his general condition was not so bad before this episode. Therefore, we were surprised that his illness became so severe. This case emphasizes that Salmonella enteritis may occasionally be a serious and lethal disease.

[A case report: horizontal infection of typhoid fever from daughter to mother (by suppository maneuver)].

A 22-year-old female was admitted to our hospital because of general fatigue and continuous high fever. Salmonella Typhi was detected from her blood. Her mother, 44-year-old female was admitted to our hospital a few days later because of high fever. She had nursed to her daughter and inserted suppositories to her daughter several times. S. Typhi was also detected from her blood. No other member of her family was infected with S. Typhi. From the study of the route of the infection, we are confident that S. Typhi was delivered from daughter to mother by insertion of suppositories.

[A case of infective endocarditis with purpura and macroscopic hematuria as initial manifestations].

A 44-year-old female admitted to our hospital because of fever, purpura and macroscopic hematuria. She had been diagnosed as having ventricular septal defect (VSD). She noticed purpura with pain on bilateral legs and macroscopic hematuria since September 18, 1994. Three weeks later she also manifested a fever. Physical examination of admission revealed numerous purpura and leg edema. Laboratory data showed macroscopic hematuria, marked anemia (Hb 3.3 g/dl), leukocytosis, azotemia (Cr 2.7 mg/dl) and positive acute phase reactants. Increased serum immune complex level and hypocomplementemia were also found. The diagnosis of allergic purpura was made initially, but positive blood culture of Streptococcus mitis and the detection of vegetation attached to the right ventricular wall near the ostium of the VSD made the definite diagnosis of infective endocarditis (IE). Chemotherapy with PCG was started for two weeks but with no effect. The chemotherapy was altered to panipenem/betamipron with a daily dose of 3 g, Then, her fever fell and purpura, macroscopic hematuria and renal failure gradually disappeared. In this case, the cause of renal manifestations was considered to be immune complex glomerulonephritis. This is the first report of IE with macroscopic hematuria due to immune complex glomerulonephritis.

[A case of brain abscess accompanied with sudden-onset hemiplegia as initial manifestation].

A 58-year-old male experienced a sudden stroke-like onset of right hemiplegia and numbness of his right upper limb while engaged in his desk-work on April 7, 1997. He had a past history of diabetes mellitus and hyperlipidemia. On admission, he had no fever and the blood pressure was 140/70 mmHg. General physical examination was unremarkable. Neurological examination showed 4/5 strength of his right unilateral extremities and numbness of his right upper limb. Clinical features and computed tomography (CT) without contrast medium at the onset of hemiplegia suggested a stroke. Seven days after admission, his consciousness worsened and body temperature fluctuated between 37 and 38 degrees C. Subsequent Gd-enhanced magnetic resonance (MR) which demonstrated an irregular shaped ring-enhancement lesion and lumbar puncture 9 days after admission was compatible with the diagnosis of brain abscess. Surgical drainage confirmed the presence of brain abscess due to alpha-streptococcus. It improved following surgical drainage and antibiotic therapy with PAPM.BP 2 g/day and PIPC 4 g/day. An afebrile patient of sudden stroke-like onset may be a rarity to be added to the differential diagnosis of brain abscess.

[A case of Plasmodium vivax malaria complicated with pancytopenia due to hypoplasia of the bone marrow].

The patient is a 39 year-old Japanese male who had traveled to Southeast Asia from March 14, 1987 and returned on April 2. On April 3 and 5, he had a high fever with chills and he was admitted to our hospital. Despite initial treatment with antibiotics, a high fever over 39 degrees C appeared with a 48 hour periodicity. On the 8th day after admission, malarial parasites were identified on the peripheral blood smear after repeated trials. Combined with a raised serum antibody titer, Plasmodium vivax malaria was diagnosed. He was successfully treated with the sulfadoxine 500 mg and pyrimethamine 25 mg (Fansidar) and body temperature was normalized after the 12th day. More interestingly, the patient showed pancytopenia without splenomegaly. The bone marrow aspiration revealed hypoplasia of erythroblasts, granulocytes and megakaryocytes. Because of this pancytopenia in the peripheral blood and hypoplasia of the bone marrow which improved after recovery from malarial infection, it was indicated that they were caused by the malarial infection. Generally, it is considered that anemia in malarial patients is caused by destruction of the blood cells by parasites and/or hypersplenism and compensatory hyperplasia of the bone marrow is seen. On the contrary, this case showed pancytopenia accompanied with hypoplasia of the bone marrow probably due to the malarial infection suggesting a new aspect of pathogenesis in the hematological abnormality of the malarial infection.

[Toxic shock-like syndrome after acupuncturation].

A 80-year-olkd male was admitted to our hospital because of severe pain and swelling on his left lower leg on January 23, 1996. He had received an acupuncture to both legs because of intermittent claudication once a week from July, 1995 to January 18, 1996. On the next day of the last acupuncture, pain and swelling on his left leg appeared. On admission, his left leg showed diffuse swelling and redness with blisters. We diagnosed this patient as toxic shock-like syndrome (TSLS), based on the rapid exacerbation of the skin changes, necrotizing superficial fasciitis, multiple organ failure with shock, and the detection of group A streptococcus from culture samples obtained from both skin blister and necrotic fascia. He recovered from the disease by amputation of the involved leg and antibiotic therapy. Acupuncture could have been the cause of streptococcal infection.

[A case report--typhoid fever complicated with liver and gallbladder abscess, treated for long-time as fever of unknown origin].

A 25-year-old male admitted to Kawasaki municipal hospital with the diagnosis of typhoid fever. He had noticed high fever since one month ago, and had been treated with prednisolone with the diagnosis of fever of unknown origin in a hospital. Then he had admitted to St. Marianna University Hospital, and Salmonella Typhi had been detected from his blood and stool. On admission, multiple liver abscess were detected by abdominal ultrasonography. S. Typhi in bile was not eliminated with CP and AMPC, but he was successfully treated with cholecystectomy and the chemotherapy of LVFX. Abscess formation was found in the resected gall bladder wall. Typhoid nodule in the lymph node, liver or other organs is a well known pathological change in the typhoid fever. But abscess formation in the liver or other organs is rare. In this case, multiple abscess is characteristic and this cause is thought to be induced by the factors that the period from onset of typhoid fever to beginning of effective therapy was too long, and that steroid therapy was done without antibiotic therapy.

[A case of Plasmodium falciparum malaria successfully treated with halofantrine].

A 44-year-old male, who had been to Lagos, Nigeria, was admitted to our hospital because of a high grade fever on July 20, 1993. On admission, Plasmodium falciparum was detected in his blood smears and the antibody titers against P. falciparum and Plasmodium vivax antigens were 1:256 and < 1:4 respectively by the indirect fluorescent antibody test. Therefore, he was diagnosed as having P. falciparum malaria. He was treated with halofantrine (Halfan: Smith Kline Beecham Pharmaceuticals, England), two tablets at six-hourly intervals, a total of six tablets (1500 mg). Parasites were cleared rapidly and remission was achieved without any adverse reactions. Halofantrine can therefore be recommended for the treatment of imported P. falciparum malaria.

[Case report: infective endocarditis caused by Streptococcus agalactiae].

A 42-year-old male was admitted to our hospital because of high grade fever on October 6, 1992. He had no history of cardiac and underlying disease. For the past 10 days, he had complained of high grade fever and noticed arthralgia on his left shoulder. Physical examination on admission revealed that there was a body temperature of 39.0 degrees C and tenderness in the left shoulder. There were no abnormal findings for the chest or abdomen. On the second hospital day, he developed a diastolic murmur which had not been present on admission. And blood culture was positive for Streptococcus agalactiae. Ultrasonic-cardiogram indicated the presence of vegetation. He was diagnosed as infective endocarditis and treated with PCG 20 million units/day, IPM/CS 2 g/day and ISP 400 mg/day. But he was not responding to the chemotherapy. Aortic valve replacement was done on 22nd, October. Valve surgery succeeded and he became well after that time. Endocarditis caused by S. agalactiae is extremely rare, and is an important condition which carries a high mortality. Only seven cases of S. agalactiae endocarditis have been reported in Japan. It is difficult to treat these cases with antibiotic therapy alone. Therefore, we suggest that early surgery should be considered in infective endocarditis caused by S. agalactiae.

[A case of bacillary dysentery caused by new quinolone-resistant Shigella flexneri 2a].

A 73-year-old male was admitted to our hospital because of detection of Shigella flexneri 2a from his stool. Antimicrobial treatment with levofloxacin (LVFX) was started, but could not eliminate the organism in the stool. In the examination of drug susceptibility, this strain was highly resistant to all new quinolones. The minimal inhibitory concentration of norfloxacin, ofloxacin and ciprofloxacin to this strain was 12.5 micrograms/ml, 6.25 micrograms/ml and 6.25 micrograms/ml, respectively. The dual mutations were detected in the codon 83 and 87 of the gyrA gene by sequencing the quinolone-resistance determining region (QRDR). There was, however, no significant difference between the intracellular uptake of ciprofloxacin in this strain and in the ciprofloxacin-sensitive strain. The amount of ciprofloxacin in this strain unchanged when carbonyl cyanide m-chlorophenyl hydrazone (CCCP) was added. These results suggest that the advanced resistance in Shigella flexneri against new quinolones could be acquired by only this dual mutations without the change of the active efflux mechanism.

[Case report: suppurative arthritis due to group B Streptococcus in a patient with rheumatoid arthritis].

A 62-year-old woman, who had been diagnosed as having rheumatoid arthritis (RA) with systemic amyloidosis and diabetes mellitus, was admitted to our hospital because of polyarthralgia on October 1, 1987. She had some subcutaneous nodules and rheumatic pleural effusion. Therefore she was treated with 20 milligrams of prednisolone (PSL) daily. On the ninth day after the beginning of steroid therapy, she complained of severe pain and a new swelling in her right knee joint. The knee joint aspirate on arthrocentesis yielded a pure growth of group B Streptococcus (GBS). Blood culture was also positive for GBS. Her suppurative arthritis gradually improved by treatment with penicillin G. However, after the discontinuance of PSL, her pleural effusion deteriorated and she died on January 10, 1988. To our knowledge, there have been no prior reports of group B streptococcal suppurative arthritis complicating RA in Japan.

[A case of vivax malaria in which remission was achieved by primaquine at 1.5 times the standard dose after two earlier relapses].

A 22-year-old male student, who had been to Indonesia, was admitted to our hospital because of a high grade fever on October 12, 1990. On admission, vivax malaria parasites were detected in his blood smears and the antibody titers against Plasmodium vivax and P. falciparum antigen were respectively 1:1024 and 1:64 by the indirect fluorescent antibody test. Therefore, he was diagnosed as having vivax malaria and was treated with sulfadoxine/pyrimethamine (Fansidar), followed by primaquine at 15 mg/day for 14 days. However, relapse occurred twice after treatment with the above-mentioned standard dose of primaquine. After his second relapse, he was treated with primaquine at 22.5 mg/day for 14 days and remission was finally achieved without any adverse reactions. The antibody titer against P. vivax did not re-increase during the first and second relapses.

[A case report of atypical tuberculous spondylitis].

A 22-year-old male was admitted to our hospital because of progressive weakness of bilateral lower limbs on October 29, 1992. He also complained of cough, sputum and back pain for 6 months. He had no history of underlying disease. His family history disclosed that his elder brother had tuberculous peritonitis. Chest radiogram on admission showed an abnormal shadow around the right first arch. Computerized tomogram revealed osteolytic lesions of the vertebral corpus and posterior elements at the level of C3, Th5, and Th11. Metastatic bone tumor was strongly suspected and surgical decompression was done on November 12. Histologic findings of the bone showed caseation and Langhans's giant cells, and acid fast bacilli were isolated. He was treated with anti-tubercular chemotherapy and showed remarkable relief of his symptoms over a period of 6 months therapy. This was a case of atypical tuberculous spondylitis. The main locus existed at the posterior elements of the vertebra and the distribution of vertebral lesions was multiple including cervical spine.