Untreated thyrotoxicosis as a manifestation of anorexia nervosa

A patient with anorexia nervosa refused treatment for her thyrotoxicosis for 18 years in an attempt to keep her weight low. Severe congestive heart failure and impending thyroid storm prompted her family to force her to seek medical attention.     

A dancer with rheumatoid arthritis: a role for biologics

A case study is presented in which a student dancer who developed rheumatoid arthritis during her degree course was able to complete her course with meticulous treatment including the use of biologics. She describes her own reactions to the disease occurring at this crucial phase in her career.     

Hyperthyroidism as a cause of persistent vomiting

A 32-year-old woman presented with persistent vomiting, epigastric pain and weight loss. A sinus tachycardia was the clue to the diagnosis of hyperthyroidism due to Graves' disease. On treatment with propylthiouracil and a beta-blocking agent, her symptoms resolved within one day, even though her free thyroxine level was still high. Hyperthyroidism is an uncommon, but previously reported cause of persistent vomiting.     

Pulmonary lymphangioleiomyomatosis: a case of progesterone receptor positive lymphangioleiomyomatosis treated with medroxyprogesterone, oophorectomy and tamoxifen

A 28-year-old woman with lymphangioleiomyomatosis had severe changes in the chest radiograph and recurrent pneumothoraces. Pathological tissue from the lung was progesterone receptor positive. After treatment with medroxyprogesterone her condition improved, but did not become clinically stable until after bilateral oophorectomy. Even after oophorectomy her condition deteriorated, when the dose of medroxyprogesterone was decreased. For one year after oophorectomy she has had no episodes of pneumothorax or breathlessness. In order to depress oestrogen activity further the anti-oestrogen drug tamoxifen was added to her usual treatment with medroxyprogesterone. During 2 weeks of treatment with tamoxifen and medroxyprogesterone marked increase of the changes in the chest radiograph were seen and a pneumothorax occurred. After discontinuation of tamoxifen the condition improved and has since been stable on medroxyprogesterone treatment.     

Lactate dehydrogenase (LDH)-linked immunoglobulin in a patient with Graves' disease treated with methimazole.

A 26-year-old woman who received methimazole treatment for Graves' disease is discussed. Two months following treatment, her serum GOT level rose to 45 K.U, her GPT to 60 K.U, and her lactate dehydrogenase (LDH) to 645 W.U; a hepatic disorder was then suspected. Later, the serum GOT and GPT concentrations decreased to a normal range, but her serum LDH continued to maintain a high level. An LDH isoenzyme analysis showed an abnormally broad LDH. The IgG that was linked to the LDH is suspected to have been the result of her underlying autoimmunity, the methimazole treatment, and the development of her hepatic disorder. Thus, this IgG was thought to be the autoantibody to LDH.     

Campylobacter fetus as cause of prosthetic valve endocarditis

A 65-year-old woman who had previously undergone aortic root replacement with a bioprosthetic valve (Bentall operation) in treatment of annuloaortic ectasia became feverish after developing dental caries and was admitted to our hospital. Transesophageal echocardiography showed an 18 × 4-mm vegetation on her prosthetic valve. Campylobacter fetus was isolated on blood cultures, and she was diagnosed with infectious endocarditis. Aggressive combined antibiotic treatment was effective for her recovery. C. fetus infection is a rarely reported cause of prosthetic valve endocarditis.     

Porphyria cutanea tarda in a patient with acute leukemia

A 20-year-old woman with acute nonlymphocytic leukemia in remission was treated postremission with intermittent courses of intensive chemotherapy. She required blood and platelet transfusions following therapy, and oral contraceptives were used to suppress her menses while she was thrombocytopenic. One and one-half years after treatment began, she developed blisters on her hands, hypertrichosis, and dark urine. The diagnosis of porphyria cutanea tarda was made. This report discusses the possible precipitating cause(s) and the subsequent treatment of both her porphyria and her leukemia.     

Falling asleep

We describe a 74-year-old woman who presented with a history of falling from bed in association with vivid dreams and physical violence towards her spouse. A clinical diagnosis of rapid eye movement sleep behaviour disorder was made and complete resolution of her symptoms was achieved with first line treatment.     

Behaviour Therapy for Phobia of Venepuncture

A 28-year-old female insulin-dependent diabetic patient developed a severe phobia to venepuncture which seriously interfered with her medical treatment. A two-month course of behaviour therapy successfully alleviated the phobia.     

Complete response to treatment with hepatic arterial infusion of cisplatin powder (IA-call) in a case of hepatocellular carcinoma with multiple lung metastases]

A 74-year-old woman was admitted to our hospital to treat her hepatocellular carcinoma (stage IVB) with multiple lung metastases. She was treated with 3 times of hepatic arterial infusion of cisplatin powder (IA-call). After the treatment, liver mass and lung tumors were disappeared and high levels of tumor markers (AFP and PIVKA-II) were markedly decreased. These data revealed that a complete response (CR) was obtained for her. She has still been maintained in CR for 2 years since the first treatment.     

Effect of intravenous immunoglobulin on a spontaneous inhibitor to factor VIII

A 71-year-old woman with a spontaneous anti-factor VIII inhibitor fractured her right wrist and 2 months later her left femur. She received treatment with porcine factor VIII for the first fracture and developed a secondary anti-porcine antibody response (from 10 to 200 Bethesda units). Following the second fracture she received intravenous immunoglobulin (i.v. IgG) (0.4 g/kg day for 5 days) in an attempt to reduce antibody activity. Despite further treatment with porcine factor VIII, the antibody level declined instead of rising as expected and the anti-human antibody activity also declined. We were not able to demonstrate neutralizing activity to her antibody but did demonstrate a reduced helper: suppressor ratio and reduced B-cell numbers and function after treatment with i.v. IgG. These changes were transient and as B-cell function improved over the following 4 months, her anti-human activity returned toward its previous level. Anti-porcine activity remained at its previous low level. We speculate that one of the mechanisms of action of i.v. IgG may be a direct cellular effect influencing both T-suppressor and B-lymphocyte function.     

Treatment of a case of severe insulin resistance as a result of a PIK3R1 mutation with a sodium–glucose cotransporter 2 inhibitor

A Japanese woman aged in her late 30s with severe insulin resistance and bodily features including a triangular face, prominent forehead, small chin, large and low‐set ears, and ocular depression was investigated. A similar phenotype was not observed in other family members with the exception of her son, suggesting that the condition was caused by a de novo mutation that was transmitted from mother to son. Exome analysis showed the presence in the proband and her son of a c.1945C>T mutation in PIK3R1, a common mutation associated with SHORT (short stature, hyperextensibility of joints and/or inguinal hernia, ocular depression, Rieger anomaly, and teething delay) syndrome. Administration of a sodium–glucose cotransporter 2 inhibitor lowered the proband's hemoglobin A_1c level and allowed a reduction in her insulin dose without treatment‐related adverse events including ketoacidosis, exaggerated loss of body mass or hypoglycemia. Sodium–glucose cotransporter 2 inhibitors might thus offer an additional option for the treatment of genetic syndromes of severe insulin resistance.     

Polycythaemia Vera and Chorea

Polycythaemia vera is often associated with neurological syndromes, but it is rare for it to present as chorea. A 71-year-old woman presented totally incapacitated by her gross chorea and responded dramatically to treatment of her underlying polycythaemia vera. A haematological relapse was accompanied by a mild recurrence of symptoms, which again improved after further venesection. The possible pathogenesis is discussed.     

A case of middle ear tuberculosis; PCR of the otorrhea was useful for the diagnosis]

A 26-year-old female was admitted to our hospital with complaints of fever, cough, otorrhea and otalgia and progressive hearing loss of her left ear. Smears of her sputum were positive for acid-fast bacilli. Smears of her otorrhea were negative for acid-fast bacilli but PCR of her otorrhea was positive. Chest X-ray showed infiltrative shadows with the cavity. She was diagnosed as middle ear tuberculosis associated with pulmonary tuberculosis. After anti-tuberculous chemotherapy, fever, cough, otorrhea and pain of her left ear were improved, but her hearing level was not improved. In the case of middle ear tuberculosis, it is necessary to make an early diagnosis and treatment. This is the first reported case in Japan in which PCR of the otorrhea is positive.     

The effect of tacrolimus on lower extremity ulcers: a case study and review of the literature

Tacrolimus, a calcineurin inhibitor, has become an increasingly valuable tool in the treatment of dermatological disorders during the last few years. However, its effect on wound healing is still under investigation and remains the subject of safety concerns. A 75-year old woman with lichen planus, diabetes mellitus, and a foot ulcer was prescribed tacrolimus for the treatment of her lichen planus. After starting the treatment, her ulcer healed and the medication was discontinued. Shortly thereafter, re-ulceration occurred, treatment was re-introduced, and the wound continued to heal until treatment was discontinued. When the third course of tacrolimus was prescribed, the ulcer started healing again but a diagnosis of osteomyelitis necessitated surgical intervention. A review of the literature suggests that tacrolimus does not adversely affect healing in vivo or in vitro and may facilitate healing lower extremity skin ulcers, especially those of inflammatory origin. Studies are needed to clarify which lower extremity wounds would improve with tacrolimus.     

Pulmonary cryptococcosis presenting as endobronchial lesions in a patient under corticosteroid treatment

We report a case of pulmonary cryptococcosis in an immunocompromised patient who had focal endobronchial lesions. When we first examined her, she complained of a productive cough that had lasted for two months. Seventeen months prior to this examination, she had been diagnosed with Sjogren syndrome and Sweet syndrome. Since that diagnosis, her condition had been maintained with low-dose prednisolone. We performed a chest CT scan, which revealed a mass, 3 cm in diameter, in the upper lobe of her left lung. A bronchoscopic examination revealed 3 white, elevated lesions in the upper lobe bronchus of her left lung. After 40 days of treatment with fluconazole, the shadow of her lung mass decreased in size to that of a scarred lesion and her white, elevated bronchial lesions disappeared.     

Radiation retinopathy--a case report

Radiation retinopathy is one of ocular complications in radiation therapy for many diseases. It is a slowly progressive, delayed-onset disease of retinal blood vessels. A 49 year-old Chinese female suffered from blurred vision for 2 years progressively. The best corrected visual acuity was 0.02 and 0.06 in her right and left eyes respectively. Ophthalmic fundus examination showed hard exudate, retinal hemorrhage, and macular edema in both her eyes. Tracing back her history, she had nasopharyngeal carcinoma and received irradiation treatment 11 years ago. She denied diabetic mellitus or other systemic disease. After laser photocoagulation treatment, she had better vision with the best corrected visual acuity 0.2 and 0.3 in her right and left eyes respectively 1 year later.     

Association of anti-tumor necrosis factor therapy with the development of multiple sclerosis

A 35-year-old woman with a history of indeterminate colitis developed symptoms of multiple sclerosis after treatment with infliximab. Neurologic examination confirmed upper and lower extremity motor and sensory deficits. MRI showed multiple enhancing white matter lesions distributed throughout her brain as well as her thoracic spine. There may be a link between inflammatory demyelinating disease of the central nervous system and anti-tumor necrosis-alpha therapy. This case report describes the onset or worsening of a demyelinating process after the initiation of infliximab therapy in a patient with indeterminate colitis.     

Cushing's disease in a patient with a 'nonfunctioning' pituitary tumor. Spontaneous development and remission

A 40-year-old woman had visual loss and a large nonfunctioning pituitary tumor. After partial surgical resection and radiation treatment, clinical and biochemical evidence of Cushing's disease developed. The pituitary source of her adrenocorticotropic hormone hypersecretion was documented on selective venous sampling. After 18 months of medical therapy with metyrapone and aminoglutethimide, the patient experienced a spontaneous remission of her hypercortisolism. A "nonfunctioning" pituitary tumor has a hypersecretory potential.     

Steroidrefrakt?re Graft-versus-host-Erkrankung

A 42 year-old woman develops steroid refractory graft-versus-host disease (GVHD) after second allogeneic stem cell transplantation for acute myelogenous leukemia with severe GVHD of her skin with blisters, severe GVHD of her gut with watery and bloody diarrhea and GVHD of her liver with cholestasis. In a further attempt to control GVHD extracorporeal photochemotherapy is administered. The treatment exposures peripheral mononuclear cells to photoactivated psoralen before they subsequently are given back to the patient. This approach apparently offers selective immune tolerance.