Recurrent goiter, hyperthyroidism, galactorrhea and amenorrhea due to a thyrotropin and prolactin-producing pituitary tumor

A 22-year-old woman with recurrent goiter, hyperthyroidism, galactorrhea, and amenorrhea due to a pituitary tumor is described. She had been treated surgically twice for recurrent goiter with tracheal compression. Despite clinical signs of hyperthyroidism and slightly elevated plasma thyroid hormone levels (T4: 11 mug/dl; T3: 189 ng/dl), without thyroid hormone replacement therapy the basal TSH level was elevated up to 23 muU/ml and could not be suppressed by exogenous thyroid hormones: even when the serum thyroid hormone levels were raised into the thyrotoxic range (T4: 16.2 mug/dl T3: 392 ng/dl), the basal TSH fluctuated between 12 and 29 muU/ml. The basal PRL level was elevated up to 6000 muU/ml. The administration of TRH (200 mug iv) led only to small increments of TSH and PRL levels. Bromocriptin (5 mg p.o.) or l-dopa (0.5 g p.o.) suppressed TSH and PRL values significantly. After transsphenoidal hypophysectomy, TSH and PRL were below normal and the patient development panhypopituitarism. The adenoma showed two cell types which could be identified as lactotrophs and thyrotrophs by electronmicroscopy and immunofluorescence. From these data we conclude that the patient had a pituitary tumor with an overproduction of thyrotropin and prolactin.     

Phacotrabeculectomy with "smile" incision, punch, and single releasable suture

A 66-year-old female had ischemic heart disease due to left main lesion complicated with calcified ascending aorta, right pelvic carcinoma and liver cirrhosis. The combined operations with coronary artery bypass by left internal thoracic artery under the hypothermic ventricular fibrillation with cardiopulmonary bypass and right nephrectomy were performed successfully. Postoperatively Treadmill exercise test and scintigram revealed no ischemihc change. She is doing well. The aortocoronary bypass to left anterior descending artery using a left internal thoracic artery under hypothermic ventricular fibrillation with cardiopulmonary bypass might be one of surgical options for high risk patient.     

Reversible left intraventricular obstruction after treatment of pheochromocytoma and hyperthyroidism

The authors report an original case of the association of three pathologies: pheochromocytoma, hyperthyroidism and cardiomyopathy with left ventricular outflow tract obstruction. This type of cardiac disease has occasionally been described in cases of pheochromocytoma and are usually induced by the endocrine disturbance because they regress with treatment of the pheochromocytoma. The associated hyperthyroidism observed in this case is very rare and may have increased the left ventricular pressure gradient. Medical treatment before surgery of the pheochromocytoma was unusual in that a triple therapy was used including betablockers, classically contra-indicated in pheochromocytoma alone. In this case, it provided excellent control of the blood pressure and decreased the left ventricular obstruction during the perioperative period.     

Therapeutic concepts of proteinuria and intra-glomerular hypertension

Leukemia has rarely been reported as a late complication of 131I therapy, occurring mostly after cumulative doses of 800 mCi. We observed two cases of acute myeloid leukemia (AML) after 131I therapy for hyperthyroidism and thyroid carcinoma, respectively. The first patient was a 45-year-old woman treated with a single dose of 27 mCi 131I for hyperthyroidism. She developed AML (FAB M2) 14 months after receiving 131I; the second patient was a 44-year-old man affected by refractory thyroid carcinoma who received a total dose of 1 Ci 131I plus radiotherapy and developed AML (FAB M6) 8 years after the first exposure to 131I. Although it is a very rare event, the occurrence of leukemia after 131I treatment should be kept in mind, considering the widespread use of 131I, particularly in the treatment of hyperthyroidism, and the unfavorable outcome of secondary leukemia.     

Progresses in the study of general surgery in China, 1997

Late systemic venous baffle obstruction after Mustard repair for complete transposition of the great arteries is a recognized complication. Balloon-expandable intravascular stents have previously been used to relieve systemic baffle narrowing in children. We report a successful stent implantation to relieve symptomatic superior vena cava obstruction and baffle dehiscence after Mustard repair in an adult patient. She had been turned down for surgery due to right ventricular dysfunction. Eighteen months after the procedure, she remains symptomless.     

Hypothyroid coma and thyrotoxic crisis

Two clinical cases of endocrinologic emergency are presented: myxedema coma and thyrotoxic crisis. These are very severe situations with a high mortality rate. In myxedema coma it reaches 50% and in thyrotoxic crisis the range is between 25 and 30%. These entities are not the always present in mind because of their rarity. Consequently, they may well be undiagnosed. In our cases, the myxedema coma was initially diagnosed as brainstem stroke, and the hyperthyroidism was taken for concealed malignant tumor. The management of both situations is briefly commented.     

Expression of p53 in reactive mesothelium

An arteriovenous fistula originating from halfway down the circumflex coronary artery, draining into the coronary sinus was first diagnosed in a patient of 81. For 10 years, she had had dyspnea on exertion, mild mitral regurgitation and permanent ventricular pacing for symptomatic bradyarrhythmia. She was treated medically and despite her age was offered transcatheter balloon embolization, but she refused to undergo such intervention.     

Scleroderma renal crisis. 7 cases and review of the literature

We report a series of seven patients who had scleroderma renal crisis. Their primary clinical and laboratory features along with the details of their management were compared with those of similar cases from the literature. The seven patients died within one to four months of the diagnosis with a pattern of acute renal failure, left ventricular failure and malignant hypertension. Histopathologic examination was performed in four of the patients, in two of whom it revealed thickening of the wall of the interlobular arteries related to the scleroderma, and in the other two patients nonspecific lesions of malignant hypertension. This histopathologic particularity led us to propose, on the basis of multiple renal biopsies performed in patients with scleroderma, a lesion chronology of the kidney in patients with scleroderma. Nevertheless, the triggering factors and pathophysiologic mechanisms of scleroderma renal crisis remain unclear and its prognosis is severe. Early treatment with angiotensin-converting enzyme inhibitors and other vasodilatators administered intravenously can prevent death and dialysis.     

Treatment of amiodarone-induced hyperthyroidism by carbimazole with maintenance of amiodarone

The authors report the case of a patient treated with amiodarone for syncopal ventricular tachycardia complicating idiopathic dilated cardiomyopathy in whom symptomatic hyperthyroidism led to a discussion of the different therapeutic options available in this type of case. Neomercazole treatment was prescribed with success enabling maintenance of the antiarrhythmic drug. The reputation of inefficacy of carbimazole should be reconsidered ; high dose therapy should probably be tried in patients with hyperthyroidism when withdrawal of the antiarrhythmic drug does not seem to be possible.     

Hyperthyroidism due to contamination of the alternative drug Ader-Rein (Vascu-Vitaal)

A 52-year-old woman developed hyperthyroidism due to the alternative Vascu-Vitaal pills. She was suffering from nephrotic syndrome due to membranoproliferative glomerulonephritis and subclinical hypothyroidism, possibly due to renal loss of thyroid hormone. For peripheral vascular disease she took the non-registered Vascu-Vitaal pills on her own initiative. According to the product information, these pills contain multiple vitamins, minerals, amino acids and tissue extracts of bovine adrenals, hypophysis and thymus. The patient developed hyperthyroidism after starting on a new batch of the preparation; it subsided after stopping the treatment. A technetium thyroid scan showed decreased uptake and subsequently the Vascu-Vitaal pills were found to contain both thyroxine and triiodothyronine. The thyroid hormone contamination was probably caused by bovine thyroid tissue. It is suggested to require a health warning statement on the package of alternative therapeutics stating that production and contents are not subject to governmental supervision.     

(Neurological CPC.55). A 60-year-old woman with progressive cerebellar ataxia, myoclonus, and dementia

We report a 60-year-old woman with progressive ataxia, myoclonus, choreoathetosis, and dementia. She was well until 27 years of the age when she noted an onset of gait disturbance and speech disturbance. She noted abnormal involuntary movements in her four limbs at 42 years of the age. Her symptoms had progressively become worse and she fell down frequently by her 52 years of the age. In addition, her family members noted gradual decline in her intelligence. She was admitted to our hospital in February of 1993 when she was 57-year-old. On admission, she showed dementia, scanning speech, ataxic gait, limb ataxia, action myoclonus, and choreic movements which involved her four limbs. Deep tendon reflexes were slightly exaggerated in the lower limbs; no Babinski sign was noted. Sensation was intact. Laboratory findings were unremarkable. Cerebral MRI revealed atrophy of the cerebellar cortex, superior cerebellar peduncle, brain stem, and the cerebral cortex; the third ventricle and the lateral ventricles were dilated; furthermore, T2-high signal lesions were seen in the cerebral white matter and in the pontine base. Her clinical course was one of the progressive deterioration of her ataxia, involuntary movements, and dementia. She expired on April 24, 1996 when she was 60-year-old. She was discussed in a neurologic CPC and the chief discussant arrived at the conclusion that the patient had dentatorubral-pallidoluysian atrophy. A minor opinion was that she might have had myoclonus epilepsy with ragged-red fibers. Postmortem examination revealed atrophy, gliosis, and neuronal loss in the external segment of the globus pallidus, subthalamic nucleus, red nucleus, and in the dentate nucleus. In addition, the gracil and cuneiform nuclei showed neuronal loss and spheroid formation; the spinocerebellar tracts were retained. The substantia nigra and the locus coeruleus were intact. No ragged-red fibers were seen in the muscle biopsy specimen taken in February, 1993. The neuropathologic findings were consistent with the diagnosis of dentatorubral-pallidoluysian atrophy.     

Prevalence of autoantibodies associated with glomerulonephritis, unaffected after extracorporeal shock wave lithotripsy for renal calculi, in a three-year follow-up

A very rare case of full trisomy 18 associated with multiple hepatoblastomas is reported. The patient also had ventricular septal defect and patent ductus arteriosus, which were repaired at 6 months of age. After the cardiac surgery, she was noted to have an abdominal mass and an elevated serum alpha-fetoprotein level. A partial hepatic lobectomy was performed at 7 months of age, and the resected tumor was diagnosed as a fetal-type hepatoblastoma. At 2 years and 4 months of age, a chest radiography disclosed an elevated left diaphragm, and abdominal ultrasonography demonstrated a tumor in the left hepatic lobe. The resected tumor was also diagnosed as a fetal-type hepatoblastoma. Chromosomal analysis demonstrated that the karyotypes of peripheral blood and hepatic tumor cell obtained on two occasions were both 47,XX, +18. She has no evidence of recurrence at 3 years of age without specific therapy.     

Addisonian crisis provoked by levothyroxine substitution therapy

A women aged 36 with a positive family anamnesis for autoimmune endocrine diseases and a history of thyroid diseases, developed major complaints of general malaise, orthostatic hypotension and loss of appetite after the start of a treatment with levothyroxin because of (sub)clinical hypothyroidism. She was found to suffer from primary adrenocortical insufficiency masked by excessive use of liquorice and a lowered metabolism, but which via the suppletion with thyroid hormone had led to an addisonian crisis.     

The progression of an infundibulum to aneurysm formation and rupture: case report and literature review

OBJECTIVE AND IMPORTANCE: Infundibula (IFs) are funnel-shaped symmetrical enlargements that occur at the origins of cerebral arteries and are apparent on 7 to 25% of otherwise normal angiograms. They are frequently considered as normal anatomic variants of no pathogenic significance. CLINICAL PRESENTATION: We report the case of a ruptured posterior communicating artery aneurysm that had developed at the site of a previously known IF in a 49-year-old hypertensive woman. She had a poor conscious level at admission, with widespread subarachnoid hemorrhage and obstructive hydrocephalus. INTERVENTION: The patient was immediately ventilated, and an external ventricular drain was inserted. The aneurysm was successfully clipped; however, secondary hemorrhage occurred both before and during craniotomy. She developed marked hypernatremia and subsequently died. CONCLUSION: This is the 11th case of IF-to-aneurysm progression reported. It suggests that in certain cases, serial investigations may be indicated with IFs to detect aneurysm formation and preempt rupture.     

Elevated numbers of reticulated platelets in hyperthyroidism: direct evidence for an increase of thrombopoiesis

We studied thrombopoietic activity in hyperthyroidism by determination of reticulated platelet counts. At the time of hyperthyroidism 14/15 patients had higher reticulated platelets than after achievement of euthyroidism (P<0.001). There was no difference in peripheral platelet counts and mean platelet volumes at the time of hyperthyroidism when compared to euthyroidism. Three patients had pan- and auto-reactive platelet antibodies during hyperthyroidism. These antibodies were directed against GPIIb/IIIa in two patients and against GPIb/IX in one patient. Our findings provide direct evidence that hyperthyroidism is associated with increased platelet production, as reflected by an increase in reticulated platelets.     

Polyarthralgia disclosing hyperthyroidism. Two case reports

BACKGROUND: We report two cases of rheumatism associated with hyperthyroidism. In both cases, arthralgia totally regressed after thyroid treatment. CASE REPORTS: Two 79-year-old and 59-year-old women developed manifestations of polymyalgia rheumatica and psoriasis arthritis respectively. Corticosteroid therapy was ineffective and followed by manifestations of hyperthyroidism. The first patient was treated with carbimazole and the second with thyroidectomy. Once the hyperthyroidism was controlled, both patients experienced a dramatically rapid cure of their arthralgias. DISCUSSION: Scalpulo-humeral periarthritis is the main articular complication of hyperthyroidism. True manifestations of "thyrotoxicosis rheumatism" are unusual and may be linked with a direct toxicity of the thyroid hormones on joint cartilage or with an autoimmune manifestation of hyperthyroidism.     

The growth of secondary infections of Hymenolepis microstoma in mice: the effect of various primary infection regimes

Two patients, ages 7 and 17, with unresectable obstructions within the left ventricular cavity, have been managed by interposing a conduit bearing a porcine aortic valve between the apex of the left ventricle and the infra-renal abdominal aorta. The younger child had idiopathic hypertrophic subaortic stenosis (IHSS) recognized in infancy. At the age of three, a right ventricular myomectomy and a trans-aortic left ventricular myotomy were performed. Symptoms were progressive with congestive failure, diaphoresis, syncope , and angina pectoris. Following construction of a second left ventricular outflow tract with relief of intraventricular obstruction, the patient has become asymptomatic. The second patient has fibrous tunnel obstruction of the left ventricular outflow tracting providing a 100 mm Hg gradient. Fibrous tissue was resected in part through the transaortic route, and a second outflow tract was constructed. A postoperative cardiac catheterization revealed an obliteration of the previous intraventricular gradients and an equal distribution of left ventricular output through the two available outflow tracts. She remains asymptomatic.     

Trends of back pain in eastern Finland, 1972-1992, in relation to socioeconomic status and behavioral risk factors

We report a 74-year-old woman with parkinsonism and dementia, who died 4 years after the onset of the disease. She was well until 70 years of the age (1993) when she noted slowness in the movement in her left hand. She also developed gait disturbance and the similar symptoms spread to the right upper and lower extremities. Two years after the onset, she had difficulty in walk, and was admitted to our hospital on March 9, 1995. Her daughter had the onset of hand tremor at 50 years of the age and gait disturbance at 52. Her gait improved after levodopa treatment, but her MRI revealed a liner T2-high signal lesion along the outer surface of each putamen. On admission, the patient was alert but slighted demented. Higher cerebral functions were normal. She had a masked face and small voice. Her gait was of small step without arm swing. Retropulsion was present. Rigidity was noted in the neck but not in the limbs. She was bradykinetic but tremor was absent. She was treated with levodopa/carbidopa, dops, and bromocriptine with considerable improvement and was discharged on March 30, 1995. On January 19, 1996, she developed fever and hallucination; she became more akinetic and admitted again. She showed marked dementia and stage IV parkinsonism. She was treated by supportive measures with improvement in the general condition, but she was found to have a gastric cancer for which a subtotal gastrectomy was performed on March 11, 1996. Post-operative course was uneventful, but her parkinsonism progressed to stage V. She was transferred to another hospital on May 13, 1996. In July 21, 1996, she developed dyspnea and fever and was admitted to our hospital again. She was somnolent. Rigidity was moderate to marked and she was unable to stand or walk. By supportive cares, her general condition improved and was discharged to home on November 4, 1996. She developed fever on June 13, 1997 and admitted to our service again. Her BP was 150/90 mmHg. She was alert but markedly demented. Laboratory examination revealed increases in liver enzymes (GOT 75 IU/l, GPT 101 IU/l) and renal dysfunction (BUN 68 mg/dl, creatinine 3.27 mg/dl). Subsequent hospital course was complicated by renal failure and thrombocytopenia (33,000/ml). She expired on July 1, 1997. The patient was discussed in a neurologic CPC, and a chief discussant arrived at the conclusion that the patient had diffuse Lewy body disease and her daughter striatonigral degeneration. Some participants thought both the patient and her daughter had diffuse Lewy body disease. Post-mortem examination revealed marked degeneration of the substania nigra and the locus coeruleus. The medial part of the nigra also showed marked cell loss. Lewy bodies were found in the remaining nigral and coeruleus neurons. Cortical Lewy bodies were very few and the striatum was intact. Pathologic diagnosis was Parkinson's disease. Dementia was in part attributed to the marked degeneration of the medial part of the substantia nigra.     

Neonatal hyperthyroidism caused by TSH receptor antibodies in maternal autoimmune hyperthyroidism

Between July 1993 and December 1994 five term infants of mothers with Graves' disease were hospitalised at the Frühgeburtenstation of the Univ.-Frauenklinik Graz. Four Mothers had elevated TSH-receptor-antibody (TRAb)--levels during pregnancy, one had normalised TRAb-titers. In one case hyperthyroidism was first diagnosed during pregnancy. Three newborns had elevated TRAb-titers; in one of them thyrotoxicosis was diagnosed clinically and biochemically at the second day of life, one newborn had mild hyperthyroidism with tachycardia at the end of the first week of life and one newborn was asymptomatic by immediately initiated therapy. The two other newborns had normal thyroid hormone and antibody levels and no symptoms or signs of hyperthyroidism. The cases are reported and discussed in detail and our overall approach to diagnosis and treatment of neonatal hyperthyroidism in case of maternal Graves' disease will be given.     

Complete remission of refractory gestational trophoblastic disease with brain metastases treated with multicycle ifosfamide, carboplatin, and etoposide (ICE) and stem cell rescue

Patients with chemotherapy-refractory gestational trophoblastic disease and brain metastasis are considered to have a very poor prognosis. We present the case of a patient who had failed several chemotherapeutic regimens. Despite transient responses to chemotherapy, she had not achieved a complete remission in 3 years, and had developed systemic disease and recurrent brain metastasis. She was treated with four cycles of high-dose ifosfamide, carboplatin, and etoposide with blood progenitor cell support. She tolerated this regimen well and has obtained a complete remission that is ongoing for 12 months.