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It has already been noted that hemangioblastoma is occasionally complicated with various diseases, especially retinal angioma, cysts of kidney and/or pancreas, vascular disorders, and furthermore about 10% of hemangioblastoma are multiple. The authors report here a case of multiple cerebellar hemangioblastomas accompanied with congenital deafness, juvenile diabetes mellitus and retinal angioma. The patient, a 38-year-old man who complained of disturbance of consciousness, headache and vomiting, admitted to our hospital on October 6, 1982. He was born in consanguineous marriage family and his elder sister was also suffered from congenital deafness and juvenile diabetes mellitus, but no angiomas. CT and angiography showed a left side cystic and a right side small cerebellar lesions with remarkable ventricular enlargement. Ventriculo-peritoneal shunt and suboccipital craniectomy & removal of the tumors were performed on October 6, and November 8, 1982 succeedingly. From a point of view of hereditary occurrence, the authors investigated these complicated lesions recognized in this patient. It revealed that this case did not belong to any other categories of hereditary syndromes which were already reported and so authors considered that this was an extremely rare condition. Moreover, in case of multiple hemangiomas, cerebral angiography could be a more useful method than CT scan to detect multiplicity of the lesions in the posterior fossa.
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PMID:[A case of multiple cerebellar hemangioblastomas with congenital deafness, juvenile diabetes mellitus and retinal angioma]. 370 51

Magnesium ions (Mg2+) are pivotal in the transfer, storage and utilization of energy; Mg2+ regulates and catalyzes some 300-odd enzyme systems in mammals. The intracellular level of free Mg2+ ([Mg2+]i) regulates intermediary metabolism, DNA and RNA synthesis and structure, cell growth, reproduction, and membrane structure. Mg2+ has numerous physiological roles among which are control of neuronal activity, cardiac excitability, neuromuscular transmission, muscular contraction, vasomotor tone, blood pressure and peripheral blood flow. Mg2+ modulates and controls cell Ca2+ entry and Ca2+ release from sarcoplasmic and endoplasmic reticular membranes. Since the turn of this century, there has been a steady and progressive decline of dietary Mg intake to where much of the Western World population is ingesting less than an optimum RDA. Geographic regions low in soil and water Mg demonstrate increased cardiovascular morbidity and mortality. Dietary deficiency of Mg2+ results in loss of cellular K+ and gain of cellular Na+ and calcium ions (Ca2+). Blood normally contains Mg2+ bound to proteins, Mg2+ complexed to small anion ligands and free ionized Mg2+ (IMg2+). Most clinical laboratories only now assess the total Mg, which consists of all three Mg fractions. Estimation of the IMg2+ level in serum or plasma by analysis of ultrafiltrates (complexed Mg + IMg2+) is somewhat unsatisfactory, as the methods employed do not distinguish the truly ionized form from Mg2+ bound to organic and inorganic anions. Because the levels of these ligands can vary significantly in numerous pathological states, it is desirable to directly measure the levels of IMg2+ in complex matrices such as whole blood, plasma and serum. Using novel ion selective electrodes (ISE's), we have found that there is virtually no difference in IMg2+, irrespective of whether one samples whole blood, plasma or serum. These data demonstrate that the mean concentration of IMg2+ in blood is about 600 mumoles/litre (0.54-0.65 mmol/L, 95% Cl); 65-72% of total Mg being free or biologically-active Mg2+. Use of the NOVA and KONE ISE's for IMg2+ on plasma and sera from patients with a variety of pathophysiologic and disease syndromes (e.g., long-term renal transplants, liver transplants, during and before cardiac surgery, ischemic heart disease [IHD], headaches, pregnancy, neonatal period, non-insulin dependent diabetes (NIDDM), end-stage renal disease [ESRD], hemodialyse [HEM], and continuous ambulatory peritoneal dialysis (CAPD), hypertension, myocardial infarction [AMI] and after excessive dietary intake of Mg), has revealed interesting data. The results indicate that long-term renal transplant patients, headache, pregnant, NIDDM, ESRD, HEM, CAPD, AMI, hypertensive, and IHD subjects exhibit, on the average significant depression in IMg2+ but not TMg. Use of 31P-NMR spectroscopy on red blood cells, from several of these disease states, to assess free intracellular Mg ([Mg2+]i demonstrates a high correlation (r = 0.5-0.8) between IMg2+ and [Mg2+]i. Increased dietary load of Mg, for only 6 days, in human volunteers, resulted in significant elevations in serum IMg2+ but not TMg. Correlations between the clinical course of several of the above disease syndromes and the fall in IMg2+ and [Mg2+]i were found. The ICa2+/IMg2+ ratio appears, from our data, to be an important guide for signs of peripheral vasoconstriction, ischemia or spasm and possibly atherogenesis. Overall, our data point to important uses for ISE's for IMg2+ in the diagnosis and treatment of disease states.
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PMID:Role of magnesium in patho-physiological processes and the clinical utility of magnesium ion selective electrodes. 886 38

The aim of the study was to assess the frequency and nature of headaches in patients with non-insulin dependent diabetes (NIDDM). The investigations included 154 patients with NIDDM, 90 females and 64 males aged between 30 to 65 years. Duration of diabetes varied from 6 months to 37 years. The control group comprised 106 persons. Of the 127 NIDDM patients complaining of headaches, 95 had migraine headaches and 32 tension-type headaches. In 50 patients the onset of migraine headaches occurred when the patients were afflicted with diabetes. In 45 patients migraine was diagnosed before they began to suffer from diabetes. In these patients the onset of diabetes significantly increased the average yearly number of headache days. In 21 patients tension-type headaches occurred in the course of diabetes. 11 patients had tension-type headaches before the onset of the disease. In these cases the onset of diabetes increased significantly the average yearly number of headache days. In the control group, migraine was diagnosed in 17 subjects and tension-type headaches in 28 subjects.
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PMID:Headaches in non insulin-dependent diabetes mellitus. 950 95

A 26-yr-old woman with type 1 diabetes and severe symptomatic autonomic neuropathy was treated with the long-acting somatostatin analogue Sandostatin LAR for intractable diarrhea. Her diarrhea had previously been successfully managed with three daily injections of octreotide without adverse consequences. She was given a single dose of Sandostatin LAR and within 2 weeks reported the development of increasingly frequent and severe headaches. Three weeks after the injection, she was admitted to hospital with severe hypertension, which eventually resolved with the administration of antihypertensive agents. No other underlying cause of the hypertension was discovered. Rechallenge of the patient with octreotide resulted in a transient hypertensive episode, which lasted 3 h. Severe hypertension, therefore, seems to be a possible adverse effect of treatment of diabetic diarrhea with somatostatin analogues, which should be used with great caution in subjects with severe autonomic dysfunction.
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PMID:Severe hypertension induced by the long-acting somatostatin analogue sandostatin LAR in a patient with diabetic autonomic neuropathy. 1072 19

Insulin-like growth factor-I (IGF-I) enhances insulin action in normal subjects and in patients with both type 1 and 2 diabetes; however, its administration is associated with significant side effects in a high percentage of patients. The coadministration of IGF binding protein-3 (IGFBP-3, the predominant IGF binding protein in serum) with IGF-I limits IGF-I inducible side effects, but it does not attenuate the ability of IGF-I to enhance protein synthesis and bone accretion; therefore, we determined whether IGF-I/IGFBP-3 would retain biological activity in type 1 DM and limit side effects associated with free IGF-I administration. Twelve patients received recombinant human IGF-I plus IGFBP-3 (2 mg/kg-day) by continuous sc infusion for 2 weeks. Each subject served as his own control; and, during a paired 2-week period, each received a placebo infusion. The order of the treatments was randomized. Subjects were placed on a constant caloric intake but were allowed to adjust insulin doses to maintain appropriate levels of glycemic control. Subjects measured blood glucose four times per day at home and kept a log of their insulin use. Frequent sampling for glucose, insulin, and GH was conducted during four inpatient study periods, one at the beginning and one at the end of each 2-week study interval. During IGF-I/IGFBP-3, insulin doses were reduced by 49%, and mean serum glucose was reduced by 23%. Free insulin levels obtained during frequent sampling in hospital fell 47% on IGF-I/IGFBP-3, compared with control, but showed no change with placebo. Concomitant glucose measurements did not differ in the two treatment groups. There was no change in body weight. Fructosamine levels decreased by 12%, but this was not significant (P < 0.1). Fasting triglyceride was unchanged, but cholesterol declined from 170 +/- 24 to 149 +/- 31 mg/dL (P < 0.05). IGFBP-2 (an IGF-I-dependent responsive variable) rose from 141 +/- 56 to 251 +/- 98 ng/mL (P < 0.01) on IGF-I/IGFBP-3. To analyze the mechanism by which IGF-I/IGFBP-3 might reduce insulin requirements, the change in serum GH was quantified. Mean GH levels were reduced by 72%, from 2.48 to 0.55 ng/mL (P < 0.001). An equal number (40%) of drug- and placebo-treated subjects had minor hypoglycemic episodes at home that required adjustment of insulin doses. No episode was classified as severe. In contrast to previous studies with free IGF-I, there were no cases of edema, headache, jaw pain, retinal edema, or Bell's palsy. No subject withdrew because of drug complications. These findings indicate that IGF-I/IGFBP-3 is biologically active on carbohydrate metabolism, as measured by a decrease in insulin requirements in patients with type 1 diabetes. Further studies will be required to determine the long-term safety and efficacy of this combination in patients with insulin resistance and diabetes.
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PMID:The combination of insulin-like growth factor I and insulin-like growth factor-binding protein-3 reduces insulin requirements in insulin-dependent type 1 diabetes: evidence for in vivo biological activity. 1077 Jan 91

A 29-year-old man developed acute visual impairment, cough, and headache. Both eyes showed serous retinal detachment in the posterior fundus. Fluorescein angiography showed subretinal pooling of fluorescein in the late phase. A diagnosis of Vogt-Koyanagi-Harada (VKH) syndrome was made based on clinical features. Treatment with systemic corticosteroids resulted in improvement of uveitis and both eyes showed "sunset glow" fundus 11 months later. Insulin-dependent diabetes mellitus (IDDM) developed 13 months later (3 months after systemic corticosteroid therapy). Despite treatment with insulin, glycemic control was poor. Human leukocyte antigen (HLA) typing showed HLADR9 and DQB 1*0303 related to IDDM. We postulated that treatment with corticosteroids precipitated IDDM, a yet unknown common autoimmune mechanism might have caused IDDM and VKH, or both conditions occurred coincidentally.
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PMID:A case of insulin dependent diabetes mellitus following systemic treatment for Vogt-Koyanagi-Harada syndrome. 1109 27

To determine the prevalence of fibromyalgia in diabetes mellitus and obesity, 121 consecutive patients have been observed: 27 with obesity (6 males and 21 females; mean age 57 years, range 20-57; mean body mass index [BMI] 34); 88 with type 2 diabetes mellitus (T2DM; 40 males and 48 females; mean age 63 years, range 44-78; mean BMI 28.8; mean glycated haemoglobin [HbA1c] in the last year 8.3%); 6 with type 1 diabetes mellitus (T1DM; 2 males and 4 females; mean age 52 years, range 26-76; mean BMI 24.5; mean HbA1c < 7%). An original questionnaire has been proposed (answer yes/not) as follows: 1) chronic (more than 3 months) and diffuse musculoskeletal pain; 2) sleep disturbances; 3) generalized fatigue; 4) paresthesias at the extremities; 5) swollen impression at hands and feet; 6) symptoms referred to irritable bowel syndrome; 7) headache; 8) symptoms change related with environmental climatic variations and/or exercise. A chronic and diffuse musculoskeletal pain has been reported by 62% of patients as well as in 9% of patients 11/18 positive tender points have been documented. In the patients with a BMI less that 26 the diagnosis of fibromyalgia was negative. Our data seem to reveal the presence of a significant clinical association between obesity, diabetes mellitus and fibromyalgia.
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PMID:[Prevalence of fibromyalgia in diabetes mellitus and obesity]. 1267 86

We report beneficial and adverse effects of sodium dichloroacetate (DCA) in three adult Japanese patients with mitochondrial disease: a 21-year-old male with involuntary movements, optic atrophy, hearing loss, and convulsions (patient 1), a 28-year-old man with mental deterioration, hemianopia, hearing disturbance, and convulsions (patient 2), and a 50-year-old woman with hearing disturbance, generalized muscle atrophy, and insulin dependent diabetes mellitus (patient 3). A3243G mutation was found in patient 2 and patient 3. Oral administration of DCA improved consciousness level and gait disturbances in patient 1, and ameliorated headaches, easy fatiguability, and muscle cramps in patient 2 and patient 3. DCA normalized high levels of lactate and pyruvate in blood and cerebrospinal fluids in all three patients. In patient 3, daily insulin needs decreased from 38 to 24 units, and urine C peptide increased from an undetectable level to 16 micrograms/day. In patient 1, DCA 23 mg/kg/day had been beneficial without adverse effects and he became free of convulsions for more than 32 months. However, despite of normal lactate and pyruvate, unsteady gait and lethargy developed after 50 mg/kg/day treatment for two months and one month in patient 2 and patient 3, respectively. In both patients, deep tendon reflexes disappeared and Romberg sign became positive. Nerve conduction studies confirmed sensory-dominant polyneuropathy and electroencephalogram showed diffuse slow basic activities. Cessation of DCA resulted in recovery of gait and consciousness, but sensory nerve action potentials did not recover in one month. Long term treatment of 50 mg/kg/day DCA may affect adversely the peripheral and central nervous systems in adult patients. Although effective plasma DCA concentration was previously reported as 25-160 micrograms/ml in patients under 18 years old, plasma DCA concentration of 10.2 micrograms/ml was sufficient in patient 1. We recommend lower dose of DCA in adult patients than in child patients.
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PMID:[Dichloroacetate treatment for adult patients with mitochondrial disease]. 1289 50

Unlike the case in adult-onset diabetes, neurologic complications in children with type 1 diabetes are uncommon. We report a case of a 14-year-old boy with type 1 diabetes who presented at our emergency unit with acute hemiparesis and intense headache without hypoglycaemia. Two hours after onset, the symptoms had subsided. It is the intention of the authors to increase the awareness of this association, whose prognosis is almost invariably good.
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PMID:Acute hemiparesis in a boy with type 1 diabetes. 1512 Jun 88

Celiac disease develops from an autoimmune response to specific dietary grains that contain gluten. Diagnosis can be made based on the classical presentation of diarrhea, fatty stools, and abdominal bloating and cramping, as well as the presence of specific serum antibodies. In addition, gluten ingestion has increasingly been found to be associated with other conditions not usually correlated with gluten intolerance. The subsequent diversity of the clinical presentation in these cases can complicate decision-making and delay treatment initiation in conditions such as ataxia, headaches, arthritis, neuropathy, type 1 diabetes mellitus, and others. This review explores the etiology and pathology of celiac disease, presents support for the relationship between gluten and other diseases, and provides effective screening and treatment protocols.
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PMID:Celiac disease and gluten-associated diseases. 1616 73


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