Lingual thyroid. Diagnosis and treatment.

We describe four patients who presented with a lingual thyroid condition (three females and one male, aged between 7 and 22 years). Only the male patient was symptomatic with mild dysphagia and hemoptysis. The diagnosis was suspected in three patients, and was confirmed by iodine 123 or 131 scanning in all patients and by a computed tomographic scan in the one patient studied. The patient with dysphagia received a 10-mCl therapeutic dose of iodine 131 before thyroxine replacement was started. The diagnosis and management of lingual thyroid is discussed. All patients need lifelong thyroxine suppression. Unenhanced computed tomographic scans have a diagnostic appearance due to the iodine content of the ectopic thyroid tissue.

Supplemental vibrational force during orthodontic alignment: a randomized trial.

This prospective 3-arm parallel-group randomized clinical trial investigated the effect of supplemental vibrational force on rate of orthodontic tooth alignment with fixed appliances. Eighty-one subjects (40 males, 41 females; mean age, 14.1 y) undergoing first premolar extraction-based fixed appliance treatment were randomly allocated to treatment supplemented with daily use (20 min) of a removable intraoral vibrational device (AcceleDent; OrthoAccel Technologies Inc.; n = 29), an identical nonfunctional (sham) device (n = 25), or fixed appliances only (n = 27). Mandibular study casts were taken at baseline (treatment start: placement of 0.014-in. nickel-titanium arch wire), initial alignment (0.018-in. nickel-titanium arch wire), and final alignment (0.019 x 0.025-in. stainless steel arch wire). Overall mean irregularity index in the mandibular arch at baseline was 8.5 ± 3.8 mm (95% CI, 7.6 to 9.3) with no significant difference between groups (P = 0.73). For the total sample, mean irregularity index at initial alignment was 2.7 ± 2.8 mm (95% CI, 2.2 to 3.4) with no significant difference between groups (P = 0.40). Mean time from baseline to initial alignment was 59 ± 25 d (95% CI, 54.5 to 65.6); from initial to final alignment, 150 ± 62.5 d (95% CI, 136 to 165); and baseline to final alignment, 209 ± 65 d (95% CI, 195 to 224). Kaplan-Meier analysis demonstrated that patterns of alignment were not significantly different among the 3 groups (P = 0.66). Multivariate linear regression for initial and overall alignment rates using initial irregularity index as the covariate showed no significant differences among groups. The most important influence on both initial and overall rates of alignment was initial irregularity (P = 0.1 × 10(-4)). This prospective randomized clinical trial found no evidence that supplemental vibrational force can significantly increase the rate of initial tooth movement or reduce the amount of time required to achieve final alignment when used in conjunction with a preadjusted edgewise fixed appliance (ClinicalTrials.gov NCT02314975).

Acute and long-term effects of octreotide in patients with ACTH-dependent Cushing's syndrome.

PURPOSE: Octreotide is of proven efficacy in the management of patients with acromegaly, thyrotropin-secreting pituitary adenomas, and certain gastrointestinal tumors, but its effect in Cushing's syndrome is less clear. PATIENTS AND METHODS: We studied 10 patients who presented with adrenocorticotropic hormone (ACTH)-dependent Cushing's syndrome, 3 of whom were previously adrenalectomized. Serum cortisol or ACTH levels were measured before and during the administration of octreotide 50 to 500 micrograms every 8 hours for 24 to 72 hours. RESULTS: Treatment was effective in four patients: serum cortisol levels decreased to within or below the normal range in Patients 1, 2, and 3, and ACTH levels were substantially lowered in Patient 4, who had previously been adrenalectomized for a metastatic islet cell tumor. These responses were sustained during long-term treatment for 2 to 72 weeks. All four patients showed no evidence of a pituitary tumor on computed tomographic or magnetic resonance imaging and had proven (Patients 3 and 4) or presumed ectopic disease. Of the six patients who did not respond, four had pituitary tumors and two had presumed ectopic ACTH production. CONCLUSION: We conclude that a short trial of octreotide is warranted in patients with ACTH-dependent Cushing's syndrome who have no demonstrable pituitary tumor. A response to treatment should alert the physician to the possibility of an ectopic ACTH source and will identify patients whose disease may be controllable using octreotide.

Comparison of 1073 MBq and 3700 MBq iodine-131 in postoperative ablation of residual thyroid tissue in patients with differentiated thyroid cancer.

In a randomized prospective study, we compared the efficacy of low dose (1073 MBq) and high dose (3700 MBq) iodine-131 administration in postoperative ablation of residual functioning thyroid tissue in 63 patients with differentiated thyroid cancer. We were unable to demonstrate any difference between the low- and the high-dose of radioactive iodine in scintigraphic ablation of remnant tissue. In 81% (21/26) of the patients, 1073 MBq ablated after the first dose, 77% (21/26 + 3/5 = 24/31) after the first plus second dose, and 69% (24/31 + 0/4 = 24/35) after the first, second, and third dose. Radioiodine (3700 MBq) ablated in 84%, 73%, and 69% of the patients after respectively 1., 1. plus 2., and 1. plus 2. plus 3. dose. Forty percent of the patients ablated with the low dose and 44% ablated with the high dose had elevated thyroglobulin levels at the time of complete scintigraphic ablation. In conclusion, we did not find any difference between 3700 MBq and 1073 MBq iodine-131 as regard to number of doses needed for complete scintigraphic ablation of residual functioning thyroid tissue.

Abnormal expression of the MOS proto-oncogene in human thyroid medullary carcinoma.

We have been studying the expression of a range of proto-oncogenes in human thyroid tumour tissue by using Northern blot analysis. We have demonstrated the expression of a MOS mRNA of 1 kb in all thyroid samples. Furthermore, in a medullary carcinoma sample we also observed additional mRNA species of 1.7 and 2.2 kb. Southern blot analysis of DNA prepared from the same tumour sample did not reveal a rearrangement of the gene. These findings are the first report of MOS expression in any human tissue, and indicate that MOS oncogene activation might be important in the development of some thyroid tumours.

Pattern of calcium metabolism in normo- and hypercalciuric patients with calcium urolithiasis in Saudi Arabia.

Urinary calcium excretion was measured in 100 consecutive normocalcemic patients with calcium urolithiasis and 12 Saudi Arabian controls while the patients and the controls were eating their usual diet of unknown calcium content. Only 16 patients were hypercalciuric using the definition of twenty-four-hour urinary calcium of more than 300 mg for males and more than 250 mg for females, and one of these patients was subsequently found to have hyperparathyroidism. The twenty-four-hour urine calcium was less than 200 mg in 60 per cent of male patients and in all the male controls. Since the dietary intake of calcium during the twenty-four-hour urinary collection was unknown, a 1,000-mg calcium loading test was performed in an attempt to differentiate various patterns of abnormal calcium excretion. Of the twenty-four-hour normocalciuric patients 10 (18%) had "absorptive hypercalciuria" and 16 patients (29%) demonstrated a "renal hypercalciuria" pattern. Thirty-nine patients (57%) and all 12 controls had normocalciuria before and after calcium loading.

Macroprolactinomas with suprasellar extension: effect of bromocriptine withdrawal during one or more pregnancies.

OBJECTIVE: To investigate the effects of bromocriptine withdrawal during one or more pregnancies in patients who presented with pituitary macroprolactinomas with suprasellar extension. DESIGN: Four infertile patients presenting with a macroprolactinoma with suprasellar extension conceived during treatment with bromocriptine on 10 occasions resulting in eight full-term normal deliveries. Treatment was withheld shortly after conception in each pregnancy. RESULTS: Serum prolactin (PRL) levels fell initially from a mean of 2,776 (range 1,682 to 4,515) to 27 micrograms/L (range 1 to 71) with the development of a partially empty sella in all patients. Recovery of visual field defects occurred in the only affected individual. In case 1, PRL levels remained within the normal range, after bromocriptine withdrawal in the first pregnancy, with the development of an empty sella. Prolactin levels, however, increased substantially in cases 2 to 4. An asymptomatic suprasellar tumor extension returned in cases 2 and 3. After two or more pregnancies (cases 1, 3, and 4), there was a progressive decline in the serum PRL levels. Although still elevated in cases 3 and 4, the PRL levels were considerably below those obtained at presentation or in the first pregnancy. Tumor regression with the development of an empty sella was observed in both these patients as well in their pregnancy or postpartum period. CONCLUSIONS: Bromocriptine may be safely withdrawn during pregnancy in patients presenting with a macroprolactinoma. With multiple bromocriptine induced pregnancies, PRL levels and tumor size may progressively decrease with the eventual development of an empty sella.

MR imaging of the lungs with hyperpolarized helium-3 gas transported by air.

Hyperpolarized noble gas MRI shows promise in the functional imaging of the pulmonary air spaces. The production of hyperpolarized (HP) gas requires specialized laser optical pumping apparatus, which is not likely to be home built in the majority of clinical MRI radiology centres. There are two routes through which HP gas will be made available to hospitals for clinical use: either the apparatus will be installed locally at a considerable expense to the centre, or a central facility will produce the gas and then deliver it to remote MRI sites as and when required. In this study, the feasibility of transporting large quantities of HP gas for in vivo MR imaging from a remote production facility in Mainz, Germany, by airfreight to Sheffield, UK, was successfully demonstrated.

Low field strength magnetic resonance imaging of the neonatal brain.

BACKGROUND: Magnetic resonance (MR) imaging of the neonate has been restricted by the need to transport the sick baby to the large magnetic resonance scanners and often the need for sedation or anaesthesia in order to obtain good quality images. Ultrasound is the reference standard for neonatal imaging. OBJECTIVE: To establish a dedicated neonatal MR system and compare the clinical usefulness of MR imaging with ultrasound imaging. DESIGN: Prospective double blind trial. SETTING: Neonatal intensive care unit, Sheffield. MAIN OUTCOME MEASURES: Imaging reports. PATIENTS: 134 premature and term babies. RESULTS: In 56% of infants with pathology suspected on clinical grounds, MR provided additional useful clinical information over and above that obtained with ultrasound. CONCLUSION: Infants can be safely imaged by dedicated low field magnetic resonance on the neonatal intensive care unit without the need for sedation at a cost equivalent to ultrasound.

Massive rise in thyroglobulin with adult respiratory distress syndrome after embolisation of thyroid cancer metastasis.

A patient with long-standing metastatic disease from a well differentiated papillary cell carcinoma of the thyroid underwent embolisation of a painful large sacral metastasis. Following embolisation she had a massive rise in circulating serum thyroglobulin (Tg) levels coinciding with the development of adult respiratory distress syndrome (ARDS) on the tenth day without any other obvious cause. The patient subsequently recovered. The time course suggests that ARDS in this case might have resulted from aggregation of Tg molecules in the pulmonary microcirculation.

Paget's disease. Radiological changes occurring in untreated patients and those on therapy with salmon calcitonin during two years' observation.

Radiological observations over two years were made in 23 patients with Paget's disease. Radiological criteria for progression and regression of disease are defined. Fourteen of the patients were treated with salmon calcitonin. Of these treated patients four showed regression of the disease and only two showed progression, both in the skull vault. Of the remaining nine untreated patients none showed regression but two showed progression. The radiological findings confirm that calcitonin will prevent the extension and sometimes induce healing of Paget's disease in long bones but that the skull appears to respond differently. Radiological changes were slow to occur and yearly or two yearly follow up is sufficient.

Subclinical pulmonary haemorrhage causing a restrictive lung defect in three siblings with a unique urticarial vasculitis syndrome.

Three siblings with urticarial vasculitis syndrome (UVS) are described. All had restrictive lung function abnormalities caused by subclinical pulmonary haemorrhage. The latter was suspected after finding haemosiderin-laden macrophages and a friable bronchial mucosa during elective bronchopulmonary alveolar lavage (BAL). The chest radiographs were normal at presentation but after steroid withdrawal symptoms worsened, haemoglobin levels fell, and Case 1 developed acute pulmonary haemorrhage. This was documented by lung biopsy, which also revealed evidence of old haemorrhage and fibrosis. We concluded that these patients had a unique familial variant of UVS with a previously unreported restrictive lung disease due to subclinical pulmonary haemorrhage.

Case report: idiopathic giant cell granuloma of the hypophysis: an unusual cause of panhypopituitarism.

A 37-year-old man presented with headache and reduced libido. He suffered from panhypopituitarism, and the walls of the pituitary fossa were noted to be thickened, sclerotic, and irregular. Computed tomography scans showed an intrasellar and suprasellar, patchy enhancing tumor. From a biopsy taken at transsphenoidal surgery, a giant cell granuloma was diagnosed, for which there was no obvious cause. Inflammatory disease within the pituitary fossa should be suspected if a reaction is seen in the surrounding bone.

Role of testicular fine-needle aspiration biopsy in the evaluation of male infertility: cytologic and histologic correlation.

A series of testicular fine-needle aspiration biopsy specimens from 272 infertile men with azoospermia were reviewed and categorized according to morphologic patterns. These included active spermatogenesis, 14 (5%); hypospermatogenesis, 106 (39%); Sertoli cells only, 70 (26%); atrophic pattern, 52 (19%); and maturation arrest, 1 (0.36%). In 29 cases (11%) the amount of material was insufficient for evaluation. The histologic and cytologic findings in 52 cases showing spermatogenesis correlated very well in 52 cases for which open testicular biopsy specimens were also available. These findings indicate that fine-needle aspiration biopsy of the testis is a reliable and useful technique for the investigation of patients with azospermia.

Hypernatremic metabolic myopathy due to hypothalamic dysfunction.

We report a patient with muscle weakness secondary to elevated serum sodium level. The cause of the elevated sodium level and the mechanism involved in producing muscle weakness are discussed.

Ultrasound guided wire localisation to aid surgical biopsy of impalpable parotid oncocytoma.

Ultrasound guided wire localisation (UGWL) is an effective way of helping a surgeon find an impalpable lesion but its use in the head and neck has only recently been described. The authors present an unusual case of bilateral multi-focal parotid oncocytoma, in which UGWL was combined with extracapsular dissection to obtain an impalpable lesion for histological examination following initial equivocal core biopsy.

A rare cause of a parotid mass: spontaneous pseudoaneurysm of the superficial temporal artery.

The authors describe a rare presentation of a spontaneous pseudoaneurysm of the superficial temporal artery which mimicked a parotid neoplasm. The clinical presentation, possible aetiology, diagnosis, and management of this condition are discussed.