Cases reported "Choristoma"

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1/9. Transposition of the lingual thyroid: A new alternative technique.

    BACKGROUND: In symptomatic lingual thyroid, surgical transposition of the gland with its vascular supply intact seems to have superior results to those obtained by surgical ablation and autotransplantation. However, the procedure should be simple, reproducible, reliable, and cause less morbidity as well as providing simple access to and evaluation of the gland postoperatively. methods: We present the case of a 33-year-old female with lingual thyroid who was treated by transposing the whole gland to the lateral pharyngeal wall through a lateral pharyngotomy incision. The transposed lingual thyroid was nourished by a random tongue muscle pedicle flap. RESULT: At the 5-month postoperative stage, iodine scanning reviewed the radioactivity uptake of the transposed gland. Even though the patient was not on postoperative thyroid hormone supplement, her thyroid function gradually returned to normal after initially showing hypothyroid postoperatively. CONCLUSION: This new technique for transposition of lingual thyroid is simple and reliable and should be considered as an alternative method in the management of symptomatic patients.
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2/9. Heterotopy of thyroid tissue--a modified therapeutical approach.

    OBJECTIVES: The authors describe the classification of heterotopic thyroid tissue. A survey of more than 600 literature cases is provided. Different therapy approaches for these anomalies are discussed. Data from seven subsequent cases have been added to the existing literature data. The authors present an alternative approach to the therapy of heterotopic thyroid tissue. MATERIALS AND methods: The records of seven patients treated for a heterotopy of the thyroid tissue as the Department of ENT and head and neck Surgery of First Medical school of the Charles University of Prague since 1.1.1991 to 1.1.2001 have been analyzed. RESULTS: The first group: No surgery. This approach was used for children. In these patients the heterotopic thyroid tissue is the only thyroid tissue they have, but its function is not damaged and there are no mechanical symptoms (no airway obstruction and dysphagia). TSH substitution-suppression therapy is necessary for this group. Cooperation and follow up by the endocrinology, otolaryngology and pediatric departments is necessary. At a later age these patients can be treated as in the second group. The second group: Surgery. All patients in this group had heterotopic thyroid tissue. The total removal of thyroid tissue has been preferred. Carcinomatous change or the development of mechanical syndrome, dysphagia or airway obstruction is possible. Total removal is the best surgical approach. Various surgical methods are described in the literature (CO2 laser, intraoral, mandibulotomy, middle hyotomy etc.). These patients are treated as after total thyroidectomy and substitution therapy with synthetic thyroid hormones is necessary. The authors describe and add seven cases to the literature data. The authors describe radical removal of aberrant and accessory tissue [corrected].
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3/9. Heterotopic pancreas in the ampulla of vater.

    We report a patient in whom heterotopic pancreatic tissue was found within the ampulla and treated by ampullectomy. Only 16 cases of pancreatic tissue located at the ampulla of vater have been reported in the literature and pancreaticoduodenectomy has been performed in more than half the cases because malignancy was suspected preoperatively, reflecting a rather aggressive surgical attitude. An accurate preoperative evaluation thus appears decisive to avoid unnecessary radical surgery. Endoscopic treatment of ampullary tumors is emerging as a viable alternative to surgical treatment. Local surgical excision is a reasonable option to endoscopic treatment. The possibility of heterotopic pancreatic tissue, though rare, should be included in the differential diagnosis of papillary tumors. Histologic confirmation (frozen or delayed) is mandatory before attempting such major surgery.
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4/9. Adrenocorticotropin hypersecretion and pituitary microadenoma following bilateral adrenalectomy in a patient with classic 21-hydroxylase deficiency.

    Bilateral adrenalectomy is an acceptable alternative treatment in salt-wasting 21-hydroxylase deficiency when conventional steroid replacement therapy fails to control hyperandrogenism. Objections to surgical adrenalectomy have been based on surgical risk, possible loss of protective adrenal function, and the risk of ACTH-induced activation of adrenal rest tissue. We report a young female with salt-wasting CAH, who underwent bilateral adrenalectomy and developed severe hyperpigmentation, progressively marked corticotropin hypersecretion to concentrations seen in Nelson's syndrome (5,000-7,000 pg/ml), a pituitary microadenoma 5 years postoperatively, and probable ectopic adrenal rest tissue. Corticotropin concentrations failed to respond to ovine corticotropin-releasing hormone (oCRH) (1 microg/kg given as an i.v. bolus), but did suppress following both hydrocortisone administration (100 mg given as an i.v. bolus) and a low dose (0.5 mg given orally every 6 h for 48 h) dexamethasone suppression test. patients with CAH have hyperactivity of the hypothalamic-pituitary-adrenal axis and are at risk for pituitary tumor formation.
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5/9. multiple endocrine neoplasia type IIB identified by magnetic resonance imaging.

    Ectopic, epinephrine-secreting pheochromocytomas are rare and difficult to diagnose. In our case, MRI was able to show the lesion, which could not be clearly identified by CT. The histologic specificity provided by T2-weighted images confirmed the biochemical diagnosis of pheochromocytoma. MRI should be considered as an alternative to CT in the diagnosis of familial pheochromocytomas.
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6/9. Intrasellar neural-adenohypophyseal choristoma. A morphological and immunocytochemical study.

    A patient with mild acromegaly had recurrence of symptoms and signs of a chiasmal-area lesion seventeen years after radiation therapy for a presumed pituitary adenoma. A mass was found anterior to the pituitary gland. Abnormal tissue removed from the sphenoid sinus and sella turcica consisted of a predominantly ganglion-cell lesion. A few ganglion cells were immunoreactive for somatostatin. There were some small cysts lined by cells with immunostaining for glial fibrillary acidic protein, growth hormone or prolactin. Some cells with vacuoles and eosinophilic granules showed immunostaining for growth hormone, prolactin, ACTH, and beta-endorphin and, thus, appeared to be of adenohypophyseal origin. Cases of intrasellar ganglion-cell lesions have been reported, most of them associated with pituitary adenomas and acromegaly. The findings in this case are discussed in relation to the hypothesis that displaced, hypothalamic-type ganglion cells may produce a growth hormone-releasing factor that stimulates the development of a growth hormone-secreting pituitary adenoma. An alternative hypothesis is suggested that includes this concept, but also allows for the influence of non-neuronal cells on neuronal differentiation and for the possible influence of adenohypophyseal hormones on the replication of hypothalamic-type neurons in the lesion.
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7/9. Phakomatous choristoma of the lower eyelid with psammoma body formation: a light and electron microscopic study.

    A firm, round tissue mass, measuring approximately 12 mm in diameter, was excised from a 5-month-old boy who had a history of a gradually enlarging swelling in the left lower nasal eyelid since birth. Histologically and ultrastructurally, the lesion appeared benign and was composed of a dense connective tissue stroma in which nests, lobules, tubules, cords, and sheets of large, pale-staining cuboidal and elongated epithelial cells were surrounded by an irregular, thick PAS-positive basement membrane having focal excrescences. The interior of the epithelial islands contained "bladder" cells, cellular debris, granuloamorphous material, and foci of dystrophic calcification; an unusual feature of the lesion was the formation of numerous psammoma bodies. Tumors of lenticular anlage are rare and probably result due to displacement of migration of the inferior lens placode cells; alternatively, it may originate from an additional locus of lens vesicle in the primitive surface ectoderm of the lower lid. The lesion enlarges because of the differentiation of the ectopic lenticular cells in an abortive attempt to form a lens within the extraocular and mesodermal environment of the lid.
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8/9. Myoepithelial hamartoma of the duodenal wall.

    A rare case of myoepithelial hamartoma of the duodenal wall is presented, and previous case reports found in the literature are reviewed. Myoepithelial hamartomas are thought to arise from displaced pancreatic anlage present along the gastrointestinal tract during embryogenesis, which can differentiate into various pancreatic elements; the most highly differentiated form is heterotopic pancreas. An alternative theory is pancreatic metaplasia of endodermal tissues. We describe a 41-year-old man who presented with abdominal pain and vomiting. CT scanning revealed a mass at the head of the pancreas. A pancreaticoduodenectomy was performed for presumed cystadenoma. histology of the mass revealed a disorderly arrangement of smooth muscle, dilated and nondilated ducts, pancreatic acinar tissue and mucus glands. The relationship of myoepithelial hamartomas involving the small bowel to similar lesions in the stomach, bile ducts and gallbladder is discussed.
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9/9. Gastric cyst of the oral cavity.

    A case of a heterotopic gastric cyst of the oral cavity is described and a review of the literature, with emphasis on the possible etiological theories of this rare lesion, is presented. This developmental lesion is found more commonly in males. It most likely arises from misplaced embryonal tissue. Surgical treatment is the preferred choice of treatment, and CO2 laser is a safe alternative surgical approach for the removal of this lesion.
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