Cases reported "Myxedema"

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1/14. Elephantiasic pretibial myxedema: insight into and a hypothesis regarding the pathogenesis of the extrathyroidal manifestations of Graves' disease.

    The basis for the extrathyroidal manifestations of Graves' ophthalmopathy (GO) and dermopathy are not well understood. We describe immunohistochemical studies on the skin of a patient with an extreme, elephantiasic form of Graves' dermopathy that developed after periods of prolonged standing with dependent edema. Excision of part of the lesion with subsequent skin grafting from a normal donor site resulted in recurrence of the disease at the original site as well as in development of disease at the donor site. A murine monoclonal antibody reacted with the thyrotropin receptor (TSHR) or a cross-reacting protein in fibroblast-like cells in the patient's upper dermis and, surprisingly, with dermal cells from unaffected individuals. The patient's dermis containing lymphoid follicles comprising B cells and CD3 , CD4 T cells, with few CD8 T cells. CD21 cells (most likely follicular dendritic cells) were also present in the dermis. Based on past and present observations, we raise an unifying hypothesis to explain the diverse extrathyroidal manifestations of Graves' disease and their apparent lack of association with TSHR autoantibodies. As opposed to the present concept that these phenomena relate to site-specific properties on preadipocytes or fibroblasts, we suggest that clinically evidence GO and dermopathy are primarily caused by local factors (particularly in the orbit) superimposed on a systemic, low-grade connective tissue inflammation.
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ranking = 1
keywords = ophthalmopathy
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2/14. myxedema coma of both primary and secondary origin, with non-classic presentation and extremely elevated creatine kinase.

    myxedema coma is a rare, often fatal endocrine emergency that concerns elderly patients with long-standing primary hypothyroidism; myxedema coma of central origin is exceedingly rare. Here, we report a 37-year-old woman in whom classical symptoms of hypothyroidism had been absent. Six years earlier, she had severe obstetric hemorrhage and, shortly after, two subsequent episodes of pericardial effusion. On the day of admission, pericardiocentesis was performed for the third episode of pericardial effusion. Because of the subsequent grave arrhythmias and unconsciousness, she was transferred to our ICU. Prior to the endocrine consultation, a silent myocardial infarction had been suspected, based on the extremely high serum levels of creatine kinase (CK) and isoenzyme CK-MB. However, based on thyroid sonography, pituitary computed tomography, elevated titers of antithyroid antibodies and pituitary stimulation tests, the final diagnosis was myxedema coma of dual origin: an atrophic variant of Hashimoto's thyroiditis and post-necrotic pituitary atrophy (Sheehan syndrome). Substitutive therapy caused a prompt clinical amelioration and normalization of CK levels. Our patient is the first case of myxedema coma of double etiology, and illustrates how its presentation deviates markedly from the one endocrinologists and physicians at ICU are prepared to encounter. In addition, cardiac problems as those of our patient should not discourage from substitutive treatment (using L-thyroxine and the gastrointestinal route of absorption), if the age is relatively low.
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ranking = 0.0073625833619453
keywords = grave
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3/14. Evidence for thyrotropin receptor immunoreactivity in pretibial connective tissue from patients with thyroid-associated dermopathy.

    Pretibial myxedema (PTM), mainly characterized by the accumulation of glycosaminoglycans in the dermis and subcutaneous tissue, is an extrathyroidal manifestation of autoimmune Graves' disease (GD), almost always associated with Graves' ophthalmopathy (GO). The thyrotropin receptor (TSH-R) has been proposed as the common target antigen in GD, GO and PTM, with evidence for receptor transcripts and/or protein in these locations. The aim of this study has been to investigate whether receptor protein is present in the pretibial tissues. skin biopsies were obtained from two patients with PTM and two normal subjects without thyroid disease. A portion of each sample was fixed to produce semi-thin sections for Toluidine Blue or periodic acid Schiff (PAS) staining. The remainder was snap frozen to generate cryostat sections for immunohistochemical analysis using three monoclonal antibodies against TSH-R. In the skin from the two patients suffering from PTM, the dermis was infiltrated by inflammatory cells (lymphocytes, B cells, macrophages, mast cells) and adipocytes. The collagen fibers were dissociated by edema and by the accumulation of a PAS-positive material. Immunodetection of TSH-R produced positive staining on cells localized in the dermis, beneath the epidermis or close to the hypodermis. These cells were elongated and resembled fibroblasts. No immunoreactivity was observed in the dermis from control patients without thyroid disease. In conclusion, we have evidence for TSH-R immunoreactivity in the pretibium of patients with GD, GO and PTM. Further studies are needed to unambiguously identify the positive cells and determine whether the reactivity is due to the receptor itself or to a cross-reacting protein.
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ranking = 2.6403390723074
keywords = ophthalmopathy, thyroid-associated
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4/14. Pretibial myxedema as the initial manifestation of Graves' disease.

    Pretibial myxedema (PM) is a localized thickening of the pretibial skin due to accumulation of acid mucopolysacharides (glycosaminoglycans). Its pathogenesis is still under investigation. Pretibial myxedema, exophthalmus and thyroid acropachy are the dassic extrathyroidal manifestations of Graves' disease. Almost invariably, PM follows the onset of ophthalmopathy, developing after the diagnosis and treatment of hyperthyroidism. Pretibial myxedema preceding Graves' ophthalmopathy is rare. We report the case of a 28-year-old Greek woman, who presented with multiple, asymptomatic nodules and plaques of the lower legs in the absence of other physical findings. Histopathologic examination revealed deposition of mucopolysacharides in the lower dermis. Laboratory investigation showed elevated serum T3 and T4 and depressed TSH levels. In our patient, pretibial myxedema was the earliest manifestation, leading to the diagnosis of Graves' disease.
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ranking = 2
keywords = ophthalmopathy
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5/14. Triad of exophthalmos, pretibial myxedema, and acropachy in a patient with Graves' disease.

    Graves' disease is an autoimmune disorder of the thyroid gland with characteristic peripheral manifestations. The most common clinical findings include ophthalmopathy in 30% of patients, dermopathy (pretibial myxedema) in 4% of patients, and thyroid acropachy in 1% of patients. The triad of exophthalmos, pretibial myxedema, and acropachy occurs in less than 1% of patients. We present a case of Graves' disease with the clinical triad of eye disease, dermopathy, and acropachy.
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ranking = 1
keywords = ophthalmopathy
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6/14. Lack of effect of long-term octreotide therapy in severe thyroid-associated dermopathy.

    Severe thyroid associated dermopathy (TAD), a rare complication of Graves' disease, currently lacks effective treatment. Mediators of growth and inflammation, including insulin-like growth factor-1 (IGF-1) and somatostatin (SST) have been implicated in its pathogenesis. octreotide, a potent SST analogue, has been used in TAD with anecdotal success. Therefore, we evaluated the efficacy of long-term octreotide therapy in moderate to severe TAD. Three obese women with TAD were studied. Baseline treatment included levothyroxine, methimazole, fluocinonide ointment under occlusive dressing, and physiotherapy for lymphedema. After establishing baseline clinical status, octreotide was started (300 microg subcutaneously daily) for 10 to 28 months. Studies obtained at baseline and every 3 months included: leg circumference, skin examination, clinical photography, self-reported clinical status, body mass index (BMI), serum thyrotropin (TSH) and free thyroxine, titers of antithyroidal and anti-TSH receptor antibodies. Minimal changes in the edema, erythema, skin texture and size of nodules were observed. The minor changes seen followed significant decreases in the patients' BMI, and hence, cannot be specifically attributed to octreotide administration. No clinically significant benefit from long-term octreotide therapy was demonstrated in three patients with moderate to severe TAD. weight loss and measures such as compression bandaging and topical corticosteroid application still remain the most cost effective treatment modalities for TAD.
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ranking = 1.6403390723074
keywords = thyroid-associated
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7/14. Euthyroid pretibial myxedema.

    Pretibial myxedema is typically associated with clinical hyperthyroidism, diffuse goiter, and ophthalmopathy in patients with Graves' disease. A case of biopsy-proved pretibial myxedema was encountered in a clinically euthyroid woman who had neither diffuse goiter nor exophthalmos. Although serum total and free thyroxine hormone concentrations were normal, the thyroid-stimulating hormone response to thyrotropin-releasing hormone was absent. This case illustrates that pretibial myxedema may present without other more common manifestations of Graves' disease. In patients with suspect pretibial skin lesions, the thyrotropin-releasing hormone stimulation test may be required to establish the presence of subtle underlying thyroid gland autonomy and the diagnosis of euthyroid pretibial myxedema.
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ranking = 1
keywords = ophthalmopathy
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8/14. Successful treatment of exophthalmos and pretibial myxoedema with plasmapheresis.

    A patient with Graves's disease with acute progressive exophthalmos and pretibial myxoedema was treated twice with plasmapheresis. Two weeks after the first treatment the symptoms recurred, but 20 weeks after the second treatment the exophthalmos was much improved and the pretibial myxoedema had disappeared. Analysis of sequential serum IgG concentrations and the thyroid-stimulating immunoglobulin index suggested that the two conditions were caused by specific IgGs. The results suggest that plasmapheresis has a useful place in the treatment of acute and rapidly progressive ophthalmopathy and pretibial myxoedema in patients with Graves's disease.
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ranking = 1
keywords = ophthalmopathy
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9/14. Acute pre-tibial myxoedema following radioiodine therapy for thyrotoxic Graves' disease.

    A 54-year-old woman was treated with an oral dose of 555MBq of 131I radioiodine for thyrotoxicosis. She had no clinically detectable extrathyroidal manifestations of Graves' disease at the time, but within two months developed moderately severe ophthalmopathy and very extensive thyroid dermopathy affecting her face, arms, hands and feet, in addition to the classic pre-tibial area. Although she developed mild post radioiodine hypothyroidism, this was detected at an early stage and its treatment had no effect on the extrathyroidal signs. Thyrotrophin receptor antibodies (TRAb) were positive before treatment (22% inhibition of TSH binding in neat serum), rose to very high levels following radioiodine (97.6% inhibition), and fell progressively over the following year during treatment with prednisolone. thyroglobulin autoantibodies became detectable following radioiodine but thyroid peroxidase antibodies were undetectable throughout. serum and purified IgG from blood samples obtained prior to steroid therapy and over the subsequent year were tested on a dermal fibroblast cell line in vitro for the stimulation of synthesis of glycosaminoglycans, protein and dna, but no increase in radiolabel incorporation was apparent for any sample when compared to controls. The temporal relation between the radioiodine and the acute onset of dermopathy and ophthalmopathy, together with the abrupt rise in TRAbs, indicates a probable causal association. However, the absence of in-vitro fibroblast stimulation would suggest that the pathogenesis of Graves' dermopathy is not dependent solely on any simple humoral factor.
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ranking = 2
keywords = ophthalmopathy
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10/14. Successful treatment of hypothyroid Graves' disease with a combination of levothyroxine replacement, intravenous high-dose steroid and irradiation to the orbit.

    A 46-year-old woman with hypothyroid Graves' disease (EMO syndrome) is reported. The patient had bilateral exophthalmos, conjunctival chemosis, periorbital edema and limitation of lateral gaze. Laboratory examination revealed the presence of primary hypothyroidism with positive thyroid-stimulating hormone (TSH) binding inhibitory immunoglobulin and thyroid stimulation antibody. These findings indicated a diagnosis of hypothyroid Graves' disease or EMO syndrome. She received levothyroxine replacement and steroid pulse therapy followed by radiotherapy. Her visual symptoms showed marked improvement and pretibial myxedema disappeared. Although several studies indicate that hypothyroid Graves' disease is a different entity from hyperthyroid Graves' disease, this report suggests that steroid pulse therapy combined with radiotherapy may be effective to treat ophthalmopathy in both diseases.
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ranking = 1
keywords = ophthalmopathy
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