Cases reported "Graves Disease"

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1/19. Congenital hyperthyroidism: autopsy report.

    We report the autopsy of a stillborn fetus with congenital hyperthyroidism born to a mother with untreated Graves' disease, whose cause of death was congestive heart failure. The major findings concerned the skull, thyroid, heart, and placenta. The cranial sutures were closed, with overlapping skull bones. The thyroid was increased in volume and had intense blood congestion. Histological examination showed hyperactive follicles. The heart was enlarged and softened, with dilated cavities and hemorrhagic suffusions in the epicardium. The placenta had infarctions that involved at least 20% of its surface, and the vessels of the umbilical cord were fully exposed due to a decrease in Wharton's jelly. hyperthyroidism was confirmed by the maternal clinical data, the fetal findings of exophthalmia, craniosynostosis, and goiter with signs of follicular hyperactivity. Craniosynostosis is caused by the anabolic action of thyroid hormones in bone formation during the initial stages of development. The delayed initiation of treatment in the present case contributed to the severity of fetal hyperthyroidism and consequent fetal death.
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2/19. propylthiouracil-induced antineutrophil cytoplasm antibody-positive anaphylactoid purpura-like vasculitis--a case report.

    propylthiouracil (PTU), a drug commonly used for treatment of hyperthyroidism, is associated with various rare side effects. Antineutrophil cytoplasm antibody (ANCA)-positive vasculitis is a relatively unusual complication among them. The pathogenesis of ANCA-positive vasculitis during PTU therapy is still obscure. We present the case of a 12-year-old boy who developed ANCA-positive vasculitis during PTU therapy for Graves' disease. His symptoms and signs were indistinguishable from anaphylactoid purpura, a common small-vessel vasculitis in children. The clinical manifestations improved after discontinuation of PTU and immunosuppressant treatment. He remained symptom-free at 11-months follow-up.
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3/19. Severe myocardial ischemia following hormone replacement in two cases of hypothyroidism with normal coronary arteriogram.

    Two cases of hypothyroidism with cardiac attack (acute myocardial infarction, AMI) following thyroxine replacement were reported. Neither of these cases showed any major coronary artery disease. The first case was a 58 year-old male who was treated with L-thyroxine (initial dose 0.025 mg/day) for hypothyroidism due to Hashimoto's disease. The dose was increased up to 0.1 mg/day within 2 weeks. Acute myocardial infarction occurred 6 weeks after the replacement was started. Angiographical study showed no notable pathological change in major coronary arteries, but echocardiography demonstrated diffuse hypokinesis of the left ventricular wall. The second case was a 61-year-old female who suffered from Graves' disease and had been treated with thiamazole (2.5 mg/day) for 15 years. Later, she became hypothyroid and was treated with thyroxine. At first, 0.05 mg/day of L-thyroxine was given, and then the dose was increased up to 0.1 mg/day after the 7th week. Acute myocardial infarction occurred 3 weeks after the dose was increased. Angiographic study of the coronary arteries revealed no abnormality. Possible causes of AMI in thyroxine replacement were discussed in relation to vascular spasm and small vessel disease of the heart. Importance of echocardiographic study before hormone replacement therapy is stressed, particularly for middle/old-aged patients with long-term hypothyroidism.
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4/19. Antiphospholipid antibody syndrome complicated by Grave's disease.

    The report describes a woman with primary antiphospholipid antibody syndrome complicated with Grave's disease. Developing symptoms included a small cutaneous nodule on her finger and subsequently ecchymotic purpura on the cheeks, ears, buttocks and lower legs. Histological examinations showed thrombosed vessels in the dermis without or with hemorrhage, respectively. Laboratory investigation revealed positive lupus anticoagulant and immunogenic hyperthyroidism due to Grave's disease. There is a close relationship between the cutaneous manifestation of antiphospholipid antibody syndrome and the activities of Grave's disease and a possible link of antiphospholipid antibody syndrome with Grave's disease was suggested both by the etiology of the disease as well as the disease activity.
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5/19. Palpebral edema as a cutaneous manifestation of hyperthyroidism.

    Three cases of palpebral edema associated with Graves' disease are described. These patients had unilateral edema and minimal erythema of the upper eyelid. Notable was that, histologically, dermal edema and dilation of lymphatic vessels were observed, but deposition of mucopolysaccharides was not. In 2 cases, edema of the eyelid was resistant to treatment with an antithyroid drug. Unilateral edema of the upper eyelid is an important cutaneous manifestation that indicates the presence of hyperthyroidism.
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6/19. Multiple intracranial arterial stenoses around the circle of willis in association with Graves' disease: report of two cases.

    OBJECTIVE AND IMPORTANCE: The association of Graves' disease with multiple intracranial arterial stenoses is rare. CLINICAL PRESENTATION: We report on two Japanese women who experienced the concurrence of Graves' disease and cerebral ischemia attributable to multiple intracranial arterial stenoses around the circle of willis. Clinically, these patients demonstrated hyperthyroidism, goiter, ophthalmopathy, and ensuing ischemic strokes. cerebral angiography demonstrated multiple intracranial arterial stenoses around the circle of willis in both cases. These cases did not meet the full diagnostic criteria for moyamoya disease, in that there were no abnormal, net-like, collateral vessels, but the other clinical and angiographic findings were consistent with this condition. INTERVENTION: After normalization of their hormonal conditions, the patients underwent cerebral revascularization procedures. Both patients achieved excellent recoveries and returned to normal daily life after treatment. CONCLUSION: Multiple intracranial arterial stenoses and Graves' disease may occur simultaneously. Such cases may offer new insights into the pathogenesis of these two conditions. It is important to study more patients with this dual condition, to obtain more evidence of the relationship between genetic and immunogenic backgrounds.
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7/19. PTU-associated cutaneous vasculitis with ANCA anti-MPO and anti-PR3 antibodies.

    A 36-year-old woman presented at our clinic with symmetrical, tender, palpable purpuric lesions on her lower legs and buttocks after restarting PTU therapy for relapsing Graves' disease. PTU-induced vasculitis was diagnosed with remarkable ANCA anti-MPO and anti-PR3 antibody positivity. The purpuric skin lesions resolved immediately after discontinuation of the drug and the ANCA titres lowered. In the presence of activated neutrophils, PTU could induce a high cytotoxity and injure the vessel walls. Treatment of choice is discontinuation of the drug. Sometimes more aggressive therapy as cyclophosphamide or plasmapheresis is warranted.
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8/19. A case of propylthiouracil-induced pyoderma gangrenosum associated with antineutrophil cytoplasmic antibody.

    A 27-year-old woman who had been receiving propylthiouracil for 2 years for Graves' disease presented with painful ulceration on the lower limbs which had first appeared 2 weeks previously. Well-circumscribed hemorrhagic ulcerations with ragged borders were noted on both legs. skin biopsy demonstrated a florid neutrophilic infiltrate and evidence of leukocytoclasis around small blood vessels in the papillary dermis compatible with the diagnosis of pyoderma gangrenosum. A highly positive perinuclear pattern of antineutrophil cytoplasmic antibody with specificities for IgM myeloperoxidase was observed. The authors think that propylthiouracil is associated with the occurrence of pyoderma gangrenosum in this patient.
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9/19. Progressive bilateral sensorineural hearing loss induced by an antithyroid drug.

    We report a patient with antithyroid drug-induced progressive bilateral sensorineural hearing loss associated with myeloperoxidase-antineutrophil cytoplasmic antibodies (MPO-ANCA). While antithyroid drugs have been linked to MPO-ANCA-associated small-vessel vasculitis, sensorineural hearing loss rarely was noted. A 36-year-old man treated for hyperthyroidism with propylthiouracil (PTU) developed progressive bilateral sensorineural hearing loss accompanied by fever and arthritis. MPO-ANCA were demonstrated in serum. Distortion product otoacoustic emissions test results suggested dysfunction of outer hair cells of the organ of corti. Inner ear blood flow impairment from ANCA-associated small-vessel vasculitis presumably caused cochlear dysfunction. PTU withdrawal and high-dose methylprednisolone administration greatly improved hearing on both sides.
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10/19. Rapidly progressive fatal bihemispheric infarction secondary to Moyamoya syndrome in association with Graves thyrotoxicosis.

    Moyamoya syndrome is the secondary form of intracranial arterial occlusive diseases that produces collateral vessels from the base of brain. We report a case of Moyamoya syndrome developing in association with Graves thyrotoxicosis; as a result of its rapid progression and severe global ischemia, it was ultimately fatal. Because of the rarity of this association, we reviewed the literature in an attempt to establish possible demographic and clinical characteristics that may suggest putative mechanisms of pathogenesis.
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