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PubMed2002

Severe graves ophthalmopathy accompanied by HTLV-1-associated uveitis (HAU) and anti-neutrophil cytoplasmic antibody-related vasculitis: a case report and a review of the literature of HAU with Graves disease.

A 50-year-old woman was admitted because of severe exophthalmos associated with Graves disease. She underwent methimazole (MMI) and methylprednisolone pulse therapy against exophthalmos. She noticed photophobia and blurred vision 3 weeks after the start of pulse therapy and she was diagnosed as having uveitis. Methylprednisolone pulse therapy was performed again for both exophthalmos and uveitis, followed by daily administration of 20 mg of prednisolone and instillation of betamethasone for 2 weeks and the uveitis was improved. Western blot analysis confirmed that human T lymphotropic virus type 1 (HTLV-1) antibody was present in her serum. Propylthiouracil was substituted for MMI and HTLV-1-associated uveitis (HAU) has not recurred. Six months after the beginning of administration of PTU, anti-neutrophil cytoplasmic antibody-related vasculitis developed in the patient. We review 43 cases of HAU with Graves disease, including the present case, in the literature. Only 2 of 27 cases (except unknown cases) (7.4%) had Graves ophthalmopathy. To the best of our knowledge, there has been no investigation of HAU and Graves ophthalmopathy.

Anti-Inflammatory Agents↗
PubMedDate not supplied

Radiotherapy for moderate-to-severe Graves' ophthalmopathy: improved outcomes with early treatment.

PURPOSE: The purpose of this review is to quantify the response to radiotherapy delivered early in the active inflammatory phase of moderate-to-severe Graves' ophthalmopathy. MATERIALS AND METHODS: A retrospective review of radiotherapy delivered early in the active phase of Graves' ophthalmopathy was performed. All 47 cases had moderate-to-severe ophthalmopathy, and 30 cases had optic neuropathy. Variables examined included exophthalmos, color vision, and resistance to retropulsion. Statistical analyses comparing these variables at presentation and at 3 and 6 months after radiotherapy were performed. Subset analyses comparing responses of patients with symptoms lasting longer or less than 6 months were also performed. RESULTS: At 6 months after radiotherapy, there was improvement in exophthalmos in 74.5% of cases and improvement in retropulsion in 83.0%, and all cases of previous color deficiency improved. The mean improvement in exophthalmos was 1.38 mm, color vision was two plates, and retropulsion was 1 grade. On subset analyses, there was a trend toward greater improvement in patients treated earlier in the course of their symptoms. CONCLUSIONS: Radiotherapy may play an important role in the treatment of Graves' ophthalmopathy, especially for patients with optic neuropathy. Early intervention (symptoms < 6 months) with radiotherapy may be the optimal timing for this treatment.

Analysis of Variance↗
PubMed1980

Graves' disease and HLA: clinical and epidemiologic associations.

Correlation between clinical and epidemiological features of Graves' disease and HLA were sought in 175 patients, eighty-three of whom were typed for HLA DR antigens. The relative risks (x) conferred by HLA-B8 and DR3 were 3.1 and 5.7 respectively (P less than 0.0005 uncorrected). The disease occurred at an earlier age in HLA-DR3 positive patients compared to negative patients (P less than 0.005 in females greater than and less than 30-years-old). Eighty-four out of ninety-two patients could specify the season of onset of hyperthyroidism; an excess of HLA-B8 positive patients in the summer and a lack of these in the spring and autumn was found. No association between B8 and exophthalmos and/or soft tissue eye changes were observed. However, significant associations between exophthalmos and either exophthalmos and/or soft tissue changes were found with DR3 (x = 3.6 and 3.8 respectively). HLA-DR3 positive patients were found to be more resistant to radioiodine therapy than patients negative for these antigens. No heterogeneity in the distribution of HLA antigens was found when the following indices were examined: sex, goitre size, severity of disease, pretibial myxoedema, antecedent psychological disorder, consumption of oral contraceptives, family history of Graves' disease or thyroid antibody titre. HLA does not appear to distinguish subvariants of Graves' disease, rather it influences the susceptibility to disease and its persistence once it becomes clinically manifest.

Adult↗
PubMed1990

Correlation of orbital computed tomography and antibodies in patients with hyperthyroid Graves' disease.

The purpose of this study was to elucidate the incidence of orbital changes in computed tomography, and the relationships of orbital changes with eye-muscle antibody (EMAb), thyrotrophin-binding inhibitor immunoglobulin (TBII), antithyroglobulin antibody (TgAb) and anti-thyroid microsomal antibody (MsAb), in 55 consecutive patients with hyperthyroid Graves' disease. EMAb was demonstrated by an immunoperoxidase technique. The results showed that the prevalence of orbital fat increase, extraocular muscle enlargement, lacrimal gland enlargement and optic nerve swelling were 54.5, 41.8, 21.8 and 16.5% respectively. There were 32.7% of patients without any orbital changes. The patients with extraocular muscle enlargement, lacrimal gland enlargement and optic nerve swelling had higher titres of EMAb than the patients without these orbital changes. The patients with lacrimal gland enlargement had a lower TBII index than those without. There was no difference in TgAb or MsAb between the patients with extraocular muscle enlargement, lacrimal gland enlargement, optic nerve swelling or orbital fat increase and those patients without these orbital changes. The patients with extraocular muscle enlargement, optic nerve swelling and orbital fat increase had a higher degree of exophthalmos than the patients without these orbital changes. EMAb was negatively correlated with TBII. However, there was no correlation between EMAb and TgAb or MsAb. The degree of exophthalmos was positively correlated with EMAb and negatively correlated with age. There was no correlation between exophthalmos and TBII, TgAb or MsAb. In conclusion, although the pathogenesis of orbital changes and hyperthyroidism is different, a close link between these two entities is suggested by the high incidence of orbital changes in hyperthyroid Graves' disease.(ABSTRACT TRUNCATED AT 250 WORDS)

Adolescent↗
PubMed1993

Correlation of orbital muscle changes evaluated by magnetic resonance imaging and thyroid-stimulating antibody in patients with Graves' ophthalmopathy.

To evaluate the relationship between eye changes and autoantibody to the thyrotropin receptor in patients with Graves' disease, we evaluated the eye changes using magnetic resonance imaging and the results were correlated with thyroid-stimulating antibody, thyrotropin binding inhibitor immunoglobulin and thyroid growth activity. Subjects were 15 patients with Graves' disease who had Graves' ophthalmopathy, including exophthalmos and other signs and symptoms, and nine patients without ophthalmopathy; all were maintained in a euthyroid state by antithyroid drugs. The thyrotropin-binding inhibitor immunoglobulin was measured by a kit, and thyroid-stimulating antibody and thyroid growth activity were evaluated by cyclic adenosine 3',5'-monophosphate production and [3H]thymidine incorporation, respectively, by cultured functional rat thyroid lined cells. The sum of the swelling ratios (muscle thickness to the diameter of the optic nerve) of the four extraocular muscles correlated well with the degree of exophthalmos. The thyrotropin-binding inhibitor immunoglobulin was positive in nine out of 15 patients with ophthalmopathy; however, no correlation was observed between the activity and exophthalmos or muscle swelling. No significant correlation was observed between muscle changes and thyroid growth activity either. On the other hand, thyroid-stimulating antibody (642 +/- 91%) in Graves' patients with ophthalmopathy was significantly (p < 0.02) higher than that (315 +/- 84%) in patients without ophthalmopathy. Moreover, the level of stimulating activity in Graves' patients with ophthalmopathy showed a significant (p < 0.02) positive correlation with the sum of the swelling ratios of the individual eight eye muscles. These results suggest that thyroid-stimulating antibody has a close relation to Graves' ophthalmopathy.

Adolescent↗
PubMed1988

A four-wall orbital decompression for dysthyroid orbitopathy.

A new surgical technique is described for the relief of the ocular manifestations of dysthyroid orbitopathy. Surgical decompression of the orbit was reserved for patients who failed to respond to medical treatment. Eleven patients (nine women and two men) were followed for periods from 3 to 48 months. Visual loss was the commonest presentation and the main indication for surgery. All patients had failed to respond to medical therapy, consisting of steroid and/or radiation therapy. Three patients had previously undergone orbital decompressions with limited success. Six patients had preoperative visual acuity of worse than 20/200. Preoperative exophthalmos ranged from 24 to 35 mm (normal 16 mm). The orbits were approached by a bifrontal scalp flap with exposure of the temporal and infratemporal fossae. The orbital roof was approached transcranially. The posterior wall of the frontal sinus was removed together with the mucous lining. To maximize the decompression, the periosteum surrounding the orbital contents was incised, allowing the contents to prolapse through the newly created windows into the maxillary antrum, temporal fossa, anterior cranial fossa, and nose. During the past 3 years, 22 orbits in 11 patients have been decompressed by this technique. Symptomatic relief was obtained in all but one patient who still had significant exophthalmos with keratitis, but did have a dramatic improvement in vision. The decrease in exophthalmos ranged from 1 to 13 mm (mean 7 mm). The most dramatic improvement was in vision, with postoperative acuity of 20/20 to 20/30 in all but two patients. There were no deaths and no major morbidity.

Adult↗
PubMedDate not supplied

[Orbital decompression by endoscopic transnasal and transantral approach in thyroid-associated ophthalmopathy].

OBJECTIVES: We evaluated the results of orbital decompression in patients with Graves' disease. PATIENTS AND METHODS: Orbital decompression was performed by endoscopic transnasal medial wall approach and transantral inferior wall approach in 18 eyes of 12 patients (8 males, 4 females; mean age 41 years; range 24 to 65 years). The degree of exophthalmos was evaluated with the use of the Hertel exophthalmometer preoperatively, and postoperatively in the 24th hour, first and third months. RESULTS: At the end of the third month, exophthalmos decreased by a mean of 4.4 mm (range 3 to 7 mm). Visual acuity and field defects improved considerably in all patients. Complications occurred in three cases (25%) including diplopia, displacement of one eye inferiorly, and maxillary sinusitis caused by the encroachment of the adipose tissue upon the ostium, respectively. CONCLUSION: With the increasing ease of surgical procedures and enhanced experience on the part of surgeons, successful treatment of exophthalmos has become possible with decreased complication rates.

Adolescent↗
PubMed1987

HLA immunogenetic heterogeneity in black American patients with Graves' disease.

Seventy-three American black patients with Graves' disease were typed for HLA-A, HLA-B, and HLA-DR antigens. There was a slight increase in HLA-DRw6 antigen frequency compared with 238 normal American black controls, but this was not significant after correction for the number of antigens tested. A significant increase in HLA-DR4 and HLA-DRw6 frequency was found in a subgroup of patients with exophthalmos (22.9% and 29.2% compared with 7.6% and 10.1% of normal black controls). There was a significant increase in HLA-DRw6 in a subgroup of patients who were thyroid antibody-positive (26.0%). The increment in HLA-DRw6 was higher in 32 patients who had both exophthalmos and who were antibody positive (37.5%). A significant increase in HLA-DR5 was found in a subgroup of patients who did not have exophthalmos and who were antibody-negative (83.3%). Our findings support previous evidence for immunogenetic heterogeneity in patients with toxic Graves' disease. In American blacks HLA-DRw6 is in some way associated with the disease in contrast to the well-recognized HLA-DR3 association in whites.

Black or African American↗
PubMed1994

The epidemiologic characteristics and clinical course of ophthalmopathy associated with autoimmune thyroid disease in Olmsted County, Minnesota.

Among incident cases of GO in Olmsted County, Minnesota: GO affected females six times more frequently than males (86% versus 14% of cases, respectively). The age-adjusted incidence rate was 16 cases per 100,000 population per year for females and 2.9 cases per 100,000 population for males. The peak incidence rates were bimodal, occurring in the age groups 40 to 44 years and 60 to 64 years in females and 45 to 49 years and 65 to 69 years in males. Among patients with GO, approximately 90% had Graves' hyperthyroidism, 1% had primary hypothyroidism, 3% had Hashimoto's thyroiditis, and 5% were euthyroid. Eyelid retraction was the most common ophthalmic feature of autoimmune thyroid disease, being present either unilaterally or bilaterally in more than 90% of patients at some point in their clinical course. Exophthalmos of one or both eyes affected approximately 60% of patients, restrictive extraocular myopathy was apparent in about 40% of patients, and optic nerve dysfunction occurred in either one or both eyes in 6% of patients with autoimmune thyroid disease. Only 5% of patients had the complete constellation of classic findings: eyelid retraction, exophthalmos, optic nerve dysfunction, extraocular muscle involvement, and hyperthyroidism. Upper eyelid retraction, either unilateral or bilateral, was documented in approximately 75% of patients at the time of diagnosis of GO. Lid lag also was a frequent early sign, being present either unilaterally or bilaterally in 50% of patients at the initial examination. At the time of diagnosis of GO, the most frequent ocular symptom was pain or discomfort, which affected 30% of patients. Some degree of diplopia was noted by approximately 17% of patients, lacrimation or photophobia was present in about 15% to 20% of patients, and 7.5% of patients complained of blurred vision. Decreased vision attributable to optic neuropathy was present in less than 2% of eyes at the time of diagnosis of GO. Thyroid dermopathy and acropachy accompanied GO in approximately 4% and 1% of patients, respectively. Myasthenia gravis occurred in less than 1% of patients. Superior limbic keratoconjunctivitis was documented in less than 4% of patients. The median age at the time of diagnosis of GO was 43 years (range, 8 to 88). Among patients with hyperthyroidism, 61% developed ophthalmopathy within 1 year of the onset of thyrotoxicosis. Symptoms and signs for which statistically significant changes occurred between the initial and final examinations included lacrimation, pain or ocular discomfort, photophobia, eyelid retraction, lid lag, eyelid fullness, conjunctival injection, chemosis, and exophthalmos.(ABSTRACT TRUNCATED AT 400 WORDS)

Adolescent↗
PubMed1996

[Intraorbital conjunctival cyst after a penetrating orbital injury: a case report].

We report a case of intraorbital conjunctival cyst following a penetrating orbitocranial injury. The patient was a 28-year-old male who was hospitalized with exophthalmos, retrobulbar pain and upper gaze disturbance of his left eye. When he was 4 years old, a thin iron rod had penetrated intracranially through the inner angle of his left orbit. He was hospitalized and treated conservatively for about two weeks. The left eye ball was intact and visual acuity was normal, although bloody fluid had continuously flowed out from the left inner angle of the conjunctival wound for a few days. He had been febrile to 39 degrees C and complained of headache for one week. Subsequentry, the symptoms gradually improved through conservative therapy. When he was a junior high school student, he noticed exophthalmos of his left eye. However, he had never been examined closely, until he was 28 years old. We suspect that he had suffered from meningitis caused by the penetrating orbitocranial injury, and had fortunately improved under the conservative therapy. On admission to our hospital, a craniogram showed fracture of the left orbital roof, and coronal and three-dimensional computed tomography (CT) scans clearly demonstrated the orbital fracture. CT revealed a cystic mass in the retrobulbar space, and a porencephalic cyst in the medial basal frontal lobe. On magnetic resonance imaging (MRI) scans, both cysts were of low intensity on T1-weighted imaging, and of high intensity on T2-weighted images. Coronal and sagittal MRI scans showed that the two cysts were connected with each other through the fracture in the orbital roof. We diagnosed therefore that the orbital cyst was a herniated porencephalic cyst of the frontal lobe. Surgery was performed by a transcranial approach. The porencephalic cyst adhered to the fractured lesion of the frontal base but did not extend into the orbita. The intraorbital cyst was totally removed by opening the orbital roof including the fractured lesion. The cyst contained milky fluid. Postoperatively, the exophthalmos, retrobulbar pain and upper gaze disturbance showed gradual improvement. On histological examination, the cyst was found to be lined by non-keratinized stratified squamous epithelium and was diagnosed as a conjunctival cyst. This case was considered to be one of traumatic conjunctival cysts caused by a penetrating orbitocranial injury. Orbital conjunctival cysts have been reported to comprise about 10% of orbital epidermoid and dermoid cysts. Of these cysts, traumatic conjunctival cysts are rare, and only a few cases have been described. The etiology and therapy of orbital conjunctival cysts are discussed.

Adult↗
PubMed1996

Graves' disease of the orbit.

Graves' disease, the most common orbital disorder, affects approximately 0.5 % of the population of the United States. It is the underlying cause in 15% to 28% of cases of unilateral exophthalmos and in 80% of cases with bilateral exophthalmos. Also called "thyroid-associated ophthalmopathy", autoimmune thyroid disease," "endocrine exophthalmos," and "thyroid eye disease", Graves' disease presents with enlargement of the extraocular muscles, increased orbital fat volume, and venous stasis caused by increased orbital pressure. This article discusses the clinical evaluation and radiologic findings of this common orbital disorder.

Diagnosis, Differential↗
PubMed1998

Presumed adenocarcinoma of the retinal pigment epithelium in a blind eye with a staphyloma.

The retinal pigment epithelium (RPE) can undergo reactive hyperplasia and metaplasia following a variety of ocular insults. However, true neoplasms of the RPE are rare. We report a case of a papillary adenocarcinoma of the RPE arising in the blind staphylomatous right eye of a 79-year-old woman with a long history of bilateral posterior staphylomas who was seen with increasing pain and exophthalmos of the right eye. Findings from ultrasonography and computed tomography demonstrated linear calcification consistent with osseous metaplasia of the RPE. Progression of the exophthalmos and worsening exposure keratitis led to enucleation of the eye. Gross pathology showed a 79-mm-long globe. Histopathologic findings revealed a largely amelanotic papillary adenocarcinoma arising from the RPE. Positive immunoreactivity for cytokeratin supported the epithelial origin of the tumor. Adenocarcinoma of the RPE is rare but may develop in a blind eye.

Adenocarcinoma, Papillary↗
PubMed1983

Choroidal folds and refractive errors associated with orbital tumors. An analysis.

Twenty patients with unilateral exophthalmos secondary to orbital tumors underwent fundus photography; 16 underwent fluorescein angiography. Choroidal folds were found in 15 eyes. The location of each tumor with respect to the globe was determined by computed tomography. In addition, most patients underwent B-scan ultrasonography and surgical exploratory procedures. Choroidal folds were more common in patients with greater amounts of exophthalmos and with anteriorly located tumors. Moreover, the pattern of choroidal folds generally reflected the location of the tumor within the orbit. Comparison with the uninvolved fellow eyes showed that preoperative refractive errors were usually shifted toward hyperopia with intraconal tumors, whereas extraconal tumors were typically associated with higher astigmatic errors on the involved side.

Diagnosis, Differential↗
PubMed2001

Orbital mucosa-associated lymphoid tissue (MALT)-type lymphoma in a patient with relapsing polychondritis.

Relapsing polychondritis is characterized by recurrent inflammation of the cartilaginous tissues of the ears, nose, peripheral joints, and the tracheobronchial tree. The eye is also a frequent target organ in relapsing polychondritis, and proptosis is a well-recognized manifestation of eye involvement. Similar to other rheumatologic diseases, an association of relapsing polychondritis with malignancy has been reported. We describe a patient with relapsing polychondritis who presented with exophthalmos. When treatment directed toward control of her underlying disease was only partially effective, further investigation revealed that she had an orbital mucosa-associated lymphoid tissue (MALT)-type B cell lymphoma. We hypothesize that the lymphoma resulted from malignant transformation of the relapsing polychondritis-induced inflammatory pseudotumor and emphasize that neoplastic disease should be considered in the differential diagnosis in patients with relapsing polychondritis presenting with exophthalmos.

Adult↗
PubMed1988

Mouse exophthalmic chronic orbital inflammatory disease. Induction by human leucocyte intracellular Mollicutes.

Mollicutes are cell wall deficient bacteria which may be overlooked or confused with viruses because of indistinct light microscopic morphology, poor staining, difficulty in cultivation, and the ability to pass 0.450 micron filters. As they have a distinctive ultrastructural appearance they can be identified using transmission electron microscopy [TEM]. Using TEM vitreous leucocytes from chronic endogenous uveitis patients may demonstrate non-cultivable intracellular 0.005-1.0 micron mollicute-like organisms [MLO], some of which develop into distinctive cell walled cocci, 0.5-0.7 micron in diameter. Inoculation of those MLO containing human vitreous into mouse eyelids produces chronic cardiac and uveal vasculitis with orbital inflammation. Similar MLO are found within the mouse lesional leucocytes. This report describes the chronic orbital inflammation with vasculitis in 67 of 100 of those MLO inoculated mice versus 0 of 200 controls (P less than 0.05). Exophthalmos with inflammation also occurred in 12 of those 67 mice (P less than 0.05). MLO were found within orbital lesional leucocytes of 10 of 10 of those mice using a TEM versus 0 of 10 controls. The results indicate that vasculitis and exophthalmos were important features of this MLO induced mouse orbital inflammation. The implication of these results for human idiopathic chronic orbital inflammatory disease is discussed.

Animals↗
PubMed1979

Recognition and differential diagnosis of enlarged extraocular muscles in computed tomography.

We found enlargement of the extraocular muscles in 70 patients out of 603 orbit studies, of whom 310 had exophthalmos. The majority (46 of 70) had the eye signs of Graves' disease. Arteriovenous malformations and carotid cavernous fistulas can cause enlargement of the extraocular muscles by a diffuse increase in orbital venous pressure, Acute orbital myositis can be distinguished from other forms of pseudotumor by the presence of a single enlarge extraocular muscle with associated inflammatory findings which responds to corticosteroid therapy. Neoplasms may invade extraocular muscles or compress their venous drainage causing secondary muscle enlargement. In all these patients the presence of a mass was correctly identified. The ability of the computed tomography scanner to recognize abnormalities of the extraocular muscles represents a significant advance in classification and diagnosis of the causes of exophthalmos.

Adult↗
PubMed2005

Bilateral orbital tumor as initial presenting sign in human T-cell leukemia virus-1 associated adult T-cell leukemia/lymphoma.

PURPOSE: To report a case of human T-cell leukemia virus type1 (HTLV-1)-associated adult T-cell leukemia/lymphoma (ATLL) whose initial sign was exophthalmos. DESIGN: Case report. METHODS: A 64-year-old Japanese man presented with exophthalmos and choroidal folds of the right eye without general symptoms and received ophthalmologic and laboratory examination, magnetic resonance imaging (MRI), and orbital biopsy. RESULTS: Antibodies against HTLV-1 were extremely high but atypical lymphocytes were not present in the peripheral blood. MRI showed multiple tumorous lesions in both orbits. Orbital biopsy revealed an orbital mass that consisted of atypical lymphocytes originally from T cells and established the diagnosis of lymphoadenopathy type of ATLL. CONCLUSION: Although ocular involvement by ATLL is extremely rare, ATLL can first present in the orbit, and only the results of a biopsy can provide definitive information for its diagnosis.

Antibodies, Viral↗
PubMed2006

Sclerosing therapy as first line treatment for low flow vascular lesions of the orbit.

PURPOSE: To evaluate the outcome of sodium morrhuate 5% injections in patients with low flow vascular lesions, which consist of orbital lymphangiomas, and in one patient with intraosseous cavernous hemangioma. DESIGN: Prospective, interventional consecutive case series. METHODS: Intralesional sodium morrhuate 5% was injected under direct visualization or under radiographic guidance to six patients with orbital lymphangiomas and one patient with intraosseous cavernous hemangioma. Comprehensive eye examination and follow-up imaging studies were performed. main outcome measures: Lesion size was evaluated by orbital imaging and clinical examination, visual acuity, exophthalmos, and posttreatment complications. RESULTS: Seven patients (four female, three male; average age, 33 years) were included. Six patients were diagnosed with orbital lymphangioma, and one patient was diagnosed with intraosseous cavernous hemangioma. Patients received an average of 2.6+/-2 intralesional injections of sodium morrhuate, with a range of one to six injections and a mean volume of 0.9+/-0.8 ml (range, 0.2 to 2.1 ml). Lesions showed a decrease in size an average of 50% (33%) and ranged from minimal (10%) to near total resolution (85%). Visual acuity and intraocular pressure remained unchanged; exophthalmos decreased an average of 1.5+/-1.8 mm. Complications included one case of orbital hemorrhage that resolved spontaneously and transient keratopathy in all patients with anterior orbital lesions. CONCLUSION: Intralesional sclerosing therapy with sodium morrhuate 5% is effective in tumor debulking in patients with orbital lymphangioma and is not associated with vision-threatening complications. It may be a better alternative to surgery for low flow orbital tumors, which includes lymphangioma.

Adolescent↗