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PubMed2003

Reconstruction of Sphenoid Wing Dysplasia with Pulsating Exophthalmos in a Case of Neurofibromatosis Type 1 Supported by Intraoperative Navigation Using a New Skull Reference System.

A patient with neurofibromatosis type 1 had pulsating exophthalmos of the right eye with diplopia resulting from severe dysplasia of the sphenoid bone and consecutive herniation of the right temporal lobe. The right orbital tectum was reconstructed with titanium mesh and iliac spongiosa via a lateral orbitotomy using intraoperative navigation. For intraoperative referencing a cortical fixed-reference system and a skin scanning laser device were used. Postoperatively, the diplopia was reduced, but the patient asked for further treatment and the procedure was repeated 6 months later. Intraoperatively, the previously implanted titanium mesh was deformed and most of the transplanted bone was resorbed, probably because of pressure. A more extended mesh was implanted and iliac spongiosa was placed on both sides. Intraoperative navigation was used during both procedures. The adverse effects of diplopia were minimized and follow-up computed tomography after seven months confirmed that the bone graft was in place.

Case Reports↗
PubMed1999

Exophthalmos, pretibial myxedema, osteoarthropathy syndrome associated with papillary fibroelastoma in the left ventricle.

EMO syndrome, a rare complication of Graves' disease, exhibits exophthalmos, pretibial myxedema, and osteoarthropathy. The presence of functional thyrotropin receptors (TSHR) in adipocytes and osteoblasts, both of which we have recently observed, may be related to these extrathyroidal manifestations of Graves' disease. In addition, the expression of TSHR in the heart has recently been reported. We describe here a patient with Graves' disease exhibiting EMO syndrome with a papillary fibroelastoma in the left ventricle. Pathological examinations showed that the fibroelastoma contained Alcian blue-stained mucinous materials that were also observed in the subcutaneous tissue of pretibial myxedema.

Fibroma↗
PubMed2004

Prevalence of asymmetric exophthalmos in Graves orbitopathy.

PURPOSE: To report the prevalence and the degree of significant asymmetric orbital involvement secondary to thyroid-related (Graves) orbitopathy in a large patient population. METHODS: The prevalence of exophthalmometric differences of > or =2 mm was determined in 391 consecutive patients diagnosed with Graves orbitopathy from the Jules Stein Eye Institute Division of Ophthalmic Plastic and Reconstructive Surgery patient registry. RESULTS: Thirty-six of 391 patients (9%) in this cohort had asymmetric proptosis with a difference of > or =2 mm, based on single-observer Hertel measurements before orbital decompression surgery. This finding was similar among men (11/95, 12%) and women (25/296, 8%) but was not statistically significant (P= 0.358; 95% CI, 0.6% to 7.8%). Also, we found no racial variations in the incidence of unilateral disease. CONCLUSIONS: Clinically, unilateral or asymmetric Graves orbitopathy is common and can lead to misdiagnosis or to unnecessary testing. Thyroid-related orbitopathy must be considered in the differential diagnosis for any case of asymmetric exophthalmos.

Cohort Studies↗
PubMed2006

Orbital morphology in exophthalmos and exorbitism.

BACKGROUND: Exophthalmos is a protrusion of the eyeball due to an increase in orbital contents in a normal bony orbit. Exorbitism is a protrusion of the eyeball due to a decrease in capacity of the orbital container, with a normal orbital content volume such as seen in a congenital form termed nonsyndromic exorbitism. High myopia can enhance proptosis. The purpose of this study was to provide values for orbital measurements from computed tomography and to suggest computed tomography criteria for nonsyndromic exorbitism. METHODS: Seventy-three computed tomography scans were collected (57 of Graves' ophthalmopathy and 16 of nonsyndromic exorbitism). Thirty-two scans from nonproptotic patients constituted a control series. Nine measurements and two indexes, performed on a reference axial computed tomography slice transecting the neuro-ocular plane, were obtained from each scan. RESULTS: The angle between the sagittal axis and the lateral orbital wall, as well as the width of the ethmoid bone (midinterorbital distance), was found to be more open in the nonsyndromic exorbitism population. A lateral orbital wall angle greater than or equal to 42 degrees and a midinterorbital distance greater than 30 mm were chosen as cutpoints. The association of these two criteria allowed the authors to obtain a sensitivity of 62 percent, a specificity of 78 percent, a positive predictive value of 80 percent, and a negative predictive value of 86 percent for nonsyndromic exorbitism. CONCLUSIONS: The different mechanisms of globe protrusion have to be taken into account before an orbital expansion/decompression procedure is planned. Only a preoperative morphological analysis of the orbital shape permits a precise analysis of the relative position of the ocular globe and orbital structures, in addition to clinical examination.

Adult↗
PubMed1981

Thyrotoxicosis and a thyrotropin-secreting pituitary tumor causing unilateral exophthalmos.

Hyperthyroidism due to a TSH-secreting pituitary tumor has been noted by a number of investigators. We describe a unique case in which a 17-yr-old female presented with clinical hyperthyroidism, a goiter, and unilateral exophthalmos. Serum T4, free T4, and T3 (RIA) were consistently elevated along with elevated TSH levels (range, 10-100 microunits/ml). Skull x-rays and computed tomography scan revealed a tumor invading the right orbit. Other pituitary function studies were normal and LATs was undetectable. Surgery performed resulted in 70% removal of the pituitary tumor and confirmed the presence of tumor infiltration into the right orbit. TRH tests done pre- and postoperatively (patient still clinically hyperthyroid with elevated T4 and TSH levels) showed TSH and PRL responsiveness. Electron microscopy of the tumor demonstrated features typical of pituitary thyrotrophs. Monolayer cultures of pituitary cells released TSH over time into the media but did not respond to TRH stimulation. Pituitary adenoma tissue content of immunoreactive TSH was 65 microunits/g wet tissue and demonstrated immunosimilarity with human standard. We conclude that the patient had a TSH-secreting pituitary tumor responsive to TRH stimulation.

Adenoma, Chromophobe↗
PubMed2005

[Tear film analysis and its relation with palpebral fissure height and exophthalmos in Graves' ophthalmopathy].

PURPOSE: To evaluate tear film quality by rose bengal staining and its stability by breakup time, relating with palpebral fissure height and exophthalmos in patients with Graves' ophthalmopathy. METHODS: We studied 54 eyes of 27 patients with Graves' ophthalmopathy, either in the inflammatory or in the chronic phase of the disease. The evaluation consisted of tear film qualitative analysis by rose bengal staining using the van Bijsterveld grading scale, tear film stability analysis by breakup time, measurement of palpebral fissure height and exophthalmometry. Qui square test was used to perform statistical analysis. RESULTS: Among 27 studied patients, 77.8% were females and 22.2% males. Mean age was 44.26 years (SD 12.67). Mean disease time was 5.85 years (SD 4.47) and mean ophthalmopathy time was 5.81 years (SD 5.37). Among 54 studied eyes, 37% had positive test by van Bijsterveld's grading scale, 33.3% tear film breakup time lower than 5 seconds, 57.4% palpebral fissure height greater than 11 mm and 55.6% exophthalmometry greater than 19 mm. When relating tear film breakup time lower than 5 seconds with palpebral fissure height greater than 11 mm we found an odds ratio of 11.2 (p=0.0008). The remaining relationships did not show statistical significance. CONCLUSIONS: Dry eye diagnosed by rose bengal staining and tear film breakup time occurs frequently in Graves' ophthalmopathy. Palpebral fissure height correlates with tear film breakup time in Graves' ophthalmopathy. Its increase may lead to tear film instability.

Adult↗
PubMed1975

Structure of an exophthalmos-producing factor derived from thyrotropin by partial pepsin digestion.

Previously reported experiments (Winand, R.J., and Kohn, L.D. (1970) J. Biol. Chem. 245, 967-975; Kohn, L.D., and Winand, R.J. (1971) J. Biol. Chem 246, 6570-6575) have demonstrated that partial pepsin digestion of bovine thyrotropin preparation yields a fragment of the thyrotropin molecule which is exophthalmogenic but has negligible or no thyroid-stimulating activity. In the present report this exophthalmogenic derivative of the thyrotropin molecule is shown to contain two major polypeptide components with approximate molecular weights of 14,000 and 6,000. Amino acid analyses, carbohydrate analyses, and tryptic digestion experiments indicate that this exophthalmogenic factor is composed of an intact or nearly intact beta subunit of thyrotropin and an NH2-terminal fragment of the alpha subunit of thyrotropin. Neither polypeptide component of the exophthalmogenic factor has the in vivo exophthalmogenic activity of the intact structure. In vitro the intact exophthalmogenic derivative of the thyrotropin molecule can bind to the thyrotropin receptor on thyroid membranes less efficiently than thyrotropin but significantly better than either its own polypeptide components or the alpha or beta subunits of thyrotropin. The exophthalmogenic factor and its parent thyrotropin molecule can stimulate adenylate cyclase activity in retro-orbital tissue membranes from guinea pigs, a mammalian model of exophthalmos; its polypeptide components have little or no such activity.

Adenylyl Cyclases↗
PubMed1990

Radiotherapy in emergency treatment of malignant exophthalmos.

18 patients with severe Graves' ophthalmopathy were treated with retrobulbar irradiation. 14/18 (78%) had at least a satisfactory immediate result to the radiotherapy, and every fourth patient had a good late result with normal eyes. A total depth dose of about 4000 cGy, with about 350 cGy daily fraction dose, and a total depth dose of about 2000 cGy, with about 200 cGy daily fraction dose, gave equal immediate and late results according to the exophthalmos. One patient lost her vision and is discussed in detail in the light of the literature.

Adult↗
PubMed1983

[Euthyroid exophthalmos: evaluation of 6 patients].

To determine if "euthyroid Graves' exophthalmopathy" is a separate autoimmune disorder, we have examined 6 clinically euthyroid male patients (aged 30-50 yrs) who have never had thyrotoxicosis, with recently developed bilateral exophthalmos (classes 2-4 in the A.T.A. classification). We have evaluated T4 and T3 levels, FTI, TSH response to TRH (200 micrograms i.v.), antithyroglobulin and antimicrosomal antibodies (Tgab and Mab) titres and thyroid stimulating antibody (TSab) activity of patients' IgG. In 5 patients we performed also orbital computerized axial tomography (CAT). The T4 and T3 levels ranged 5.6-7.9 micrograms/d1 and 1.2-2.1 ng/ml respectively; FTI ranged 7-9.7 U. 5 patients showed an impaired TSH response to TRH; only one had a normal TSH increase. The Tgab and Mab titre was undetectable in 5 patients; only one showed high autoantibody titre and developed a clear hypothyroidism 8 months later. TSab activity was detectable in the overall group. The CAT revealed a consistent infiltrative involvement of extraocular muscles in 4 patients; the 5th showed evidence of increased density of retroorbital fat, without any muscle involvement. In conclusion, the presence of TSab activity (6/6), the impaired TRH test (5/6) and the infiltrative retroorbital muscle involvement (4/5) seem to suggest an autoimmune thyroid disease even if apparently normal iodothyronine levels were found.

Antibodies↗
PubMed1981

Plasma exchange in conjunction with immunosuppressive drug therapy in the treatment of endocrine exophthalmos.

We describe three patients who developed severe endocrine ophthalmopathy at 3, 12 and 18 months after treatment with iodine-131. In two cases with congestive ophthalmopathy steroids alone and plasma exchange alone failed to produce a satisfactory response, but the combination of plasma exchange with steroids or steroids plus cyclophosphamide was followed by resolution of papilloedema and return of visual acuity to normal as well as definite improvement in the other signs of ophthalmopathy. In the remaining case, which did not have papilloedema the signs of ophthalmopathy were progressive despite treatment with steroids. Combined treatment with plasma exchange, steroids and cyclophosphamide resulted in improvement in visual acuity and resolution of periorbital oedema and diplopia. Plasma exchange may be a useful adjunct to immunosuppressive therapy in the treatment of acute progressive endocrine exophthalmos.

Aged↗
PubMed1993

Rhipicephalus exophthalmos sp. nov., a new tick species from southern Africa, and redescription of Rhipicephalus oculatus Neumann, 1901, with which it has hitherto been confused (Acari: Ixodida: Ixodidae).

Rhipicephalus exophthalmos sp. nov., a species which has in the past been confused with Rhipicephalus oculatus Neumann, 1901, is described and illustrated from laboratory-reared specimens. Preferred hosts for the adults of this tick are various domestic and wild ungulates and hares. It is widely distributed in Namibia and in the southeastern Cape Province, South Africa, with only scattered records from elsewhere. The adults of R. oculatus are redescribed and illustrated, and the immature stages are described and illustrated for the first time. All stages feed virtually exclusively on leporids. It is common in parts of the eastern and southern Cape Province, with few records as yet from other parts of South Africa and Namibia.

Africa, Southern↗