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A case of a slipped medial rectus muscle after strabismus surgery.

BACKGROUND: A slipped muscle after strabismus surgery is a well known complication, but only a few cases have been reported in the medical literature. Here, we report a case of a slipped medial rectus muscle that occurred after strabismus surgery for esotropia. CASE REPORT: The patient was a 10 year old male with an acquired esotropia of 20 PD at distance. For treatment, 3.5 mm bilateral medial rectus recessions was performed. There were no complications during the procedure. On postoperative day 8, the eye position was orthotropic in primary position, but a right exotropia was observed in left gaze. The boy was frequently observed rubbing his right eye after surgery. The right exotropia increased to 30 PD 2 months after surgery. Limited adduction of the right eye, a large widening of the lid fissure on adduction, and mild exophthalmos were observed. Based on the above findings, a slipped medial rectus was suspected, and a second surgery, advancement of the right medial rectus muscle was performed 2.5 months after the initial procedure. After this second procedure, the eyes became orthotropic, and the adduction disorder, large widening the lid fissure, and mild exophthalmos resolved. CONCLUSION: The slipped muscle may have been caused by insufficient suture and/or excessive rubbing of the eye by the patient. When divergent strabismus is observed after recession of the medial rectus muscle, a slipped muscle should be considered as a differential diagnosis.

Child↗

Pyocele of the middle turbinate: a case report.

Mucoceles are uncommon and the majority of cases occur in the frontoethmoidal complex. The accumulation of mucous secretion and eventual secondary infection may result in the formation of a pyocele of the middle turbinate, which is exceptionally rare. This case report presents a patient with a large pyocele of the middle turbinate invading the orbit. A 48-year-old woman presented with a three-week history of nasal obstruction, slight rhinorrhea, and headache. There was an external swelling of the nasal pyramid just near the medial canthus and right exophthalmos with bulging of the medial canthus of the ipsilateral eye. A computed tomography scan revealed a rounded, expansive lesion of soft tissue density. There was a thin compact bone at the margin. Under general anesthesia, resection of the lateral and inferior walls of the middle turbinate was performed. The large cystic mass was filled with pus. Exophthalmos and bulging of the medial canthus immediately disappeared. Histologic examination showed the mucocele wall with a ciliary respiratory epithelium.

Diagnosis, Differential↗

[A case of orbital hematic cyst].

A case of orbital hematic cyst is reported. The patient was a 3-year-old boy in whom swelling of the left upper lid and left exophthalmos appeared after blunt trauma in the region of the left eye. A hematoma was thought to be present from upper lid to the orbit. The hematoma was absorbed but the swelling of the eyelid and exophthalmos increased though more than seven days elapsed after the trauma. The existence of the cyst located from the upper lid to the orbital deep portion and collection of blood were confirmed during operation. Histopathologically the cyst consisted of thickened fibrogranulomatous tissue and the inner wall lacked endothelial or epithelial lining. The blood collected in the cyst hardly coagulated and was examined hematologically. Extreme delay of activated partial thromboplastin-time and prothrombin time, extreme decrease of fibrinogen and abnormal increase of fibrin degradation products were recognized. This indicated that the blood in the cyst had been in a localized accelerated fibrinolytic state. The results support the hypothesis of the mechanism leading to the enlargement of hematic cyst proposed by Pearson et al.

Child, Preschool↗

[Radiation therapy for thyroid-associated ophthalmopathy].

OBJECTIVE: To observe indication and effectiveness of radiation therapy (RT) in the treatment of the patients with thyroid-associated ophthalmopathy (TAO). METHODS: 23 patients of TAO who received RT in Sichuan University were collected from 1992 to 2004. Among those patients, 9 cases of infiltrative exophthalmos and 14 cases of compressive optic neuropathy were ineffectively with glucocorticoid treatment or could not treated with glucocorticoid, or could not perform orbital decompression due to severe diabetic mellitus or hypertension, or feared to receive the operation, all of patients were active ophthalmopathy and with short duration. Outer orbital radiation was applied using linear accelerator with Donaldson's method, radiation treatment fields was 4 cm x 5 cm, exposure energy was 2 GY fractions with total of 20 GY. In 11 cases with severe inflammation prednisone was administered during radiotherapy. Photos and CT scan were taken for each patient before and after RT. RESULTS: Visual acuity (VA) of the patients was improved from before RT 0.04 - 0.2 to after RT 0.1 - 0.8 in 14 cases of compressive optic neuropathy. Extraocular muscle of patients decreased in size confirmed by CT scan. VA improvement was correlated with the degree of extraocular muscle decreased in size. Eyelid and conjunctive swelling, eyelid incompletely closure, exposure keratitis, limitation of motion and proptosis were improved after RT in 9 patients with infiltrative exophthalmos. Following up the patients for 1 - 3 years, it was found that VA decreased in 3 cases and inflammation recurred in 4 cases, eyelids could not closed in 2 cases after RT. CONCLUSIONS: RT could be used in severe, active cases of TAO. If there is severe inflammation, steroids could be combined with RT therapy.

Adult↗

Cranioorbital fibrous dysplasia: with emphasis on visual impairment and current surgical management.

Fibrous dysplasia is a benign but slowly progressive disorder of bone in which normal cancellous bone is replaced by immature woven bone and fibrous tissue. Significant deformity and both acute and chronic visual impairment can result. A contemporary understanding of fibrous dysplasia, emphasizing the origins of visual impairment, indications for decompressive surgery, and the techniques for correction of the cosmetic deformity are presented. In their experience and review of the literature, the authors found the most frequent clinical presentations to be exophthalmos, displacement of the globe, abnormalities of extraocular motility, cosmetic deformity, and visual impairment. Although traditionally the cause of visual impairment has been ascribed to impingement of the optic canal on the optic nerve, the authors' experience is that the most common cause of visual loss is cystic degeneration of the tumor, particularly with those involving the anterior clinoid process. Exophthalmos and optic canal stenosis are less common causes of visual impairment. Indications for surgical intervention include acute and/or serially radiographically documented and relentless visual impairment and significant cosmetic deformity. Individualized management strategies are also discussed.

Adolescent↗

[Clinical analysis of 11 cases of orbital Langerhans cell histiocytosis].

OBJECTIVE: To study the clinical and imaging features, the diagnosis and management of orbital Langerhans cell histiocytosis. METHODS: The symptoms, signs, imaging features, treatment and follow-up results of 11 cases of orbital Langerhans cell histiocytosis were reviewed and analyzed, including 7 cases of eosinophilic granuloma and 4 cases of Hand-Schueller-Christian disease. RESULTS: Eosinophilic granuloma occurred primarily in children and teenagers and appeared as orbital lesions only. Hand-Schueller-Christian disease occurred in children under 5 years old, with multiple lesions of histiocytosis. Typical cases showed the Hand-Schueller-Christian triad: exophthalmos, diabetes insipidus and bony skull defects. Patients usually complained of exophthalmos and orbital masses. B scan and color Doppler showed soft tissue masses in the orbit. CT showed soft tissue masses and bony skull defects. MRI showed orbital tumor and intracranial involvement. CONCLUSIONS: Characteristic imaging features are helpful for the diagnosis of orbital Langerhans cell histiocytosis. The treatment includes surgical excision, local radiotherapy or chemotherapy. The prognosis is related to the types of lesion, eosinophilic granuloma shows the best prognosis and infantile Hand-Schueller-Christian disease shows the worst prognosis.

Adolescent↗

[Tonometry in the diagnosis of subclinical ophthalmopathy in patients with Basedow's disease].

Ophthalmopathy in addition to hyperthyreosis with goitre and dermopathy is characteristic of Basedow's exophthalmos is frequently associated with elevated intraocular pressure (IOP) on upgaze. We wanted to examine whether these changes of IOP exist in patients with Basedow's disease without clinical manifestations of ophthalmopathy. We measured IOP with Goldmann's applanation tonometry in two positions: the primary position, when the patient looks straight ahead and then on upgaze. Forty-six patients with Basedow's disease were examined. The minority [5 (11%)] of the 46 patients had exophthalmos and 22 (47%) of them had abnormal IOP (delta IOP greater than or equal to 3 mmHg). The average interval between the onset of Basedow's disease and this study was 12.8 +/- 7.4 years for those patients who had exaggerated positional changes in IOP, as compared with 5.8 +/- 3.3 years for those with normal IOP, P less than 0.01. We conclude that Basedow's ophthalmopathy is more common than is recognized clinically and that ophthalmopathy is more frequent in patients suffering of Basedow's disease for a longer period. Measuring IOP in two positions is a very simple method which could help us in early diagnosis of ophthalmopathy in patients with Basedow's disease.

Adult↗

[Adenoid cystic carcinoma of the lacrimal gland: report of a case].

Adenoid cystic carcinoma of the lacrimal gland is rarely reported in Japan compared with other countries in Europe and the U.S.A. Here is a report of this rare case. A 26-year-old female had complained of left visual disturbance and left exophthalmos with orbital pain. CT scan and MRI revealed a lacrimal gland tumor in that eye. The tumor was removed via a left frontal craniotomy, and diagnosed as adenoid cystic carcinoma. After operation, she was locally irradiated with a total dose of 5,000 rads using 15 MeV Lineac source. Chemotherapy was started with 5-FU after radiation therapy. An abnormal uptake was observed in the sternum in the bone scintigraphy performed after operation, suggesting metastasis. Visual disturbance improved and exophthalmos and orbital pain disappeared. At the last follow-up at 8 months after operation, she had no signs and symptoms of recurrence or metastasis. Adenoid cystic carcinoma is reported to be the most frequent among the malignant tumors of the lacrimal gland in the Western countries. Prognosis is generally unfavorable with the mortality of 45% to 70%. The 5-year and 10-year survival rate are 45% and 25%, respectively. The mean post-operative survival time is 4.5 years. Since the tumor grows infiltratively into the surrounding tissues, radical excision with total exenteration of the orbit is the treatment of choice. However, it is a difficult decision whether or not to remove the eye with the intact visual function. As for operation, an approach from above through the frontal craniotomy provides a wider operative field and an easier access to the tumor than the lateral approach employed by ophthalmologists.

Adult↗

[Carotid-cavernous fistula successfully treated with embolization and radiation therapy: report of three cases].

Three cases of mixed internal and external carotid-cavernous fistula (CCF) were successfully treated with embolization of feeders from the external carotid artery (ECA) and focal irradiation to the cavernous sinus (CS). Cases 1 and 2 were females, 63 and 69 years old respectively, both with spontaneous left CCF. Case 3 was a 55 year old male with posttraumatic left CCF. Symptoms of case 1 were double vision, left chemosis and exophthalmos; those of case 2 were double vision, left retroobital pain, left forehead dysesthesia and blurred vision; and case 3 complained of double vision, left chemosis, left exophthalmos and pulsatile tinnitus. In all three cases, angiography disclosed left CCF fed by ipsilateral dural branches from the internal maxillary artery (IMA) and the internal carotid artery (ICA). In case 1, small branches from the ascending pharyngeal artery also fed the CS. In cases 2 and 3, feeders from the ECA were arising only from branches of the IMA. In case 3, hypertrophy of the meningohypophyseal trunk was visible. In cases 1 and 2, although the CS was opacified, feeders from the ICA were not clearly visible. Embolizations of branches of the IMA were performed in all cases using Ivalon under selective catheterization. In case 1, symptoms partially improved, but in cases 2 and 3, visual symptoms were transiently aggravated. Focal irradiation to the CS was done with total doses of 30, 30 and 40 Gy each for cases 1, 2, and 3 respectively. In case 1, clinical symptoms gradually improved about one third way through irradiation.(ABSTRACT TRUNCATED AT 250 WORDS)

Aged↗

[The treatment of Graves' disease with antithyroid drugs and small doses of radioiodine (author's transl)].

Three hundred and thirty Graves' disease patients (258 F. 72 M. ; mean age = 50 +/- 07 years) were treated by 131I from 1961 to 1979 ; 57 % had a goiter and 32 % had exophthalmos. One or several doses of radioiodine were given to each patient without dosimetry. The mean total dose (+/- SEM) was = 8,51 +/- 0,44 mCi and the mean for the first dose 4,34 +/- 4 ans 54,8 % of the patients received only one dose but 10,3 % of them had more than 3 doses. Data obtained by actuarial methods showed 66 % of the cases to be in remission in less than 2 years ; the frequency of hypothyroidism increased steadily by 3 % every year to give 15 % at 5 years and 30 % at 10 years. Remission was slower to occur in goitrous subjects and hypothyroidism was less frequent in this group despite doses significantly higher of radioiodine (p less than 0,001). Recovery was also slower among exophthalmos patients. Finally, the mean 3-hour radioiodine uptake and plasma T3 level were significantly lower six months after the first therapeutic dose among those who were cured of thier hyperthyroidism within two years.

Antithyroid Agents↗

[Case of histiocytosis X with a large intracranial mass].

The authors experienced a case of histiocytosis X with a large intracranial mass resulting in a convulsive seizure. The patient showed left exophthalmos and a skin rash one year and two months after birth. Histiocytosis X was diagnosed from a skin biopsy, and predonine, endoxan and vincristine were administered. The rash disappeared, but the exophthalmos remained. At the age of two years and nine months, punched-out lesions appeared in the skull and 4,000 rads of radiation was applied. Thereafter, the exopthalmos persisted but there was no particular problem in the course. However, a convulsive seizure with fever suddenly appeared at nine years and ten months of age and the patient was hospitalized. At the time of admission, the general condition was good and there were no abnormalities in neurological tests. In neuroradiological examinations, a calcified and poorly vascularized mass 8 cm in maximum diameter was found to occupy the left middle cranial fossa. Chondrosarcoma was strongly suspected from these findings, but there was also symmetrical thickening of bone cortex in the peripheries of the long bones of the extremeties which appeared to be the recovery process from bone destruction caused by histiocytosis X. Therefore, the formation of an intracranial mass by histiocytosis X was diagnosed and surgery was performed. When left osteoplastic fronto-temporal craniotomy was performed, the mass was found to be raising the temporal lobe and it could be easily separated from the surrounding tissue. However, these was firm adherence to dura mater of the middle cranial fossa (especially that of the superior orbital fissure). Histologically, there were many cells with small nuclei, no polymorphism, abundant and clear cytoplasm which were darkly stained and slightly atypic. These findings matched those for histiocytosis X. Cases of histiocytosis X rarely show symptoms of the central nervous system or infiltration of the central nervous system. Only 31 such cases were seen in the literature investigated by the authors. Neurological symptoms include pyramidal symptoms such as hemiparesis and impairment of the cranial nerves, particularly paresis of the optic, trigeminal, facial and acoustic nerves. Convulsive seizures were seen in only five cases including the one reported here. It is also rare for intracranial masses to be formed in cases of histiocytosis X and only six cases, including the authors', have been found with masses of a maximum diameter of more than 5 cm.

Brain Diseases↗

Thyroid acropachy.

Thyroid acropachy (TA) developed in a 48-year-old man at least six years prior to clinically evident and symptomatic hyperthyroidism. The TA prompted a clinical workup for acromegaly on two occasions, with negative results. It usually develops after diagnosis or treatment of hyperthyroidism and usually occurs in conjunction with exophthalmos. In our patient, TA preceded the diagnosis and treatment of hyperthyroidism and was not associated with exophthalmos.

Humans↗

Direct carotid-cavernous sinus fistulas.

1. Direct carotid-cavernous sinus fistulas usually result from multi-trauma or head injuries. These patients are more likely to be seen at trauma-based hospitals. 2. Ocular manifestations of direct carotid-cavernous sinus fistulas include exophthalmos, swollen eyelids, ocular pulsation, pulsating exophthalmos, conjunctival chemosis, arterialization of conjunctival vessels, exposure keratopathy, a bruit over the affected eye (heard by the patient as a swishing sound), diplopia, visual loss, and ophthalmoscopic abnormalities that include moderate dilation of retinal veins, optic disc swelling, and intraretinal hemorrhage. 3. The ultimate diagnostic test for direct carotid-cavernous sinus fistulas is cerebral arteriography of both the internal and external carotid arteries. 4. The optimal treatment of a direct carotid-cavernous sinus fistula is closure of the abnormal arteriovenous communication with preservation of internal carotid artery patency. Detachable, flow-guided balloons are presently used to close most direct carotid-cavernous sinus fistulas.

Cardiovascular Diseases↗

[Transarterial platinum coil embolization for direct carotid-cavernous fistula].

Two cases of direct carotid-cavernous fistula (CCF) were treated by transarterial platinum coil embolization (TACE) following unsuccessful transarterial balloon embolization (TABE). Case 1 was a 47-year-old man who complained of pulsatile left exophthalmos, chemosis and bruit. Left carotid angiograms showed a CCF with anterior, posterior and cortical venous drainage. Near total obliteration of the CCF was achieved by TABE, but it showed recurrence in the next morning. At this time, left carotid angiograms showed a CCF which drained only into the cortical veins via the enlarged sphenoparietal sinus. Because of high risk of intracranial hemorrhage, TACE was performed immediately. The result was successful. Case 2 was an 82-year-old woman who suffered from traumatic subarachnoid hemorrhage. First right carotid angiograms showed a small CCF which drained only into the inferior petrosal sinus. Right exophthalmos, swelling of the eyelids, severe eye pain and bruit appeared gradually. The second right CAG performed three months after the head trauma showed markedly dilated superior ophthalmic vein which was the new main draining root of the CCF. Because of progressive symptoms, TACE was performed immediately after the angiography, which proved successful. Direct CCFs must be treated aggressively because they don't cure by spontaneous obstruction of fistula. Although TABE is the first choice for direct CCF, complete occlusion of CCF in difficult in some cases. Those cases have; 1) small fistula of CCF for balloon insertion, 2) large fistula for occlusion by balloons, 3) not enough space for inserting a balloon after recurrence of CCF, and 4) sharp objects (bone fracture fragments, foreign objects) may puncture the balloon. If TABE couldn't provide successful treatments, TACE should be considered as an alternative treatment for direct CCF after angiography without delay because it is less complex compared with TABE.

Aged↗

Clinical experience of radiation therapy for Graves' ophthalmopathy.

The effect of radiation therapy for Graves' ophthalmopathy was evaluated. Ten patients with Graves' ophthalmopathy were treated with radiation therapy between 1992 and 1993 in Gunma University Hospital. All patients had a past history of hyperthyroidism and received 2,000 cGy to the retrobulbar tissues in 20 fractions. Nine of ten patients were treated with radiation therapy after the failure of corticosteroids. Six patients (60%) showed good or excellent responses. The exophthalmos type was more responsive to radiation therapy than the double vision type in this series. Two of five patients with the exophthalmos type demonstrated excellent responses, and their symptoms disappeared almost completely. The improvement of symptoms appeared within 3-6 months, and obvious clinical effects were demonstrated after 6 months of radiotherapy. Radiation therapy was well tolerated, and we have not observed any side effects of radiation therapy. In conclusion, radiation therapy is effective treatment for Graves' ophthalmopathy.

Adult↗

[Spontaneous carotid-cavernous fistula: correlations between clinical findings and venous drainage].

We reviewed retrospectively 6 cases of spontaneous carotid cavernous fistulas to discuss the differences in clinical findings depending on venous patterns. Orbit US was performed in all patients but one and all patients were examined with CT and intraarterial digital subtraction angiography (DSA). Orbit US, performed in 5 patients with ocular signs, showed dilation of the ophthalmic veins, in 3 cases bilaterally. CT was performed to demonstrate possible lesions of the orbit apex or skull base and showed dilation of the superior ophthalmic vein in 4 patients, bilateral in only one case. Moreover, CT detected enlargement of the cavernous sinuses in 4 cases (3 unilateral, one bilateral). Dynamic CT, performed in 2 patients, showed early opacification of both cavernous sinuses. DSA was carried out on the basis of clinical data and US and CT findings. It diagnosed carotid cavernous fistulas in all cases. The fistulas were bilateral in 3 subjects. In all the patients with exophthalmos, conjunctival chemosis and dilated episcleral veins, the superior ophthalmic vein was the main venous drainage from the cavernous sinus. Only 4 of these patients had abducent nerve palsy; bruit was present in 2 cases only. The ocular signs were contralateral to the fistula in one patient. In the patient presenting III, IV and VI nerve palsy, the venous drainage was direct from the cavernous sinus to pericarotid plexus, pterygoid plexus and inferior petrosal sinus; in this case there were no ocular signs. The clinical findings of spontaneous cavernous fistulas are caused by the arterial supply and, especially, by the venous drainage of the fistula; for the early diagnosis and treatment it is important to know that some patients do not exhibit the classic triad of symptoms, characterized by pulsating exophthalmos, bruit and conjunctival chemosis. Our experience has confirmed that spontaneous carotid cavernous fistulas may be characterized by atypical clinical findings, such as ocular signs contralateral to the fistula side or palsies of cranial nerves only.

Adult↗

Primary orbital Ewing sarcoma in a middle-aged woman.

A 43-year-old woman had unilateral exophthalmos caused by primary orbital Ewing sarcoma. Specialized immunohistochemical stains, primarily MIC-2 (CD99), aided in the diagnosis of Ewing sarcoma. Twenty-two months after radiotherapy and multiagent chemotherapy, the patient remained tumor free. To our knowledge, this is the first reported case of orbital Ewing sarcoma to present in an adult beyond the fourth decade of life.

12E7 Antigen↗