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• W2060874662 abstract "A 69-year-old female came to Oculoplastic Clinic, Farabi Eye Hospital, Tehran, Iran, with the complaint of new-onset proptosis and loss of vision in her left eye from 4 months ago. She had no history of any trauma. On examination, there was no visible skin scar of any previous surgery around the orbit. The right eye had best corrected visual acuity (BCVA) of 9/10, and slit-lamp examination of its anterior segment and funduscopy showed no pathology except mild cataract. In her left eye, she had BCVA of 2/10, mild conjunctival injection and chemosis, severe proptosis (10-mm difference in exophthalmometry; Fig. 1A), lateral gaze limitation, and a reactive pupil with 2+ relative afferent pupillary defect. The left eye had also mild cataract, and its funduscopy showed no significant abnormal finding. Radiologic evaluation for the eye was started with CT scan (Fig. 1B). CT scan revealed a well-defined, dumbbell-shaped cystic lesion—internal density similar to water, delineated by a perceptible hyperdense rim—in lateral wall of orbit, with destruction of the bone and extension into the orbit, compressing orbital contents and pushing the globe forward. For further evaluation of the lesion, magnetic resonance imaging (MRI) was performed (Fig. 1C, 1F). MRI showed a multiloculated cystic lesion with better depicting peripheral fibrous capsule and internal septations. The lesion had low signal intensity in T1-wighted imaging without enhancement and high signal intensity in T2-weighted images. The signal intensity of the lesion was consistent with an isolated multiloculated cystic lesion with content density similar to water or cerebrospinal fluid (CSF), with no internal enhancement, which could be a hydatid cyst, a dermoid cyst, an abscess, a mucocele, a hematocele, or an encephalocele. However, in T1-weighted images, hypersignal intensity is expected in dermoid cysts (because of their fat content) and hematocele (because of methemoglobin inside them). Moreover, abscess usually has a notable wall enhancement in contrast studies, whereas thin peripheral enhancement is sometimes evident in mucocele and hydatid cyst, mostly as a result of superinfection. In the case of encephalocele, a cranial defect, and hence a connection with intracranial extra-axial CSF space, could be depicted. With these findings, the most probable diagnosis was an intraosseous hydatid cyst. Considering its compressive effect, the lesion had to be excised as soon as possible. Surgical en bloc excision of the cyst is the mainstay of treatment. To avoid intraorbital dissemination of the cyst’s content, considering its location, we approached through an incision 1.5 cm posterior to lateral orbital rim (Fig. 2A). Although the cyst ruptured during last step, there was no spillage into orbital space and it could be totally removed (Fig. 2B). Pathology confirmed hydatid cyst (Fig. 3A, 3B). After surgery, albendazole was prescribed for the patient for 3 months. After 2 years of follow-up, no sign of recurrence has been detected in this patient.Fig. 3Histopathologic view of the patient’s lesion showing laminated hyalinized cystic wall of hydatid cyst (A) attached to some scolexes (B). Hematoxylin and eosin stains. Original magnification ×100 (A); ×400 (B).View Large Image Figure ViewerDownload (PPT) Brain imaging was performed for evaluation of any intracranial extension or dissemination, which was negative. To assess whether the patient had pulmonary hydatidosis, we performed a chest x-ray, which showed no finding. Also, abdominal ultrasonography was performed to evaluate the liver, which showed no abnormal finding. Echinococcosis is one of the most common parasitoses that affect humans in its endemic areas. When contaminated, it mostly affects liver, lung, and kidneys.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar, 2Aouchiche M. Benrabah R. Abanou A. et al.[Computed x-ray tomographic aspects of intra-orbital hydatid cyst. Apropos of 10 cases]. [Article in French].J Fr Ophtalmol. 1983; 6: 901-916PubMed Google Scholar Orbital involvement is rare, and only about 1% of hydatidoses occur in orbit.3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar, 4Danziger A. Price H. Computed tomographic findings in orbital echinococciasis.J Comp Assist Tomogr. 1980; 4: 128-129Crossref PubMed Scopus (9) Google Scholar It has been reported that hydatidosis accounts for only 1% of orbital tumours2Aouchiche M. Benrabah R. Abanou A. et al.[Computed x-ray tomographic aspects of intra-orbital hydatid cyst. Apropos of 10 cases]. [Article in French].J Fr Ophtalmol. 1983; 6: 901-916PubMed Google Scholar; but among orbital cystic lesions, it has been the second most frequent lesion (25.8%) after dermoid cysts (29.7%).5Günalp I. Gündüz K. Cystic lesions of the orbit.Int Ophthalmol. 1996-1997; 20: 273-277PubMed Google Scholar Although in endemic areas such as Spain, Lebanon, Iraq, and Turkey, the incidence rate of hydatid cyst has been reported to be 13.6%, 6%, 20%, and 6.6% among orbital tumours, respectively.6Kars Z. Kanzu T. Ozcan O. et al.Orbital echinococcosis, report of two cases studied by computerized tomography.J Clin Neuro-ophthalmol. 1982; 2: 197-199PubMed Google Scholar In North America, Echinococcus granulosus is rarely reported in Canada and Alaska, and a few human cases have also been reported in Arizona and New Mexico in sheep-raising areas. In the United States, most infections are diagnosed in immigrants from countries where cystic echinococcosis is endemic.7Centers for Disease Control and Prevention. Cystic echinococcosis. http://www.cdc.gov/parasites/echinococcosis/gen_info/ce-faqs.htmlGoogle Scholar Orbital hydatidosis mostly affects children and young adults. This occurs most frequently when individuals handle or contact infected dogs or other infected carnivores, or inadvertently ingest food or drink contaminated with fecal material that contains tapeworm eggs. In our case, we think that she has ingested vegetables possibly contaminated with fecal material of an infected dog. It is usually unilateral with a trend for left eye involvement,3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar and occurs with an insidious onset, causing slowly progressive proptosis, which mostly leads to visual loss and limited eye movements.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar, 3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar, 4Danziger A. Price H. Computed tomographic findings in orbital echinococciasis.J Comp Assist Tomogr. 1980; 4: 128-129Crossref PubMed Scopus (9) Google Scholar, 8Benazzou S. Arkha Y. Derraz S. et al.Orbital hydatid cyst: review of 10 cases.J Craniomaxillofac Surg. 2010; 38: 274-278Abstract Full Text Full Text PDF PubMed Scopus (24) Google Scholar There can be sudden symptoms, for example, complete loss of vision, proptosis, pain, among others, caused by spontaneous cyst rupture. These lesions are usually not associated with hydatid cysts of other organs3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar; however, accompanied involvement of liver, lung, brain, spleen, and thyroid has been reported.9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar Thus, chest x-ray and abdominal ultrasonography should be performed as a routine work-up for all patients.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar About 2% of hydatid cysts have been reported to be located in skeleton,9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar and there is small number of case reports on intraosseous hydatid cysts,10Erman T. Tuna M. Göçer I. et al.Intracranial intraosseous hydatid cyst. Case report and review of literature.Neurosurg Focus. 2001; 11 (ECP1)Google Scholar, 11Yazdani N. Basam A. Heidarali M. et al.Infratemporal hydatid cyst: a case presenting with blindness.J Laryngol Otol. 2010; 124: 456-459Crossref PubMed Scopus (10) Google Scholar, 12Pelegri C. Gaertner E. Bernard E. et al.Recurrence of femoral echinococcosis 5 years after a primary surgical procedure.Orthop Traumatol Surg Res. 2010; 96: 94-96Crossref PubMed Scopus (3) Google Scholar, 13Fyfe B. Amazon K. Poppiti Jr, R.J. et al.Intraosseous echinococcosis: a rare manifestation of echinococcal disease.South Med J. 1990; 83: 66-68Crossref PubMed Scopus (10) Google Scholar although periorbital bone involvement has not been reported. Eosinophilia can be seen in a few patients. Although serologic tests are generally positive in systemic hydatidosis, they are almost always negative in orbital hydatidosis, and thus are not reliable.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar, 9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar Imaging studies including CT scan and MRI are now standard diagnostic evaluation for these lesions.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar, 3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar On CT scan, they are shown as well-defined encapsulated cystic lesions with internal density of water or CSF, which can show increased peripheral density at its capsule area (a hyperdense rim); this can show rim enhancement with contrast.6Kars Z. Kanzu T. Ozcan O. et al.Orbital echinococcosis, report of two cases studied by computerized tomography.J Clin Neuro-ophthalmol. 1982; 2: 197-199PubMed Google Scholar, 9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar These lesions can result in bone destruction (mostly medial wall into nasal cavity).6Kars Z. Kanzu T. Ozcan O. et al.Orbital echinococcosis, report of two cases studied by computerized tomography.J Clin Neuro-ophthalmol. 1982; 2: 197-199PubMed Google Scholar, 14Murthy R. Honavar S.G. Vemuganti G.K. et al.Polycystic echinococcosis of the orbit.Am J Ophthalmol. 2005; 140: 561-563Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar On MRI, we can see the lesion as a well-defined cystic mass with low-intensity signal in T1-weighted imaging, which can show peripheral rim enhancement with contrast and high internal signal intensity, and a hyposignal peripheral rim (caused by fibrous component of the capsule) in T2-weighted images.9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar Surgical en bloc resection of the cyst is the mainstay of treatment and it can be curative if we are sure there has been no spillage of the contents intraoperatively3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar; however, it frequently has leakage through small punctures in the cyst wall, made during the procedure, which necessitates antihelmintic therapy, albendazole or mebendazole, postoperatively to reduce recurrence.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar, 3Limaiem F. Bellil S. Bellil K. et al.Primary orbital hydatid cyst in an elderly patient.Surg Infect (Larchmt). 2010; 11: 393-395Crossref PubMed Scopus (10) Google Scholar, 8Benazzou S. Arkha Y. Derraz S. et al.Orbital hydatid cyst: review of 10 cases.J Craniomaxillofac Surg. 2010; 38: 274-278Abstract Full Text Full Text PDF PubMed Scopus (24) Google Scholar, 9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar Also, preoperative medical therapy has been advised to sterilize the lesion before the procedure, causing reduced risk for complications such as anaphylaxis and recurrence.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar For intraosseous lesions, extensive bony curettage is suggested.12Pelegri C. Gaertner E. Bernard E. et al.Recurrence of femoral echinococcosis 5 years after a primary surgical procedure.Orthop Traumatol Surg Res. 2010; 96: 94-96Crossref PubMed Scopus (3) Google Scholar Irrigation of the affected area with hypertonic saline has also been advised, especially in the case of spillage.8Benazzou S. Arkha Y. Derraz S. et al.Orbital hydatid cyst: review of 10 cases.J Craniomaxillofac Surg. 2010; 38: 274-278Abstract Full Text Full Text PDF PubMed Scopus (24) Google Scholar, 9Turgut A. Turgut M. Kosar U. Hydatidosis of the orbit in Turkey: results from review of the literature 1963–2001.Int Ophthalmol. 2004; 25: 193-200Crossref PubMed Scopus (34) Google Scholar, 11Yazdani N. Basam A. Heidarali M. et al.Infratemporal hydatid cyst: a case presenting with blindness.J Laryngol Otol. 2010; 124: 456-459Crossref PubMed Scopus (10) Google Scholar One important key in managing these patients is to have regular follow-up visits to detect any recurrence, especially in adjacent organs.1Siddiqui M.A. Rizvi S.W. Rizvi S.A. et al.Atypical multifocal hydatid disease of cranial vault: simultaneous orbital and extradural meningeal involvement.Emerg Radiol. 2010; 17: 427-430Crossref PubMed Scopus (12) Google Scholar, 11Yazdani N. Basam A. Heidarali M. et al.Infratemporal hydatid cyst: a case presenting with blindness.J Laryngol Otol. 2010; 124: 456-459Crossref PubMed Scopus (10) Google Scholar, 12Pelegri C. Gaertner E. Bernard E. et al.Recurrence of femoral echinococcosis 5 years after a primary surgical procedure.Orthop Traumatol Surg Res. 2010; 96: 94-96Crossref PubMed Scopus (3) Google Scholar In conclusion, we should consider the hydatidosis as a cause of progressive unilateral proptosis and visual loss in its endemic areas, which can be diagnosed with the help of imaging studies. Immediate intervention should be performed because of its compressive effects, and its recurrence must always be kept in mind, which can result in poor visual outcomes." @default.
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• W2060874662 title "Intraosseous orbital hydatid cyst: report of a rare case" @default.
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