scholarly journals Idiopathic Sclerosing Inflammation Presenting as Sinusitis

2012 ◽  
Vol 3 (2) ◽  
pp. ar.2012.3.0031 ◽  
Author(s):  
Henry P. Barham ◽  
Megan K. Dishop ◽  
Jeremy D. Prager

Idiopathic sclerosing orbital inflammation is a rare finding that is poorly delineated, immune mediated, and causes severe symptoms and disability. It has been described affecting the orbit in addition to other sites within the head and neck, but has rarely been described presenting as sinusitis. A case report and literature review were performed. A 14-year-old girl with right-sided face and eye pain and pressure for >1 month presented 3 days after endoscopic sinus surgery for presumed acute sinusitis. She subsequently developed ipsilateral vision loss and hypesthesia of the infraorbital nerve. MRI revealed a mildly enhancing soft tissue intensity lesion extending from the right maxillary sinus into the pterygopalatine fossa and orbital apex through the inferior orbital fissure. Biopsy specimens of the lesion were consistent with a sclerosing inflammatory lesion. High-dose steroids led to rapid improvement in vision and pain; however, the patient was unable to tolerate steroid weaning because of recurrence of eye pain and headache. Repeat imaging showed progression of the lesion. Rheumatology was consulted and the patient's steroid therapy was altered and her medications were expanded to include azathioprine. The patient's symptoms improved and subsequent imaging showed a reduction in the size and extent of the lesion. Idiopathic sclerosing inflammation is characterized by primary, chronic, and immunologically mediated fibrosis. Patients typically have a poor response to corticosteroid treatment or radiotherapy. Immunosuppressive therapy in addition to corticosteroids is the recommended treatment.

2021 ◽  
pp. 3-6
Author(s):  
Jiraporn Jitprapaikulsan ◽  
M. Tariq Bhatti ◽  
Eric R. Eggenberger ◽  
Marie D. Acierno ◽  
John J. Chen

A 51-year-old White woman sought care for vision loss 1 week after a nonspecific upper respiratory tract infection. She reported pain in both eyes exacerbated by eye movement, which lasted for several days, followed by bilateral vision loss to the level of counting fingers–only vision. Optic neuritis was diagnosed, and she was treated with 1 g intravenous methylprednisolone for 3 days. Her vision improved substantially, and the pain resolved during the corticosteroid treatment. However, 1 week later, she woke up with right eye pain and vision loss. She was again treated with 5 days of intravenous methylprednisolone, with visual improvement nearly back to baseline. Two weeks later, she had recurrence of painful vision loss in both eyes. A diagnosis of chronic relapsing inflammatory optic neuropathy was made. Tests for serum angiotensin-converting enzyme, antineutrophil cytoplasmic antibody, antinuclear antibody, Lyme disease, syphilis, tuberculosis, and aquaporin-4-immunoglobulin G antibodies were negative. Serum was definitively positive for myelin oligodendrocyte glycoprotein-immunoglobulin G antibodies at a titer of 1:1,000. Myelin oligodendrocyte glycoprotein-immunoglobulin G–associated recurrent optic neuritis was diagnosed. After her diagnosis of recurrent corticosteroid-dependent optic neuritis associated with myelin oligodendrocyte glycoprotein-immunoglobulin G positivity, the patient was treated with 5 days of intravenous methylprednisolone. The eye pain resolved, and her vision returned to normal. At follow-up evaluation, the patient’s visual acuity, color vision, and visual fields were normal in both eyes, but there was mild bilateral optic disc pallor. She has not had recurrent demyelinating episodes while on chronic immunotherapy. Optic neuritis is an inflammatory demyelination of the optic nerve manifesting as acute to subacute vision loss, classically associated with pain with eye movement. The long-term prevention and prognosis depend on the cause of the optic neuritis.


2014 ◽  
Vol 128 (11) ◽  
pp. 1018-1021 ◽  
Author(s):  
A Hariri ◽  
N Choudhury ◽  
H A Saleh

AbstractBackground:Scytalidium dimidiatum is a soil and plant pathogen that frequently affects fruit trees, but can also cause human infection. There are only two reported cases of invasive fungal sinusitis involving this rare micro-organism.Objective:This paper reports the first case of invasive fungal sinusitis caused by Scytalidium dimidiatum occurring in a young immunocompetent patient from a non-endemic region, and discusses potential sources of exposure and relevance of local factors.Method:Case report.Results:The patient was treated successfully with a combination of functional endoscopic sinus surgery, and antifungal and corticosteroid treatment.Conclusion:This paper describes the first reported case of invasive fungal sinusitis secondary to Scytalidium dimidiatum in a young immunocompetent patient from a non-endemic region. Importance is placed on following a systematic process of investigation and management, and adhering to well-defined basic surgical principles.


2021 ◽  
Vol 39 (15_suppl) ◽  
pp. 11527-11527
Author(s):  
Emanuela Palmerini ◽  
Catalina Marquez ◽  
Cristina Meazza ◽  
Angela Tamburini ◽  
Gianni Bisogno ◽  
...  

11527 Background: Overexpression of ABCB1/P-glycoprotein (Pgp) predicts poor outcome in retrospective osteosarcoma series.Two prospective trials with Pgp expression and post-induction histologic response as stratification factors were activated in Italy (ISG/OS-2) and Spain (GEIS-33). Methods: Patients ≤ 40 years with extremity non-metastatic high-grade osteosarcoma were eligible. Analysisi of Pgp expression from diagnostic biopsy was centralized. Preoperatively, all patients received methotrexate, adriamycin, cisplatinum (MAP). Surgery was performed at week 8. All patients received a dose of adriamycin following surgery. In case of Pgp overexpression (Pgp+), mifamurtide (2 mg/m2 twice/week for 3 months then weekly for 6 months) was added after surgery, with 4 consecutive cycles of ifosfamide 3 gr/m2/day, day 1-5 (HDIFO) in case of poor histologic response (necrosis < 90%) to MAP. Patients without overexpression of Pgp (Pgp-) received MAP postoperatively, regardless the pathological response. From March 2013, an amendment increased high dose methotrexate cumulative dose from 60 g/m2 (5 cycles) to 120 mg/m2 (10 cycles). The post-amendment regimen was adopted in the observational prospective study by GEIS. Here we present the merged analysis of ISG/OS-2 patients treated post-amendment and GEIS-33. Results: From March 2013 to April 2018, 274 patients were included. Median age was 14 years (range 4-38), male/female: 163/111; 90 were Pgp-, 164 were Pgp+, 20 not evaluable. With a median follow-up of 48 months (1.3-78.5 months), the 3-year EFS and OS were 71.9% (95%CI 66-76.9) and 88% (95%CI: 83.2-91.5) respectively, with no inferior survival for Pgp positive patients and improved survival for good responders (Table). Conclusions: In this prospective uncontrolled study with a risk-adapted strategy for non-metastatic osteosarcoma, survival is superior to that of all ISG/GEIS previous series. The 3-year EFS of 71.9% compares favorably with other reports. Pgp+ patients performed well in this study, in which mifamurtide and HDIFO were added after a poor response to MAP. Clinical trial information: NCT01459484; NCT04383288. [Table: see text]


2015 ◽  
Vol 9 (11) ◽  
pp. 1289-1293 ◽  
Author(s):  
Kavitha Saravu ◽  
Rajagopal Kadavigere ◽  
Ananthakrishna Barkur Shastry ◽  
Rohit Pai ◽  
Chiranjay Mukhopadhyay

Two distinct and potentially deceitful cases of neurologic melioidosis are reported. Case 1: A 39-year-old alcoholic and uncontrolled diabetic male presented with cough, fever, and left focal seizures with secondary generalization. An magnetic resonance imaging (MRI) brain scan revealed a small peripherally enhancing subdural collection along the interhemispheric fissure suggestive of minimal subdural empyema. Blood culture grew Burkholderia pseudomallei. Patient was diagnosed with disseminated bacteraemic melioidosis with subdural empyema. He was successfully treated with ceftazidime-cotrimoxazole-doxycycline. Case 2: A 45-year-old male presented with left lower limb weakness, difficulty in passing urine and stool, and back pain radiating to lower limbs. Neurological examination revealed flaccid left lower limb with absent deep tendon reflexes and plantar reflex. Spinal MRI showed T2 hyperintensity from D9 to L1 suggestive of demyelination. Patient was treated with high dose methylprednisolone. By day 3 of steroid treatment, lower limb weakness progressed. Subsequent MRI showed extensive cord hyperintensity on T2 weighted sequence extending from C5 to conus medullaris consistent with demyelination. Cerebrospinal fluid (CSF) culture grew B. pseudomallei, and the patient was given meropenem-cotrimoxazole. After three weeks of parenteral treatment, the lower limbs remained paralyzed. Patient was discharged on oral cotrimoxazole-doxycycline. Conclusions: Melioidosis should be considered as a differential in focal suppurative central nervous system (CNS) lesions, meningoencephalitis, or encephalomyelitis in endemic areas. CNS infections must be ruled out prior to steroid administration. The role of corticosteroids in demyelinating CNS melioidosis has been refuted. This is a rare documentation of effect of unintentional corticosteroid treatment in melioidosis.


1996 ◽  
Vol 10 (1) ◽  
pp. 11-16 ◽  
Author(s):  
Ramzi T. Younis ◽  
Rande H. Lazar

Subperiosteal abscess is a serious orbital complication of sinusitis that occurs predominantly in children. Infection may spread to the orbital site through a preexisting defect in the lamina papyracea or by means of a thrombophlebitic process. Although the traditional approach to treating a subperiosteal abscess has been drainage through an external incision, we treated this serious complication with functional endonasal sinus surgery. Ten pediatric patients with clinical and CT evidence of orbital subperiosteal abscess underwent endoscopic surgery at LeBonheur Children's Hospital in Memphis, Tenn., between July 1990 and June 1992. The four girls and six boys were between 3 and 12 years of age at the time of surgery. Despite 2 to 4 days of intravenous antibiotic therapy, their signs and symptoms progressed, and axial and coronal CT scans demonstrated orbital subperiosteal abscess. Seven patients had bilateral ethmoid and maxillary sinusitis, and the remaining three patients had unilateral ethmoiditis. Ophthalmologic assessments were performed for all patients immediately after hospital admission. The patients were started on regimens of high-dose intravenous cefuroxime sodium to subdue infections and pediatric decongestant nasal spray to facilitate drainage. All 10 patients with the confirmed diagnosis of subperiosteal abscess were treated with endoscopic surgery to drain the site of infection. All 10 patients improved significantly 12 to 24 hours after surgery and were discharged from the hospital to their homes 48 to 72 hours after surgery. Patients were continued on a regimen of broad-spectrum oral antibiotics and beclomethasone nasal spray for 2 weeks beyond the final examination. Follow-up endoscopic examinations were performed under general anesthesia in the operating room 2 to 3 weeks after surgery. No complications or adverse sequelae occurred. Our study appears to confirm that endoscopic drainage of subperiosteal abscess offers the patient an alternative to incisional surgery. The endoscopic approach prevents an external scar without increasing morbidity or length of hospitalization.


Author(s):  
N. Vegas-Revenga ◽  
V. Calvo-Río ◽  
I. González-Mazón ◽  
L. Sánchez-Bilbao ◽  
E. Beltrán ◽  
...  

2018 ◽  
Vol 8 (1) ◽  
pp. 7-12
Author(s):  
Rupesh Gautam ◽  
Deepak Adhikari ◽  
Matrika Dhital ◽  
Sudip Thakur ◽  
Bhawana Adhikari

Introduction: The variation in the course of infraorbital canal and protrusion of the infraorbital nerve through it to the maxillary sinus may lead to its accidental injury during reconstructive or endoscopic sinus surgery. Preoperative identification of this variant will prevent unintended injuries. Methods: A retrospective study of 307 patients who underwent CT scan study of the paranasal sinuses at Chitwan Medical College, Nepal was conducted. The protrusion of infraorbital nerve to the maxillary sinus was identified and the length of the bony septum along with the infraorbital nerve was measured. It was further classified as Class I to III according to the length of the septum.Results: The prevalence of protrusion of inferior orbital nerve in our study was 11.40 % and bilateral protrusion was 5.8 %.  The median length of the protruding component along with the septum was 4.9 mm. Conclusion: Preoperative identification of the normal protrusion of infraorbital nerve to the maxillary sinus will prevent accidental injuries during sinus surgery. CT scan of the paranasal sinus would be the modality of choice for identification of this variant. 


2020 ◽  
Vol 19 (2) ◽  
pp. E196-E200 ◽  
Author(s):  
Ritam Ghosh ◽  
Fadi Al Saiegh ◽  
Aria Mahtabfar ◽  
Nikolaos Mouchtouris ◽  
Omaditya Khanna ◽  
...  

Abstract BACKGROUND AND IMPORTANCE Carotid-cavernous fistulas (CCF) are pathological connections between carotid artery branches and the cavernous sinus. They can lead to a variety of symptoms, such as chemosis and double vision, or more insidious events, such as vision loss and intracranial hemorrhage. Although these patients are often treated by endovascular means, we describe a case in which the patient's CCF was not able to be accessed by usual methods and required an open surgical approach. CLINICAL PRESENTATION The patient had progressive chemosis, double vision, and periorbital pain. Angiogram showed an indirect type D CCF with cortical venous drainage with a large sylvian vein that was directly draining the fistula. The patient did not have a dilated superior ophthalmic vein, and the petrosal sinuses could not be catheterized. Therefore, because of the patient's increased risk for intracranial hemorrhage, she was taken to the operating room for an image guided burr hole for direct catheterization of the sylvian vein. From this point, the fistulous point could be catheterized, and the CCF was embolized using onyx. Follow-up angiogram showed complete occlusion. CONCLUSION This is the first report in literature of an indirect CCF being treated through a transsylvian approach with onyx. This combined open-surgical-and-endovascular approach was necessary to get full resolution of the lesion, and patient had rapid improvement of symptoms.


2019 ◽  
Vol 10 (2) ◽  
pp. 227-234 ◽  
Author(s):  
Magdalene Yin Lin Ting ◽  
Meghan Shan ◽  
Oliver Gantz ◽  
Sandy Zhang-Nunes ◽  
Bozena Wrobel

Mucoceles of the paranasal sinus commonly involve the frontal sinuses, the ethmoid sinuses, and rarely the maxillary or sphenoid sinuses. They often present with sinus pain or pressure, but rarely can present with more severe symptoms such as changes in mental status or vision due to expansion and invasion through the skull base or orbit. A 62-year-old male presented with optic neuropathy, a relative afferent pupillary defect with proptosis and lateral gaze palsy of the left eye. The patient was found to have a large mucocele extending from the left posterior ethmoid sinus into the left orbital apex. Urgent endoscopic sinus surgery was performed jointly between Oculoplastics and Otolaryngology. Post-operatively, the patient had improvement in diplopia, extraocular motion, and proptosis with stable vision. This case demonstrates the importance of early identification and intervention in a rare presentation of a sinus mucocele to prevent serious complications such as vision loss.


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