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"en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Preoperative MRI and intra-surgical imaging. (A and D) Axial and coronal T1-weighted sequences without gadolinium, respectively, showing an intraconal nodular mass (green dashed line). Note the easy identification of the optic nerve (red arrows). (B and E) Axial and coronal T1-weighted contrast-enhanced sequences, respectively, confirming homogeneous enhancement of the lesion. (C) Axial T2-weighted sequences showing clear medial displacement of the right optic nerve. (F) Image showing the position of the tumour (light green) from 7 o'clock to 10 o'clock following the "clock" model. (G) Image of the surgical incision. (H) Lateral orbitotomy performed with piezoelectric saw. (I) Opening of the periorbita and lateralisation of the external lateral muscle. No tumour infiltration of the muscle is observed. (J) Detailed image of the lesion with a whitish appearance and sparse arterial vascularisation. The eyeball can be seen in the anterior part. (K) Final repositioning of the zygomatic bone with titanium mini-plates. (L) Final image of the sutured incision with intradermal technique.</p>"
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"textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Immunoglobulin G4-related disease (IgG4-RD) is a fibro-inflammatory immune-mediated disorder characterised by lymphoplasmacytic infiltration (predominantly IgG4 plasma cells) of tissues and organs, which can manifest with local or multiple organ and tissue involvement.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The general incidence of IgG4-RD is estimated to be 0.78–1.39 per 100,000 population/year and although it has been well documented since the early 2000s, it can be difficult to diagnose, as histopathological confirmation is often required.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Eye involvement occurs in approximately 17–23% of patients and can affect both the muscles and the orbital and periorbital tissues.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Some association with the simultaneous occurrence of head and neck lymphomas (MALT lymphomas) has been reported, but it is not clear whether there is a true causal relationship between the two processes.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Orbital tumours cover various categories, from inflammatory and infectious to cancerous and metastatic. This poses challenges in terms of treatment and requires the collaboration of several medical disciplines, with neurosurgery offering an optimal solution for some of these patients.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">We present the case of a 64-year-old woman with no relevant previous history who was referred to our department for right mydriasis, proptosis and partial ophthalmoplegia of the right eye attributable to paralysis of the third cranial nerve. A head MRI scan revealed a lesion in the apex of the right orbit measuring approximately 3 × 1.6-cm in diameter, located between the external and inferior rectus muscles and the optic nerve, which was displaced and homogeneously enhanced after the administration of contrast (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A–F). After clinical-radiological assessment and without a clear diagnosis, it was decided to approach the mass directly by means of lateral orbitotomy with preservation of the lateral rectus muscle according to the standard technique described by Rhoton.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> With the head turned 15 ° to the left side and positioned with Mayfield®, an S-shaped skin incision is made from the orbital rim to the horizontal branch of the zygoma. The subcutaneous tissues are dissected down to the periosteum of the lateral orbital margin and zygoma. The periosteum and temporalis muscle are reflected posteriorly, exposing the lateral wall of the orbit in the temporal fossa. Two to three 2 by 2 gauze pads are inserted into the temporal fossa to aid retraction and haemostasis. A straight saw incision is made approximately 5 mm above the frontozygomatic suture and just above the zygomatic arch inferiorly. In our patient, after opening the periorbita and lateralising the extraocular musculature, we located a lesion with a hard consistency and several pieces were removed without definitive intraoperative pathology results, so we decided to continue with the excision. Using a SONOPET ultrasonic aspirator (Stryker® Neuro Spine ENT, USA), aggressive debulking of the lesion was performed, with the most distal part of the lesion surrounding the optic nerve, but without infiltrating it, achieving a satisfactory macroscopic excision. The bone fragment was then repositioned with titanium mini-plates and the skin closed with intradermal suture (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>G–L). The patient recovered well after surgery, with no neurological symptoms. Her proptosis had been corrected, but with persistent paralysis of the third cranial nerve in the postoperative period, and was discharged 48 h after the operation (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was followed up in our outpatient clinic at three, six, 12 and 24 months with no signs of tumour recurrence and with a clear improvement in the ophthalmoplegia without diplopia. MRI at two years ruled out tumour recurrence without any associated complications (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><p id="par0030" class="elsevierStylePara elsevierViewall">Pathology examination revealed a lymphoplasmacytic infiltration with fibrosis of the periorbital tissue with histological and immunohistochemical findings suggestive of IgG4-RD (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The patient was therefore referred to rheumatology, involvement of any other organs or tissues was ruled out and treatment was started with dexamethasone 4 g/8 h orally in a tapering regimen for two weeks without subsequent recurrences.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">IgG4-RD is a chronic condition characterised by the infiltration of one or more organs.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> However, due to the non-specific radiological features of these pseudotumours, patients with IgG4-RD are often misdiagnosed as having cancer, as they can mimic tumours, infections or autoimmune diseases. The main abnormal findings include lymphoplasmacytic infiltrates with predominantly IgG4 plasma cells in the affected tissue, as well as storiform fibrosis and obliterative phlebitis.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> For the diagnosis of IgG4-RD, a minimum of 30–50 IgG4 plasma cells per high-power field is considered a sufficient criterion.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> During the acute phase, elevated serum IgG4 levels are found in 51–70% of patients, and these patients usually recover well after treatment with systemic glucocorticoids, sometimes in combination with immunomodulatory or immunosuppressive drugs.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> However, in some cases, such as the one presented here, local progression of the disease and the development of new symptoms make surgical treatment necessary.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7–9</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The differential diagnosis is broad and includes several possibilities such as ocular adnexal lymphoma, as patients may present with an orbital space-occupying lesion associated with exophthalmos, decreased visual acuity and diplopia, similar to what was initially observed in our patient.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Inflammatory myofibroblastic tumour, which tends to occur in the paediatric age group and may manifest as a well-defined soft tissue mass, should also be considered.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Orbital meningioma, Graves' orbitopathy and idiopathic orbital inflammation, formerly known as benign orbital lymphoid hyperplasia, within which IgG4-RD appears to account for 25%–50% of orbital pseudotumours, although less likely, are also considered within the differential diagnosis.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8,10</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Whether patients with IgG4-RD should receive maintenance therapy is still under debate.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Reintroduction of glucocorticoid therapy is indicated in patients who relapse after successful induction. After relapse, the introduction of a glucocorticoid-sparing agent is often the key to controlling the disease.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The surgical approach used in our patient aims to access the orbit to achieve complete excision of the tumour, in addition to freeing the optic nerve, in a minimally invasive manner.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">11–13</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Lateral orbitotomy is an approach first described in 1888 by Kronlein et al.,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> which has subsequently undergone several modifications to become a safe and useful technique for orbital lesions lateral to the optic nerve.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,12</span></a> This type of approach provides improved visualisation and manipulation of structures, allowing rapid identification of the optic nerve and the eyeball, and achieving wide resections. As it is a familiar approach from a neurosurgical point of view, it is another tool that we can offer to this type of patient by taking on an active position on a disorder on the boundaries of our scope of action.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">14,15</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The size, location and type of tumour are the main factors determining the approach to the orbit. The "round the clock" concept described by Paluzzi A et al. advocates the use of the right orbit (viewed from the front) as a clock model, where the approach is determined by the time position of the tumour. Lateral micro-orbitotomy allows access to the orbit from 8 to 10 o'clock, while frontotemporal craniotomy with orbital osteotomy allows access to the orbit from 9 to 1 o'clock; if a zygomatic osteotomy is added, access is extended from 6 to 8 o'clock.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> Accordingly, in our patient, as the lesion was found between 7 and 11 o'clock, a lateral micro-orbitotomy was chosen.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Other authors suggest that minimally-invasive endoscopic techniques using a transorbital upper eyelid approach or an endonasal endoscopic approach successfully provide minimally-invasive 360 ° circumferential access to the entire orbit with acceptable morbidity compared to transcranial approaches.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10,11</span></a> However, a number of complications have been reported following endoscopic approaches, including injury to the internal carotid artery, ophthalmic artery or optic nerve, cerebrospinal fluid fistula and difficult-to-contain epistaxis. Because of this, we believe that extensive experience in the use of the endoscopic technique is necessary to recommend the treatment of lesions lateral to the optic nerve, especially when there is a microsurgical alternative with good results.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13,16</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Complications associated with orbital surgery, regardless of modality, include decreased visual acuity, cerebrospinal fluid fistula, nerve paralysis (supraorbital nerve, frontal branches of the facial nerve and third cranial nerve) and enophthalmos.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">14,15</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">In our experience, lateral micro-orbitotomy is a plausible, elegant and low-comorbidity option for intraconal and extraconal tumours with lateral location. Different specialities may be involved in orbital disorders (maxillofacial, ophthalmology, plastic surgery, neurosurgery) due to the special location of the orbit, and it is necessary to defend the role of neurosurgical approaches as a viable, safe and effective option. Cosmetic results appear to be excellent, and this minimally invasive approach may avoid morbidities associated with a wider transcranial approach.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conclusion</span><p id="par0080" class="elsevierStylePara elsevierViewall">The application of the microsurgical approach using lateral orbitotomy has emerged as a useful tool in the treatment of symptomatic patients with orbital pseudotumour associated with IgG4-related diseases. The results obtained in our clinical case support the efficacy and safety of this technique, which allows us to perform anything from a simple biopsy to a complete resection of the lesion, with significant improvement in the patients' symptoms. This surgical approach we describe offers a viable and promising alternative in the management of this complex disorder.</p></span></span>"
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"resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Immunoglobulin G4-related disease (IgG4-RD) is characterized by a systemic fibroinflammatory infiltrate that often involves the orbit in addition to other tissues. Thus it has to be considered in the differential diagnosis of orbital tumors. We report the clinical case of a 64-year-old woman who presented with right mydriasis, progressive proptosis and paralysis of the third cranial nerve of 1 year of evolution. Cranial MRI identified an intraconal lesion of the right orbit, located between the external and inferior rectus muscles and the optic nerve, and she was scheduled for surgery by transcranial approach with lateral micro-orbitomy. A satisfactory macroscopic excision was achieved with no remarkable complications and a definitive deferred histological result of pseudotumor by IgG4-RD. Follow-up for 24 months showed no tumor recurrence, and the patient clinically improved from ophthalmoplegia. This case highlights the efficacy of lateral orbitotomy in the etiologic diagnosis and successful therapeutic outcome of complex orbital lesions associated with IgG4-RD pseudotumor.</p></span>"
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"resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">La enfermedad relacionada con Inmunoglobulina G4 (ER-IgG4) se caracteriza por un infiltrado fibroinflamatorio sistémico que a menudo afecta a la órbita además de otros tejidos. Así pues se ha de considerar en el diagnóstico diferencial de los tumores orbitarios. Se expone el caso clínico de una mujer de 64 años que presentó midriasis derecha, proptosis progresiva y parálisis del III par craneal de 1 año de evolución. Mediante RM craneal se identificó una lesión intraconal de la órbita derecha, situada entre los músculos recto externo e inferior y el nervio óptico programándose para cirugía mediante abordaje transcraneal con micro-orbitomia lateral. Se consiguió una escisión macroscópica satisfactoria sin complicaciones reseñables y un resultado histológico diferido definitivo de pseudotumor por ER-IgG4. El seguimiento durante 24 meses no mostró recidiva tumoral, y la paciente mejoró clínicamente de la oftalmoplejía. Este caso subraya la eficacia de la orbitotomía lateral en el diagnóstico etiológico y los resultados terapéuticos satisfactorios de las lesiones orbitarias complejas asociadas al pseudotumor por ER-IgG4.</p></span>"
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"en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Preoperative MRI and intra-surgical imaging. (A and D) Axial and coronal T1-weighted sequences without gadolinium, respectively, showing an intraconal nodular mass (green dashed line). Note the easy identification of the optic nerve (red arrows). (B and E) Axial and coronal T1-weighted contrast-enhanced sequences, respectively, confirming homogeneous enhancement of the lesion. (C) Axial T2-weighted sequences showing clear medial displacement of the right optic nerve. (F) Image showing the position of the tumour (light green) from 7 o'clock to 10 o'clock following the "clock" model. (G) Image of the surgical incision. (H) Lateral orbitotomy performed with piezoelectric saw. (I) Opening of the periorbita and lateralisation of the external lateral muscle. No tumour infiltration of the muscle is observed. (J) Detailed image of the lesion with a whitish appearance and sparse arterial vascularisation. The eyeball can be seen in the anterior part. (K) Final repositioning of the zygomatic bone with titanium mini-plates. (L) Final image of the sutured incision with intradermal technique.</p>"
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