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8. Chang BY, Moriarty P, Cunniffe G, et al. Accelerated growth of a primary orbital schwannoma during pregnancy. Eye (Lond) 2003;17:839–41. 9. Lee BJ, Schoenfield L, Perry JD. Orbital intramuscular hemangioma enlarging during pregnancy. Ophthal Plast Reconstr Surg 2009;25:491–3. 10. Birkholz ES, Lee AG, Nerad JA, et al. A pregnant pause. Surv Ophthalmol 2010;55:162–8. 11. Sugo N, Yokota K, Nemoto M, et al. Accelerated growth of an orbital schwannoma during pregnancy. J Neuroophthalmol 2007;27:45–7. 12. Das JK, Sharma AS, Deka AC, et al. Solitary fibrous tumor of the orbit presenting in pregnancy. Indian J Ophthalmol 2009;57:238–40. 13. Shields JA, Shields CL, Christian C, Eagle RC. Orbital nodular fasciitis simulating a dermoid cyst in an 8-month old child. Ophthal Plast Reconstr Surg. 2001;17:144–8.

Tension Pneumocephalus Following Orbital Exenteration Wen-Shi Shieh, M.D.*; Christopher Farrell, M.D.†; Joseph Curry, M.D.‡; Ann P. Murchison, M.D., M.P.H.§; and Jurij R. Bilyk, M.D.§ Abstract: Pneumocephalus is a known complication of skull base surgery, but is rarely seen by orbital surgeons. We report a case of postoperative mental status changes after exenteration due to tension pneumocephalus. After surgical and medical management, the patient’s pneumocephalus resolved and she recovered fully. Risk factors for tension pneumocephalus, mechanism, clinical presentation, and management techniques are discussed.

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ension pneumocephalus, defined as the accumulation of air under pressure within any intracranial compartment, can lead to neurologic deterioration from mass effect and parenchymal herniation, and is considered a neurosurgical emergency.1 It is most commonly associated with skull base-related trauma2 or prior neurosurgical procedures such as craniotomy3,4, and with transnasal endoscopic surgery for paranasal sinus disease or pituitary lesions.5 Symptomatic pneumocephalus following orbital surgery is a rare event. The authors describe a case of tension pneumocephalus following uncomplicated orbital exenteration. This report is HIPPA-compliant.

CASE REPORT An 87-year-old woman with a previously excised periocular basal cell carcinoma presented with a frozen globe and was found to have tumor recurrence invading the right orbit and surrounding bone on biopsy. An exenteration of the right orbit including removal of the lateral orbital wall to the level of greater wing of the sphenoid, rotation of a temporalis muscle flap, and a myocutaneous midfacial flap repair was performed with no obvious intraoperative complications. On the second postoperative day, altered mental status and increased confusion Accepted for publication December 11, 2014. *Wills Eye Hospital; †Department of Neurosurgery, Thomas Jefferson University Hospital; ‡Department of Otolaryngology Head Neck Surgery, Thomas Jefferson University Hospital; and §Skull Base Division, NeuroOphthalmology Service, Wills Eye Hospital, Philadelphia, Pennsylvania, U.S.A. The authors have no financial or conflict of interest to disclose. Address correspondence and reprint requests to Ann P. Murchison, M.D., M.P.H., Department of Research, Wills Eye Hospital, 840 Walnut Street, Suite 802, Philadelphia, PA 19107; Email: [email protected] DOI: 10.1097/IOP.0000000000000407

Case Reports

were noted. A head CT scan revealed an extensive pneumocephalus with associated intracranial mass effect on the frontal lobes bilaterally along with the “Mount Fuji sign” (Fig. 1).6 Neurosurgical consultation was initiated and conservative management with supplemental oxygenation was recommended to expedite reabsorption of the intracranial air. Despite these measures, no clinical or radiographic improvement was noted on the following day. The patient subsequently underwent orbital exploration and 3 small areas of bony dehiscence with associated dural defects were identified in the orbital roof. In addition, a bony dehiscence was noted in the lacrimal sac fossa communicating with the nasopharynx. The pneumocephalus was relieved by gentle irrigation of the subdural space with saline to allow for reexpansion of the brain. The dural defects were then primarily repaired with DuraMatrix (Stryker Corporation, Kalamazzo, MI, U.S.A), a collagen-engineered membrane dural substitute, and a fat graft harvested from the abdomen was utilized to cover the bony orbital defects. Finally, the previous temporalis muscle flap was revised to provide reinforcement of the newly placed fat graft. The patient remained intubated to facilitate 100% inspired oxygen during recovery in the intensive care unit. Subsequent serial CT scans demonstrated decreased air volume within the intracranial space and frontal lobe expansion (Fig. 2), which was mirrored clinically by the patient’s rapid mental status improvement. The patient has done well, with no evidence of tumor recurrence or focal neurologic deficits 1 year after surgery.

DISCUSSION Air within the intracranial space has many well-described clinical signs, with headache being the most common presenting complaint. Other signs and symptoms include cerebrospinal fluid (CSF) rhinorrhea, meningismus, seizures, altered mental state, and hemiparesis.7 Peri- and intraoperative risk factors that have been cited for development of pneumocephalus include head position during surgery, surgical duration, nitrous oxide anesthesia, hydrocephalus, hyperventilation, spinal or epidural anesthesia, barotrauma, continuous CSF drainage via lumbar drain, infection, and neoplasms. Additional factors include incomplete reconstruction following skull base procedures and positive pressure events such as coughing, sneezing, nose blowing, or Valsalva maneuvers.8,9 A small amount of pneumocephalus (“nontension pneumocephalus”) is a common finding following intracranial surgery and typically resolves with observation alone. Tension pneumocephalus is a significantly more serious finding and is a neurosurgical emergency.6 In tension pneumocephalus, intracranial air causes a mass effect, most commonly on the frontal lobes. Radiographically, this may manifest as the “Mount Fuji sign.”6 Compression and distortion of the frontal lobes by increased pressure results in widening and separation of the frontal lobes in addition to compression; on axial CT images, these features mimic the silhouette of Mount Fuji (Fig. 1).6,10 Interestingly, the volume of air seen on CT is not an accurate predictor of tension pneumocephalus.10 There are 2 postulated theories that explain the development of tension pneumocephalus. The first is the “inverted bottle” mechanism, where intracranial negative pressure created by drainage of CSF is relieved by influx of air. Alternatively, the “ball-valve” theory suggests that positive pressure induced during episodes of sneezing, coughing, or vomiting force air through cranial defects.11 In our case, the bony defects in the lacrimal sac fossa and the orbital roof may have facilitated the influx of air beneath the soft tissue flaps; a slight positive pressure through this tract most likely occurred postoperatively with

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FIG. 1.  CT images of patient following mental status changes. Note the separation of the frontal lobes (arrows). This finding, along with the shape of the intracranial air on axial views, has been coined the “Mount Fuji sign” and is characteristic of tension pneumocephalus. Left, axial. Center, coronal. Right, parasagittal.

FIG. 2.  Axial CT images of patient. Left, following mental status changes. Center, 2 days later, immediately following surgical exploration. Right, 1 month later.

mask ventilation and coughing. Furthermore, the flaps may have had a ball-valve effect, trapping air within the cranium. Currently, few studies with significant evidence exist in the literature with regard to the treatment of tension p­ neumocephalus. The main principles of management include decompression of the aerocele and closure of the defect. A common approach to address the former utilizes supplemental oxygen, which has been demonstrated to accelerate the resolution of intracranial air.12 Conservative measures aimed at closure of the fistula include bed rest with head elevation and CSF diversion via serial lumbar punctures or subarachnoid drains.5,7 In the setting of tension pneumocephalus with acute neurologic decline, emergent neurosurgical intervention in the form of burr hole placement and air aspiration may be necessary for immediate relief of mass effect. Larger defects often fail conservative treatment and require early surgical intervention with multilayered repairs of bone and mucosa or fascia. According to DelGaudio and Ingley,5 exploration and defect repair with mucosal grafting should be considered

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even if pneumocephalus resolves with conservative therapy. The authors cite the possibility of persistent CSF leak due to incomplete or compromised mucosal regeneration and increased risk of subsequent ascending meningitis as reasons for this approach. In a study of 160 patients with traumatic CSF leaks, Eljamel and Foy13 report an overall risk of 30.6% for meningitis. Conservative treatment of CSF leaks has a similar reported risk (29%) of meningitis.14 Pneumocephalus has been reported following orbital barotrauma from either compressed air or helium.15–19 In each of these cases, the pneumocephalus was small, nonprogressive, and either neurologically asymptomatic or minimally symptomatic. Pneumocephalus may also occur secondary to orbital injury with penetration of the skull base.20 Fortunately, pneumocephalus following orbital surgery is a rare complication. To the authors’ knowledge, there is only 1 other reported case of delayed tension pneumocephalus in a patient with recurrent adenoid cystic carcinoma of the lacrimal gland that was treated by orbital exenteration and reconstruction.21

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In an article by Gil et al.,22 various approaches for anterior skull base reconstruction after oncological resection are discussed, including the use of a temporalis muscle flap to cover the orbital socket in cases of exenteration, as was performed in our patient. They report a 5% incidence of CSF leak, intracranial infection, and tension pneumocephalus in their series, concluding that double-layer fascial graft alone for repair of skull base defects was adequate in prevention of these complications in the majority of cases. Although rare, tension pneumocephalus should be considered in any patient experiencing headache or altered mental status following orbital exenteration.

REFERENCES 1. Sweni S, Senthilkumaran S, Balamurugan N, et al. Tension pneumocephalus: a case report with review of literature. Emerg Radiol 2013;20:573–8. 2. Finch MD, Morgan GA. Traumatic pneumocephalus following head injury. A complication of general anaesthesia. Anaesthesia 1991;46:385–7. 3. Reasoner DK, Todd MM, Scamman FL, et al. The incidence of pneumocephalus after supratentorial craniotomy. Observations on the disappearance of intracranial air. Anesthesiology 1994;80:1008–12. 4. Pitts LH, Wilson CB, Dedo HH, et al. Pneumocephalus following ventriculoperitoneal shunt. Case report. J Neurosurg 1975;43:631–3. 5. DelGaudio JM, Ingley AP. Treatment of pneumocephalus after endoscopic sinus and microscopic skull base surgery. Am J Otolaryngol 2010;31:226–30. 6. Michel SJ. The Mount Fuji sign. Radiology 2004;232:449–50. 7. Campanelli J, Odland R. Management of tension pneumocephalus caused by endoscopic sinus surgery. Otolaryngol Head Neck Surg 1997;116:247–50. 8. Schirmer CM, Heilman CB, Bhardwaj A. Pneumocephalus: case illustrations and review. Neurocrit Care 2010;13:152–8. 9. Pepper JP, Lin EM, Sullivan SE, et al. Perioperative lumbar drain placement: an independent predictor of tension pneumocephalus and intracranial complications following anterior skull base surgery. Laryngoscope 2011;121:468–73. 10. Ishiwata Y, Fujitsu K, Sekino T, et al. Subdural tension pneumocephalus following surgery for chronic subdural hematoma. J Neurosurg 1988;68:58–61. 11. Markham J. Pneumocephalus. In: Vinken PJ, Bruyn GW, ed. Handbook of Clinical Neurosurgery. Amsterdam: North Holland Publishing Co; 1976:201–13. 12. Standefer M, Bay JW, Trusso R. The sitting position in neurosurgery: a retrospective analysis of 488 cases. Neurosurgery 1984;14:649–58. 13. Eljamel MS, Foy PM. Acute traumatic CSF fistulae: the risk of intracranial infection. Br J Neurosurg 1990;4:381–5. 14. Bernal-Sprekelsen M, Bleda-Vázquez C, Carrau RL. Ascending meningitis secondary to traumatic cerebrospinal fluid leaks. Am J Rhinol 2000;14:257–9. 15. Kang BS. Disseminated head and neck emphysema with pneumocephalus due to air compressor injury into orbit. Am J Emerg Med 2007;25:223–5. 16. Yuksel M, Yuksel KZ, Ozdemir G, et al. Bilateral orbital emphysema and pneumocephalus as a result of accidental compressed air exposure. Emerg Radiol 2007;13:195–8. 17. Williams TR, Frankel N. Intracerebral air caused by conjunctival laceration with air hose. Arch Ophthalmol 1999;117:1090–1. 18. Walrath JD, Kazim M. Compressed helium injury to the or bit resulting in pneumocephalus. Ophthal Plast Reconstr Surg 2006;22:234–5. 19. Koenig RP. Traumatic eye and intracranial air-movement from a subconjunctival to an intracranial position. Am J Ophthalmol 1977;83:915–7. 20. Detorakis ET, Drositis I, Drakonaki EE, et al. Pneumocephalus and presumed meningitis following inconspicuous penetrating periocular trauma. Acta Ophthalmol Scand 2004;82:603–5.

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21. Esson MD, Blanco-Guzman M, Douglas PS. Delayed tension pneumocephalus complicating orbital exenteration. Br J Oral Maxillofac Surg 2005;43:123–5. 22. Gil Z, Abergel A, Leider-Trejo L, et al. A comprehensive algorithm for anterior skull base reconstruction after oncological resections. Skull Base 2007;17:25–37.

Forget Me Not: A Case of Gossypiboma (Textiloma) Mimicking an Orbital Tumor Kaustubh Mulay*, Vishal Sharma†, and Santosh G. Honavar† Abstract: Retained foreign bodies are not infrequent following surgical procedures and are associated with medico-legal issues. Gossypiboma following ocular surgeries is rare but should be included in the differential diagnosis of a postoperative patient presenting with pain or mass. We report on one such case of gossypiboma following surgical excision of lacrimal gland.

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surgical sponge is the most frequently encountered retained foreign-body (RFB). Gossypiboma (textiloma) is a rare surgical complication resulting in the formation of a mass around a surgical swab retained inside the body following a surgery. The term “gossypiboma” is derived from 2 words, “gossypium” (Latin) meaning “cotton” and “boma” (Swahili) meaning “place of concealment.” “Textiloma” is derived from the words “textilis” (Latin = weave) and “oma” (Greek = disease, tumor).The earliest report on “gossypiboma” was by Wilson in 1884.1 The actual incidence of gossypiboma is difficult to estimate because of the low reporting rates due to medico-legal complexities associated. Gossypiboma occurs most commonly following intraabdominal surgery, the reported rate of occurrence being 1 in 1000 to 1500 interventions.1 Gossypiboma in the eye or orbit is rare. We report a case of orbital gossypiboma in a 63-year-old female following excision of lacrimal gland.

CASE REPORT Eleven months after surgical excision of left lacrimal gland for a suspected adenoid cystic carcinoma, a 63-year-old female presented to our clinic with a burning sensation and stickiness in the left eye since the surgery. On examination, proptosis (2 mm) of the left eye (Fig. 1A) was observed with restriction of left eye movements in the lateral gaze. Schirmer test showed wetting of 35 mm and 1 mm of paper strip in the right and left eyes, respectively. A soft mass was palpable in the temporal orbit. CT scan showed a homogenously enhancing, diffuse mass involving the lateral, superior and inferolateral orbit (Fig. 1B,C). Per-operatively, a surgical gauze (Fig. 1D) was found at the center of a firm mass in the inferotemporal orbit. Microscopy showed granulation tissue composed of proliferating vascular channels, fibrocollagenous stroma, and dense

Accepted for publication December 8, 2014. *National Reporting Centre for Ophthalmic Pathology; and †Oculoplastic, Facial Aesthetic and Ocular Oncology, Centre For Sight, Hyderabad, India The authors have no financial or conflict of interest to disclose. Address correspondence and reprint requests to Kaustubh Mulay, Centre For Sight, Ashoka Capitol Building, Banjara Hills, Road No. 2, Hyderabad 500034. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000406

© 2015 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

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Tension Pneumocephalus Following Orbital Exenteration.

Pneumocephalus is a known complication of skull base surgery, but is rarely seen by orbital surgeons. We report a case of postoperative mental status ...
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