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Fig. 1. Fundoscopy in the acute phase showing tortuous veins, dot and blot hemorrhages, and marked disk edema (A) as well as exudates on fluorescein angiography (B) typical for central retinal vein occlusion. One month later, the fluorescein angiography is normal and no edema is present (C).
patients with transient monocular visual disturbances and initially normal fundoscopy, as the misdiagnosis of CRVO is common [11]. Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References [1] Olesen J. The headaches. Philadelphia: Lippincott Williams & Wilkins; 2006. [2] Richards W. The fortification illusions of migraines. Sci Am 1971;224:88–96. [3] Russell MB, Olesen J. A nosographic analysis of the migraine aura in a general population. Brain 1996;119:355–61. [4] Daroff RB. Retinal migraine. J Neuroophthalmol 2007;27:83. http://dx.doi.org/ 10.1097/WNO.0b013e3180331435. [5] Eriksen MK, Thomsen LL, Olesen J. The Visual Aura Rating Scale (VARS) for migraine aura diagnosis. Cephalalgia 2005;25:801–10. http://dx.doi.org/ 10.1111/j.1468-2982.2005.00955.x.
[6] Grosberg BM, Solomon S, Friedman DI, et al. Retinal migraine reappraised. Cephalalgia 2006;26:1275–86. http://dx.doi.org/10.1111/j.1468-2982.2006. 01206.x. [7] Headache Classification Committee of the International Headache Society (IHS). The International Classification of Headache Disorders, 3rd edition (beta version). Cephalalgia 2013;33:629–808. http://dx.doi.org/10.1177/ 0333102413485658. [8] Fong ACO, Schatz H. Central retinal vein occlusion in young adults. Surv Ophthalmol 1993;37:393–417. http://dx.doi.org/10.1016/0039-6257(93) 90138-W. [9] McIntosh RL, Rogers SL, Lim L, et al. Natural history of central retinal vein occlusion: an evidence-based systematic review. Ophthalmology 2010;117:1113–23. http://dx.doi.org/10.1016/j.ophtha.2010.01.060. [10] Friedman MW. Occlusion of central retinal vein in migraine. AMA Arch Ophthalmol 1951;45:678–82. http://dx.doi.org/10.1001/archopht.1951. 01700010693010. [11] Hill DL, Daroff RB, Ducros A, et al. Most cases labeled as ‘‘retinal migraine’’ are not migraine. J Neuroophthalmol 2007;27:3–8. http://dx.doi.org/10.1097/ WNO.0b013e3180335222.
http://dx.doi.org/10.1016/j.jocn.2015.03.055
A massive pelvic mucocele presenting as a cystic sacral mass Christine Boone a, C. Rory Goodwin a, Genevieve Crane b, James Pendleton a, Daniel Sciubba a,⇑ a b
Neurosurgery Department, The Johns Hopkins University School of Medicine, 600 North Wolfe Street, Baltimore, MD 21287, USA Pathology Department, The Johns Hopkins University School of Medicine, Baltimore, MD, USA
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Article history: Received 30 March 2015 Accepted 5 April 2015
Keywords: Complication Crohn’s disease Mucinous cystadenoma Sacrum Spine Surgery
a b s t r a c t We present a man, with a complex medical and surgical history, who had a large pelvic non-appendiceal cystadenoma, presenting as a cystic sacral mass causing obstructive urinary symptoms and renal failure. Mucocele should be included in the differential diagnosis of patients who present with large sacral masses, and who have a significant history of pelvic and abdominal surgery and inflammatory bowel disease. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction
⇑ Corresponding author. Tel.: +1 141 0502 9982. E-mail address:
[email protected] (D. Sciubba).
Sacral and presacral masses are often asymptomatic and difficult to diagnose. Abdominal or pelvic mucoceles are rare, and most commonly arise in the appendix as benign, neoplastic mucinous cystadenoma.
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Fig. 1. Preoperative sacral mucocele. (a) Sagittal CT scan demonstrating a large, multi-compartmented cystic mass (+) displacing the bladder (⁄) anteriorly and superiorly. (b) Sagittal T2-weighted MRI without contrast, showing no communication between the mass (+) and the subarachnoid space. (c) An axial CT scan of the pelvis shows the multiloculated cystic structure of the mass and involvement with the right inferior sacral neural foramina.
Fig. 2. Low grade mucinous neoplasm. (a) Histology revealed a cystic structure with copious acellular mucin containing necrotic debris (hematoxylin and eosin [H&E]; 64 magnification). (b) The fibrous capsule demonstrated a scant, low grade mucinous epithelial lining (H&E; 160 ), positive for (c) cytokeratin 20 (100 ) and (d) cytokeratin 7 (160 ). This figure is available in colour at www.sciencedirect.com.
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2. Case report
for cytokeratin 7 and cytokeratin 20 (Fig. 2). By exclusion, the capsule was diagnosed as a low grade mucinous neoplasm.
2.1. Preoperative A 59-year-old man, with a medical history of chronic kidney disease and Crohn’s disease with intestinal rupture, was diagnosed with an extremely large pelvic mass, presumably causing obstructive renal failure. His surgical history was significant for over 40 colorectal operations. He was referred to neurosurgery after a pelvic MRI revealed a large mass, appearing to abut and erode the ventral sacral surface and invade several sacral foramina (Fig. 1). 2.2. Radiologic findings A CT myelogram, performed for possible pseudomeningocele, revealed a multi-compartmental cystic mass measuring 14.0 9.6 cm in the presacral space, compressing and displacing the bladder anteriorly. Chronic remodeling of the anterior sacral cortex with widening of the foramina of S2 and S3 was observed. There was no evidence of direct communication between the mass and the subarachnoid space, excluding pseudomeningocele or meningocele. A computed tomography guided needle biopsy demonstrated mucin, with no evidence of cerebrospinal fluid, but did not confirm a diagnosis. 2.3. Operation
2.5. Postoperative His postoperative MRI demonstrated the expected postsurgical changes, including removal of the cyst contents. He was discharged from the hospital to home on postoperative day 6, neurologically intact, and with drains in place. At the 3 month follow-up, his MRI suggested a recurrence of the mucocele in the resection cavity, measuring 8.4 6.3 cm (Fig. 3). The general surgery team, in conjunction with the patient, decided that further attempts to resect the mass could lead to more trauma without improved bladder function. In addition, his long history of obstructive uropathy from both urethral disease and bilateral ureteral stones had resulted in stage 5 chronic kidney disease. 3. Discussion We present the first report, to our knowledge, of a giant sacral mucinous adenoma mucocele invading the sacrum and causing urinary incontinence and retention. Our patient underwent a low sacrectomy to confirm the diagnosis, drain the mucocele, and remove the capsule that was non-adherent to critical structures. Mucoceles are most commonly found in the appendix, and are rare occurrences in the abdomen or pelvis [2]. The location of the mass
A definitive diagnosis could not be made preoperatively. Surgery was proposed with a presumptive differential diagnosis of sacral tumor versus mucocele (from incomplete removal of glandular tissue in previous colorectal surgeries). A complete resection of the mass was recommended, in conjunction with general surgery to remove the mass and remaining capsule from other pelvic organs in a staged anterior approach. However, general surgery suggested that if a mucocele were present, complete excision may not be possible due to extensive involvement of the pelvic viscera and an inability to remove further bowel tissue in this already compromised patient (with an already limited bowel). A posterior approach was selected because of the significant scarring and complications from previous abdominal procedures. The man was positioned prone on an Andrews frame with his head pinned with a Mayfield Clamp, as previously reported [1]. After a subperiosteal dissection of the mid to distal sacrum below the S4 level, the dorsal foramina were exposed and mucinous exudate was observed. An amputation of the distal sacrum exposed a capsular layer of tissue, which released approximately 4 L of mucinous material upon incision. The pathology confirmed that the lining of the mass was a mucin producing epithelium, and contained mucin. Therefore, an aggressive cyst wall resection was attempted. A dissection and excision of the capsule adjacent to the sacrum was performed, however, the portions of the capsule that were adherent to the pelvic structures were left behind. Plastic surgeons then placed two Jackson–Pratt drains in the abdomen and performed a posterior complex closure. There were no intraoperative complications. 2.4. Pathological findings The capsule attached to the portion of amputated sacrum consisted of fibrous tissue with scant mucinous epithelium, focal dissection of mucinous material through the fibrous capsule, and some calcifying debris. The content of the mass was extracellular mucin, without evidence of mucinous epithelium floating within the extensive sampled mucin, which would have suggested a carcinoma. The immunostaining of the epithelial lining was positive
Fig. 3. Postoperative sagittal CT scan depicting a recurrence of the mucocele (+) but with reduced displacement of the bladder (⁄).
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raised suspicion of anterior sacral meningocele or pseudomeningocele, but we excluded these with a CT myelogram scan, which showed no evidence of a dural breach. Although the CT guided biopsy did not yield a confirmatory diagnosis, the mucin indicated a mucocele. The man’s extensive history of Crohn’s disease and surgeries also supported the differential diagnosis of a pelvic mucocele.
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important to consider mucocele as a diagnosis. Long term follow-up is also essential in these patients. Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References
4. Conclusion This man is the first reported patient, to our knowledge, with an extra-appendiceal mucinous cystadenoma with sacral spine involvement. In patients with histories of inflammatory bowel disease and/or extensive pelvic and abdominal surgeries, it is
[1] Goodwin CR, Recinos PF, Omeis I, et al. Prevention of facial pressure ulcers using the Mayfield clamp for sacral tumor resection. J Neurosurg Spine 2011;14:85–7. http://dx.doi.org/10.3171/2010.9.SPINE09516. [2] Ai XB, Feng JC, Gong FY, et al. Endoscopic therapy of colonic liver flexure mucocele. Case Rep Gastroenterol 2011;5:433–7. http://dx.doi.org/10.1159/ 000330484.
http://dx.doi.org/10.1016/j.jocn.2015.04.006
Sacrum fracture following L5–S1 stand-alone interbody fusion for isthmic spondylolisthesis Kevin Phan, Ralph J. Mobbs ⇑ Neuro Spine Clinic, Prince of Wales Private Hospital, Suite 7a, Level 7, Barker Street, Randwick, NSW 2031, Australia NeuroSpine Surgery Research Group, Sydney, NSW, Australia
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Article history: Received 21 March 2015 Accepted 28 March 2015
Keywords: Anterior lumbar interbody fusion Cage Complications Implant Isthmic spondylolisthesis Sacrum Stand-alone
a b s t r a c t We report a 72-year-old man with a rare sacral fracture following stand-alone L5–S1 anterior lumbar interbody fusion for isthmic spondylolisthesis. The man underwent a minimally invasive management strategy using posterior percutaneous pedicle fixation and partial reduction of the deformity. We also discuss the current literature on fusion procedures for isthmic spondylolisthesis. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction Isthmic spondylolisthesis is characterized by subluxation of one vertebral body over another, causing spinal instability which is usually initially attributed to lytic defects in the pars interarticularis [1]. A pars defect disconnects the anterior column segment from its posterior support, which may produce excessive loading, leading to disc degeneration, subluxation and nerve impingement. Surgical treatment of isthmic spondylolisthesis remains controversial. At the L5–S1 level, decompression and instrumented fusion can be achieved using several techniques, including a posterior and/or anterior approach, or non-instrumented technique. Traditionally, a posterior lumbar interbody fusion or transforaminal lumbar interbody fusion is commonly performed for direct nerve decompression and stabilization [2]. However, these posterior approaches may be associated with increased operative duration, blood loss and longer hospitalization. For low grade isthmic spondylolisthesis with radicular symptoms, multiple studies have reported excellent results with the anterior lumbar interbody ⇑ Corresponding author. Tel.: +61 2 9650 4766. E-mail address:
[email protected] (R.J. Mobbs).
fusion (ALIF). The anterior approach offers the advantages of minimal tissue and neural disruption, with restoration of disc height, sagittal alignment and indirect nerve decompression [3]. To provide fixation, the ALIF cage design has evolved over the years to a stand-alone construct with an integrated distraction cage and fixation plate. We report a man with a delayed sacral fracture complication of ALIF for isthmic spondylolisthesis, who was managed with the minimally invasive strategy of posterior percutaneous pedicle fixation and partial reduction of the deformity.
2. Case report A 72-year-old man presented with Grade I isthmic spondylolisthesis, with several years of progressive lower back pain and predominant bilateral L5 radiculopathy (Fig. 1A). Following some unsuccessful conservative treatment options, the decision was made to proceed with surgical intervention. The senior author adopted a strategy of stand-alone ALIF (ANCHOR-L Implant; Stryker, Kalamazoo, MI, USA) to restore the foraminal height and stabilize the spondylolisthesis (Fig. 1B).