Orbit, 2014; 33(2): 152–155 ! Informa Healthcare USA, Inc. ISSN: 0167-6830 print / 1744-5108 online DOI: 10.3109/01676830.2013.853807
C ASE REPORT
Spontaneous Subperiosteal Orbital Hematoma as Initial Presentation of Metastatic Lung Adenocarcinoma to the Skull: Case Report John Kirk Capua1, Eric Scott Stiner2, and Tina Grace Li3 1
Department of Neurological Surgery, Arrowhead Regional Medical Center, Colton, California, USA, Department of Neurological Surgery, Kaiser Permanente - Fontana Medical Center, Fontana, California, USA, and 3Department of Ophthalmology, Kaiser Permanente - Fontana Medical Center, Fontana, California, USA
2
ABSTRACT Introduction: Subperiosteal orbital hematoma is a rare occurrence, typically developing as a result of orbital trauma. The spontaneous formation of a subperiosteal orbital hematoma (sSOH) may also occur but is less frequent. To date there has been no documented cases of sSOH as the initial presentation of an unknown metastatic neoplasm to the skull. We provide a case of a woman with unknown lung adenocarcinoma that metastasized to the skull which caused the formation of a sSOH resulting in orbital compression syndrome. Case Report: A 57-year-old female presented with double vision, retro-orbital right eye pain, and vision loss in the right eye. A magnetic resonance imaging revealed a right orbital compressive lesion with an adjacent supraorbital skull lesion and separate left frontal skull lesion. Intra-operative findings along with post-operative immunohistochemistry staining revealed sSOH resulting from a metastatic lung adenocarcinoma to the skull. Further metastatic work up also revealed an occult lung mass and multiple spinal lesions. Conclusion: Differential diagnosis of etiologies causing the formation of sSOH in an adult without history of trauma should include metastatic neoplasm to the skull and warrants metastatic workup. Treatment options of sSOH have included observation with spontaneous resolution; however, we opted for surgical decompression of the eye and biopsy of the skull mass. Keywords: Orbital compression, skull metastasis, spontaneous subperiosteal orbital hematoma
INTRODUCTION
20 14
The incidence of skull metastases is estimated to be 3%.6 There has been an increase in detection of skull metastasis as a result of advancement of imaging technology.7 The most common neoplasms that metastasize to the skull are breast, prostate, lung, melanoma, renal, and thyroid.6 Bone metastases are often clinically symptomatic and present as the first manifestation of malignancy in 2.3% of lung cancer patients.8 Life expectancy after skull metastases is estimated to be 5 months.9 We provide a case of a woman with unknown lung adenocarcinoma that metastasized to the skull which caused the formation of a sSOH resulting in orbital compression syndrome. To our knowledge and review
Subperiosteal orbital hematoma (SOH) is a rare entity, most often caused by trauma.1 Formation of SOH may also occur spontaneously (sSOH).2 The formation of sSOH may originate from blood dyscrasias, sinusitis, anti-thrombolytic agents, vascular malformations, and orbital venous congestion.3 Pathogenesis of sSOH is thought to occur from increased pressure of the orbital veins that lack valves.4 Surgical decompression is typically the treatment of choice for SOH in symptomatic patients, though conservative management with resolution of sSOH has been reported.4,5
Received 2 May 2013; Revised 30 September 2013; Accepted 7 October 2013; Published online 15 November 2013 Correspondence: Dr. John Kirk Capua, Department of Neurological Surgery, Arrowhead Regional Medical Center, 400 North Pepper Ave. Colton, Ca. 92324, USA. E-mail: [email protected]
152
Spontaneous Subperiosteal Orbital Hematoma 153 of the available literature, this is the first documented case of the initial presentation of a metastatic neoplasm to the skull being the formation of a sSOH.
CASE REPORT A 57-year-old Caucasian female presented to the emergency department with complaint of one week history of diplopia, retro-orbital eye pain, and vision loss in the right eye. She denied any headaches, scalp pain, nor any other complaints of pain throughout the rest of the body. There was no history of trauma. Her past medical and surgical histories were also unremarkable. She did report of having smoked tobacco for 20 plus years.
Physical examination revealed significant right eye proptosis, periorbital erythema, conjunctival edema, and ophthalmoparesis resulting in diplopia. Visual fields of the right eye were diminished. There were no palpable masses of the scalp. No recent fevers or infections. Lab results revealed mildly elevated erythrocyte sedimentation rate, a normal level of C-reactive protein, and normal leukocyte count. Liver function and coagulation levels were within normal limits. An MRI of the brain was then obtained and showed the following characteristics (Figures 1A-2D). The patient was taken for immediate decompression of the globe as well as biopsy of the supraorbital skull lesion. A supraorbital eye brow incision was made, to approach both the superior retrobulbar lesion and supraorbital skull lesion. Intraoperative
FIGURE 1. T1 post-GAD imaging with the retrobulbar mass enhancement on axial view (A). Coronal T1 post-GAD view also showing the enhancing retrobulbar mass (B). T1 post-GAD sagittal view verifying the right supraorbital rim enhancing skull lesion (C). The T1 post-GAD coronal view revealed a second enhancing skull mass in the left frontal region (D). !
2014 Informa Healthcare USA, Inc.
154 J. K. Capua et al.
FIGURE 2. DWI sequence shows the retrobulbar mass to by hypointense (A). The right supraorbital skull lesion is hyperintense (B). T2 axial views showing the isointense right supraorbital skull mass (C) and the hyperintense retrobulbar lesion (D).
findings revealed a nonpurulent darkened color fluid in the subperiosteal space. There was no bony erosion but discoloration of the outer table of the skull in the supraorbital region. Engorged vessels adjacent to the discolored area were present. Abnormal tissue was seen in the diploic space as well as erosion through the inner table of the skull. Involvement of the dura but not the adjacent cerebral parenchyma was identified. Post-operatively she regained partial movement of the extraocular muscles and her visual field cuts improved significantly. Final immunohistochemistry staining revealed the skull lesion to be
undifferentiated metastatic lung adenocarcinoma (TTF-1 and CK7 positive). The subperiosteal fluid collection contained no malignant cells and was comprised of fibrin clot and granulation tissue consistent with hematoma. Cultures were negative. Further metastatic work up revealed a small right lung mass and multiple thoracic vertebral body lesions. The patient was started on adjuvant therapies. Two months later she returned to the clinic with slight ophthalmoparesis and visual field cut impairment, though much improved from her pre-operative status. The patient, however, expired 7 months after the surgical date. Orbit
Spontaneous Subperiosteal Orbital Hematoma 155
COMMENT
REFERENCES
The authors find this to be a unique initial manifestation of metastatic lung adenocarcinoma which has not been described previously in literature. We theorize that the cause of the sSOH was due to vascular congestion, microaneurysm, or vascular erosion from the metastasis as there was no macroscopic evidence of bony erosion nor malignant cells found in the cytological evaluation of the hematoma. A differential diagnosis of etiologies causing the formation of sSOH in an adult without history of trauma should include metastatic neoplasm to the skull and warrants metastatic workup. Furthermore we believe that surgical intervention of a sSOH would be the most beneficial for patients in similar circumstances as presented here, though observation may be an option for treatment.
1. Leovic´ D, Zubcˇic´ V, Kopic´ M, et al. Posttraumatic subperiosteal orbital hematoma. J Craniomaxillofac Surg 2011;39(2):131–134. 2. Russell DJ, Dutton JJ. Bilateral spontaneous subperiosteal orbital hemorrhages following endoscopic retrograde cholangiopancreatography. Ophthal Plast Reconstr Surg 2011;27(3):e49–e50. 3. Matsumoto S, Yamamoto T, Ban S, et al. Spontaneous subperiosteal hematoma of the orbit: Case report. Neurol Med Chir (Tokyo) 1994;34:27–29. 4. Crawford S, Patel M. Bilateral supraorbital masses after prolonged headlock injury an unusual manifestation of orbital subperiosteal hematomas. Am J Rad 2005;184: S2–S3. 5. Balasubramaniam S, Mahore A, Dange N. Spontaneous resolution of spontaneous subperiosteal orbital hematoma. Neurol India 2011;59(3):463–464. 6. Sugiura H, Yamada K, Sugiura T, et al. Predictors of survival in patients with bone metastasis of lung cancer. Clin Orthop Relat Res 2008;466(3):729–736. 7. Nemeth AJ, Henson JW, Mullins ME, et al. Improved detection of skull metastasis with diffusion-weighted MR imaging. AJNR Am J Neuroradiol 2007;28(6):1088–1092. 8. Kagohashi K, Satoh H, Ishikawa H, et al. Bone metastasis as the first manifestation of lung cancer. Int J Clin Pract 2003;57:184–186. 9. Mitsuya K, Nakasu Y, Horiguchi S, et al. Metastatic skull tumors: MRI features and a new conventional classification. J Neurooncol 2011;104(1):239–245.
DECLARATION OF INTEREST The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
!
2014 Informa Healthcare USA, Inc.
Copyright of Orbit is the property of Taylor & Francis Ltd and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
C ASE REPORT
Spontaneous Subperiosteal Orbital Hematoma as Initial Presentation of Metastatic Lung Adenocarcinoma to the Skull: Case Report John Kirk Capua1, Eric Scott Stiner2, and Tina Grace Li3 1
Department of Neurological Surgery, Arrowhead Regional Medical Center, Colton, California, USA, Department of Neurological Surgery, Kaiser Permanente - Fontana Medical Center, Fontana, California, USA, and 3Department of Ophthalmology, Kaiser Permanente - Fontana Medical Center, Fontana, California, USA
2
ABSTRACT Introduction: Subperiosteal orbital hematoma is a rare occurrence, typically developing as a result of orbital trauma. The spontaneous formation of a subperiosteal orbital hematoma (sSOH) may also occur but is less frequent. To date there has been no documented cases of sSOH as the initial presentation of an unknown metastatic neoplasm to the skull. We provide a case of a woman with unknown lung adenocarcinoma that metastasized to the skull which caused the formation of a sSOH resulting in orbital compression syndrome. Case Report: A 57-year-old female presented with double vision, retro-orbital right eye pain, and vision loss in the right eye. A magnetic resonance imaging revealed a right orbital compressive lesion with an adjacent supraorbital skull lesion and separate left frontal skull lesion. Intra-operative findings along with post-operative immunohistochemistry staining revealed sSOH resulting from a metastatic lung adenocarcinoma to the skull. Further metastatic work up also revealed an occult lung mass and multiple spinal lesions. Conclusion: Differential diagnosis of etiologies causing the formation of sSOH in an adult without history of trauma should include metastatic neoplasm to the skull and warrants metastatic workup. Treatment options of sSOH have included observation with spontaneous resolution; however, we opted for surgical decompression of the eye and biopsy of the skull mass. Keywords: Orbital compression, skull metastasis, spontaneous subperiosteal orbital hematoma
INTRODUCTION
20 14
The incidence of skull metastases is estimated to be 3%.6 There has been an increase in detection of skull metastasis as a result of advancement of imaging technology.7 The most common neoplasms that metastasize to the skull are breast, prostate, lung, melanoma, renal, and thyroid.6 Bone metastases are often clinically symptomatic and present as the first manifestation of malignancy in 2.3% of lung cancer patients.8 Life expectancy after skull metastases is estimated to be 5 months.9 We provide a case of a woman with unknown lung adenocarcinoma that metastasized to the skull which caused the formation of a sSOH resulting in orbital compression syndrome. To our knowledge and review
Subperiosteal orbital hematoma (SOH) is a rare entity, most often caused by trauma.1 Formation of SOH may also occur spontaneously (sSOH).2 The formation of sSOH may originate from blood dyscrasias, sinusitis, anti-thrombolytic agents, vascular malformations, and orbital venous congestion.3 Pathogenesis of sSOH is thought to occur from increased pressure of the orbital veins that lack valves.4 Surgical decompression is typically the treatment of choice for SOH in symptomatic patients, though conservative management with resolution of sSOH has been reported.4,5
Received 2 May 2013; Revised 30 September 2013; Accepted 7 October 2013; Published online 15 November 2013 Correspondence: Dr. John Kirk Capua, Department of Neurological Surgery, Arrowhead Regional Medical Center, 400 North Pepper Ave. Colton, Ca. 92324, USA. E-mail: [email protected]
152
Spontaneous Subperiosteal Orbital Hematoma 153 of the available literature, this is the first documented case of the initial presentation of a metastatic neoplasm to the skull being the formation of a sSOH.
CASE REPORT A 57-year-old Caucasian female presented to the emergency department with complaint of one week history of diplopia, retro-orbital eye pain, and vision loss in the right eye. She denied any headaches, scalp pain, nor any other complaints of pain throughout the rest of the body. There was no history of trauma. Her past medical and surgical histories were also unremarkable. She did report of having smoked tobacco for 20 plus years.
Physical examination revealed significant right eye proptosis, periorbital erythema, conjunctival edema, and ophthalmoparesis resulting in diplopia. Visual fields of the right eye were diminished. There were no palpable masses of the scalp. No recent fevers or infections. Lab results revealed mildly elevated erythrocyte sedimentation rate, a normal level of C-reactive protein, and normal leukocyte count. Liver function and coagulation levels were within normal limits. An MRI of the brain was then obtained and showed the following characteristics (Figures 1A-2D). The patient was taken for immediate decompression of the globe as well as biopsy of the supraorbital skull lesion. A supraorbital eye brow incision was made, to approach both the superior retrobulbar lesion and supraorbital skull lesion. Intraoperative
FIGURE 1. T1 post-GAD imaging with the retrobulbar mass enhancement on axial view (A). Coronal T1 post-GAD view also showing the enhancing retrobulbar mass (B). T1 post-GAD sagittal view verifying the right supraorbital rim enhancing skull lesion (C). The T1 post-GAD coronal view revealed a second enhancing skull mass in the left frontal region (D). !
2014 Informa Healthcare USA, Inc.
154 J. K. Capua et al.
FIGURE 2. DWI sequence shows the retrobulbar mass to by hypointense (A). The right supraorbital skull lesion is hyperintense (B). T2 axial views showing the isointense right supraorbital skull mass (C) and the hyperintense retrobulbar lesion (D).
findings revealed a nonpurulent darkened color fluid in the subperiosteal space. There was no bony erosion but discoloration of the outer table of the skull in the supraorbital region. Engorged vessels adjacent to the discolored area were present. Abnormal tissue was seen in the diploic space as well as erosion through the inner table of the skull. Involvement of the dura but not the adjacent cerebral parenchyma was identified. Post-operatively she regained partial movement of the extraocular muscles and her visual field cuts improved significantly. Final immunohistochemistry staining revealed the skull lesion to be
undifferentiated metastatic lung adenocarcinoma (TTF-1 and CK7 positive). The subperiosteal fluid collection contained no malignant cells and was comprised of fibrin clot and granulation tissue consistent with hematoma. Cultures were negative. Further metastatic work up revealed a small right lung mass and multiple thoracic vertebral body lesions. The patient was started on adjuvant therapies. Two months later she returned to the clinic with slight ophthalmoparesis and visual field cut impairment, though much improved from her pre-operative status. The patient, however, expired 7 months after the surgical date. Orbit
Spontaneous Subperiosteal Orbital Hematoma 155
COMMENT
REFERENCES
The authors find this to be a unique initial manifestation of metastatic lung adenocarcinoma which has not been described previously in literature. We theorize that the cause of the sSOH was due to vascular congestion, microaneurysm, or vascular erosion from the metastasis as there was no macroscopic evidence of bony erosion nor malignant cells found in the cytological evaluation of the hematoma. A differential diagnosis of etiologies causing the formation of sSOH in an adult without history of trauma should include metastatic neoplasm to the skull and warrants metastatic workup. Furthermore we believe that surgical intervention of a sSOH would be the most beneficial for patients in similar circumstances as presented here, though observation may be an option for treatment.
1. Leovic´ D, Zubcˇic´ V, Kopic´ M, et al. Posttraumatic subperiosteal orbital hematoma. J Craniomaxillofac Surg 2011;39(2):131–134. 2. Russell DJ, Dutton JJ. Bilateral spontaneous subperiosteal orbital hemorrhages following endoscopic retrograde cholangiopancreatography. Ophthal Plast Reconstr Surg 2011;27(3):e49–e50. 3. Matsumoto S, Yamamoto T, Ban S, et al. Spontaneous subperiosteal hematoma of the orbit: Case report. Neurol Med Chir (Tokyo) 1994;34:27–29. 4. Crawford S, Patel M. Bilateral supraorbital masses after prolonged headlock injury an unusual manifestation of orbital subperiosteal hematomas. Am J Rad 2005;184: S2–S3. 5. Balasubramaniam S, Mahore A, Dange N. Spontaneous resolution of spontaneous subperiosteal orbital hematoma. Neurol India 2011;59(3):463–464. 6. Sugiura H, Yamada K, Sugiura T, et al. Predictors of survival in patients with bone metastasis of lung cancer. Clin Orthop Relat Res 2008;466(3):729–736. 7. Nemeth AJ, Henson JW, Mullins ME, et al. Improved detection of skull metastasis with diffusion-weighted MR imaging. AJNR Am J Neuroradiol 2007;28(6):1088–1092. 8. Kagohashi K, Satoh H, Ishikawa H, et al. Bone metastasis as the first manifestation of lung cancer. Int J Clin Pract 2003;57:184–186. 9. Mitsuya K, Nakasu Y, Horiguchi S, et al. Metastatic skull tumors: MRI features and a new conventional classification. J Neurooncol 2011;104(1):239–245.
DECLARATION OF INTEREST The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
!
2014 Informa Healthcare USA, Inc.
Copyright of Orbit is the property of Taylor & Francis Ltd and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
