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Successful treatment of intraorbital lymphangioma with tissue fibrin glue
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Surgical Neurology 72 (2009) 722 – 724 www.surgicalneurology-online.com
Neoplasm
Successful treatment of intraorbital lymphangioma with tissue fibrin glue Aiko Hayasaki, MD, Hideo Nakamura, MD, PhD⁎, Tadashi Hamasaki, MD, PhD, Keishi Makino, MD, PhD, Shigetoshi Yano, MD, PhD, Motohiro Morioka, MD, PhD, Jun-ichi Kuratsu, MD, PhD Department of Neurosurgery, Kumamoto University Medical School, Kumamoto 860-8556, Japan Received 4 March 2009; accepted 13 April 2009
Abstract
Background: Although surgical resection is the first treatment choice in patients with cystic lymphangioma, the complete resection of orbital lymphangioma is often difficult. After partial resection of the cyst wall, some cystic lymphangiomas recur. The injection of tissue fibrin glue may prevent the recurrence of orbital lymphangioma. Case Description: We present a 2-year-old girl with left progressive exophthalmos. Magnetic resonance imaging revealed a cystic mass lesion behind the left eyeball. At the first operation, the cyst wall was partially resected, and all cyst fluid was totally removed by suction. One week after the first operation, the cyst showed regrowth. At a second procedure, we injected tissue fibrin glue into the cyst. The cyst was completely sealed, and there was no recurrence. Conclusion: Tissue fibrin glue is adhesive and hemostatic and highly useful in the treatment of orbital cystic lymphangioma. © 2009 Elsevier Inc. All rights reserved.
Keywords:
Lymphangioma; Orbita; Tissue fibrin glue
1. Introduction The natural course of lymphangioma remains unclear, some lymphangiomas are contiguous with the venous circulation, and they are rarely associated with arteriovenous malformation [2,5]. The management of orbital lymphangioma can be challenging. Although some instances of successful surgical excision have been reported [3], the complete resection of lymphangiomas is difficult due to their diffuse infiltration and intertwining with adjacent tissues. We report a 2-year-old girl with orbital lymphangioma who was successfully treated by surgical resection of the cyst wall followed by the intralesional injection of tissue fibrin glue [1]. 2. Case report This 2-year-old girl presented with a 3-week history of progressive left exophthalmos and upper eyelid swelling. Abbreviation: MRI, magnetic resonance imaging. ⁎ Corresponding author. Tel.: +81 96 373 5219; fax: +81 96 371 8064. E-mail address: [email protected] (H. Nakamura). 0090-3019/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.surneu.2009.04.013
Fig. 1. Pretreatment photographs. A: Note left ocular proptosis and congestion of the left eyelid. B: The left conjunctiva is congested.
A. Hayasaki et al. / Surgical Neurology 72 (2009) 722–724
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Fig. 2. Magnetic resonance imaging scans obtained before (A-C) and 1 week after the first operation (D). A: T2-weighted image. B and C: Gadolinium-enhanced T1-weighted images. Note the cystic lesion behind the eyeball. D: T2-weighted image after the first operation. Note the reappearance of the multicystic lesion with niveau.
The visual acuity of her left eye had rapidly decreased for the preceding 2 weeks. The left pupil was dilated and without light reflex. The left eyeball was dislocated upward, and ocular mobility was severely restricted (Fig. 1). Orbital contrast-enhanced magnetic resonance imaging (MRI) showed several multilobular intraorbital cysts; some of these manifested a niveaulike pattern suggestive of recent intracystic bleeding (Figs. 2A-C). She underwent frontal craniotomy, and using an upper orbital wall approach, we punctured the intraorbital cyst and drained its xanthochromic fluid content. We also partially resected the cyst wall. Immediately after the operation, her exophthalmos and dilation of the left pupil were improved. However, within a week, her exophthalmos worsened again, and orbital MRI showed regrowth of intraorbital cysts (Fig. 2D). A second operation was performed via frontal craniotomy, the cysts were punctured and drained again, the wall of the cysts was resected as much as possible to render the intracystic space contiguous, and tissue fibrin glue was injected. After the second operation, her exophthalmos was improved. Orbital MRI showed no apparent intraorbital lesions (Fig. 3). Although immediately after the second operation there was some residual dislocation of the left eyeball and eye movement
remained slightly restricted (Fig. 4), there was complete recovery after a few weeks. 3. Discussion Surgical resection is the first treatment choice in patients with cystic lymphangioma; however, complete resection can be difficult if the cysts are located in the orbital region. Tunç et al [4] reported that 15 (58%) of 26 orbital lymphangiomas recurred. A goal of the treatment of cystic lymphangioma is cyst shrinkage, and sclerosing agents have been injected into the lesions. Yoon et al [6], who reported the successful use of OK-432 in patients with orbital lymphangioma, suggested that this agent was useful for the treatment of cystic lymphangioma and produced minimal adverse effects. However, their patients experienced low-grade fever for a few days and local inflammation. We injected tissue fibrin glue, not a sclerosing agent, and our patient experienced neither fever nor inflammation. We suggest that precise cyst puncture, cystic fluid aspiration, and the injection of fibrin glue are useful for the treatment of orbital lymphangioma. It is important that, in patients with multiple cysts, cysts should be combined into one cavity to be filled with tissue fibrin glue.
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A. Hayasaki et al. / Surgical Neurology 72 (2009) 722–724
Fig. 3. Magnetic resonance imaging scan obtained after the second operation. A: T2-weighted image. B and C: Gadolinium-enhanced T1-weighted image. Note complete shrinkage of the cystic lesions.
Fig. 4. Photograph obtained just after the second operation. There is slight dislocation of the left eyeball; however, there is no proptosis.
References [1] Boulos PR, Harissi-Dagher M, Kavalec C, et al. Intralesional injection of Tisseel fibrin glue for resection of lymphangiomas and other thinwalled orbital cysts. Ophthal Plast Reconstr Surg 2005;21:171-6. [2] Garrity JA. Orbital venous anomalies. A long-standing dilemma. Ophthalmology 1997;104:903-4. [3] Hemmer KM, Marsh JL, Milder B. Orbital lymphangioma. Plast Reconstr Surg 1988;82:340-3. [4] Tunc M, Sadri E, Char DH. Orbital lymphangioma: an analysis of 26 patients. Br J Ophthalmol 1999;83:76-80. [5] Wright JE, Sullivan TJ, Garner A, et al. Orbital venous anomalies. Ophthalmology 1997;104:905-13. [6] Yoon JS, Choi JB, Kim SJ, Lee SY. Intralesional injection of OK-432 for vision-threatening orbital lymphangioma. Graefes Arch Clin Exp Ophthalmol 2007;245:1031-5.
Commentary Although rare in the practice of most neurosurgeons, this lesion is extremely difficult to eradicate and has a definite propensity for recurrence. The technique of injecting fibrin glue into residual cysts may have some benefit as suggested by the authors. Obviously, this one case does not provide proof positive, but it is an interesting consideration to keep in mind when operating on such a lesion. Joseph C. Maroon, MD Tri-State Neurosurgical Associates-UPMC Pittsburgh PA 15213, USA
Surgical Neurology 72 (2009) 722 – 724 www.surgicalneurology-online.com
Neoplasm
Successful treatment of intraorbital lymphangioma with tissue fibrin glue Aiko Hayasaki, MD, Hideo Nakamura, MD, PhD⁎, Tadashi Hamasaki, MD, PhD, Keishi Makino, MD, PhD, Shigetoshi Yano, MD, PhD, Motohiro Morioka, MD, PhD, Jun-ichi Kuratsu, MD, PhD Department of Neurosurgery, Kumamoto University Medical School, Kumamoto 860-8556, Japan Received 4 March 2009; accepted 13 April 2009
Abstract
Background: Although surgical resection is the first treatment choice in patients with cystic lymphangioma, the complete resection of orbital lymphangioma is often difficult. After partial resection of the cyst wall, some cystic lymphangiomas recur. The injection of tissue fibrin glue may prevent the recurrence of orbital lymphangioma. Case Description: We present a 2-year-old girl with left progressive exophthalmos. Magnetic resonance imaging revealed a cystic mass lesion behind the left eyeball. At the first operation, the cyst wall was partially resected, and all cyst fluid was totally removed by suction. One week after the first operation, the cyst showed regrowth. At a second procedure, we injected tissue fibrin glue into the cyst. The cyst was completely sealed, and there was no recurrence. Conclusion: Tissue fibrin glue is adhesive and hemostatic and highly useful in the treatment of orbital cystic lymphangioma. © 2009 Elsevier Inc. All rights reserved.
Keywords:
Lymphangioma; Orbita; Tissue fibrin glue
1. Introduction The natural course of lymphangioma remains unclear, some lymphangiomas are contiguous with the venous circulation, and they are rarely associated with arteriovenous malformation [2,5]. The management of orbital lymphangioma can be challenging. Although some instances of successful surgical excision have been reported [3], the complete resection of lymphangiomas is difficult due to their diffuse infiltration and intertwining with adjacent tissues. We report a 2-year-old girl with orbital lymphangioma who was successfully treated by surgical resection of the cyst wall followed by the intralesional injection of tissue fibrin glue [1]. 2. Case report This 2-year-old girl presented with a 3-week history of progressive left exophthalmos and upper eyelid swelling. Abbreviation: MRI, magnetic resonance imaging. ⁎ Corresponding author. Tel.: +81 96 373 5219; fax: +81 96 371 8064. E-mail address: [email protected] (H. Nakamura). 0090-3019/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.surneu.2009.04.013
Fig. 1. Pretreatment photographs. A: Note left ocular proptosis and congestion of the left eyelid. B: The left conjunctiva is congested.
A. Hayasaki et al. / Surgical Neurology 72 (2009) 722–724
723
Fig. 2. Magnetic resonance imaging scans obtained before (A-C) and 1 week after the first operation (D). A: T2-weighted image. B and C: Gadolinium-enhanced T1-weighted images. Note the cystic lesion behind the eyeball. D: T2-weighted image after the first operation. Note the reappearance of the multicystic lesion with niveau.
The visual acuity of her left eye had rapidly decreased for the preceding 2 weeks. The left pupil was dilated and without light reflex. The left eyeball was dislocated upward, and ocular mobility was severely restricted (Fig. 1). Orbital contrast-enhanced magnetic resonance imaging (MRI) showed several multilobular intraorbital cysts; some of these manifested a niveaulike pattern suggestive of recent intracystic bleeding (Figs. 2A-C). She underwent frontal craniotomy, and using an upper orbital wall approach, we punctured the intraorbital cyst and drained its xanthochromic fluid content. We also partially resected the cyst wall. Immediately after the operation, her exophthalmos and dilation of the left pupil were improved. However, within a week, her exophthalmos worsened again, and orbital MRI showed regrowth of intraorbital cysts (Fig. 2D). A second operation was performed via frontal craniotomy, the cysts were punctured and drained again, the wall of the cysts was resected as much as possible to render the intracystic space contiguous, and tissue fibrin glue was injected. After the second operation, her exophthalmos was improved. Orbital MRI showed no apparent intraorbital lesions (Fig. 3). Although immediately after the second operation there was some residual dislocation of the left eyeball and eye movement
remained slightly restricted (Fig. 4), there was complete recovery after a few weeks. 3. Discussion Surgical resection is the first treatment choice in patients with cystic lymphangioma; however, complete resection can be difficult if the cysts are located in the orbital region. Tunç et al [4] reported that 15 (58%) of 26 orbital lymphangiomas recurred. A goal of the treatment of cystic lymphangioma is cyst shrinkage, and sclerosing agents have been injected into the lesions. Yoon et al [6], who reported the successful use of OK-432 in patients with orbital lymphangioma, suggested that this agent was useful for the treatment of cystic lymphangioma and produced minimal adverse effects. However, their patients experienced low-grade fever for a few days and local inflammation. We injected tissue fibrin glue, not a sclerosing agent, and our patient experienced neither fever nor inflammation. We suggest that precise cyst puncture, cystic fluid aspiration, and the injection of fibrin glue are useful for the treatment of orbital lymphangioma. It is important that, in patients with multiple cysts, cysts should be combined into one cavity to be filled with tissue fibrin glue.
724
A. Hayasaki et al. / Surgical Neurology 72 (2009) 722–724
Fig. 3. Magnetic resonance imaging scan obtained after the second operation. A: T2-weighted image. B and C: Gadolinium-enhanced T1-weighted image. Note complete shrinkage of the cystic lesions.
Fig. 4. Photograph obtained just after the second operation. There is slight dislocation of the left eyeball; however, there is no proptosis.
References [1] Boulos PR, Harissi-Dagher M, Kavalec C, et al. Intralesional injection of Tisseel fibrin glue for resection of lymphangiomas and other thinwalled orbital cysts. Ophthal Plast Reconstr Surg 2005;21:171-6. [2] Garrity JA. Orbital venous anomalies. A long-standing dilemma. Ophthalmology 1997;104:903-4. [3] Hemmer KM, Marsh JL, Milder B. Orbital lymphangioma. Plast Reconstr Surg 1988;82:340-3. [4] Tunc M, Sadri E, Char DH. Orbital lymphangioma: an analysis of 26 patients. Br J Ophthalmol 1999;83:76-80. [5] Wright JE, Sullivan TJ, Garner A, et al. Orbital venous anomalies. Ophthalmology 1997;104:905-13. [6] Yoon JS, Choi JB, Kim SJ, Lee SY. Intralesional injection of OK-432 for vision-threatening orbital lymphangioma. Graefes Arch Clin Exp Ophthalmol 2007;245:1031-5.
Commentary Although rare in the practice of most neurosurgeons, this lesion is extremely difficult to eradicate and has a definite propensity for recurrence. The technique of injecting fibrin glue into residual cysts may have some benefit as suggested by the authors. Obviously, this one case does not provide proof positive, but it is an interesting consideration to keep in mind when operating on such a lesion. Joseph C. Maroon, MD Tri-State Neurosurgical Associates-UPMC Pittsburgh PA 15213, USA