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Orbital lymphatic malformation showing the symptoms of orbital complications of acute rhinosinusitis in children: A report of 2 cases
International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case report
Orbital lymphatic malformation showing the symptoms of orbital complications of acute rhinosinusitis in children: A report of 2 cases A.P. Zimmermann *, B. Eivazi, S. Wiegand, J.A. Werner, A. Teymoortash Department of Otolaryngology, Head and Neck Surgery, Philipp University, Deutschhausstr. 3, 35037 Marburg, Germany
A R T I C L E I N F O
A B S T R A C T
Article history: Received 30 January 2009 Received in revised form 23 April 2009 Accepted 29 June 2009 Available online 30 July 2009
Orbital lymphatic malformations are benign cystic malformations of the lymphatic system. The present report shows two cases with symptoms of orbital complications of acute rhinosinusitis with proptosis, compressive optic neuropathy, loss of vision and cellulites in children. Magnetic resonance imaging (MRI) revealed a well-demarcated intraorbital mass with heterogeneous signal conformable with lymphatic malformation in both cases. A tumor extirpation was performed via lateral orbitotomy in both cases. Postoperatively the symptoms and especially the loss of vision improved completely. Histological analysis of the surgical specimens verified lymphatic malformations of the orbit. Orbital lymphatic malformations can mimic the symptoms of orbital complications of acute rhinosinusitis. The existence of lymphatic malformation should be considered in every orbital complication of rhinosinusitis in children. ß 2009 Elsevier Ireland Ltd. All rights reserved.
Keywords: Lymphatic malformation Acute rhinosinusitis Orbital complications
1. Introduction Lymphatic malformations are benign malformations of the lymphatic system. Morphologically, lymphatic malformations are composed of thin-walled, cystically dilated vascular channels lined by inconspicuous endothelial cells and filled with proteinaceous lymph fluid. Traditionally, lymphatic malformations are classified in analog to their histological appearance into capillary, cavernous, and cystic lymphatic malformations. This classification, however, is being replaced by a classification based on morphologic aspects into macrocystic, microcystic and combined lymphatic malformations [1]. Lymphatic malformations often grow proportionally to the patients’ body growth. Depending on the location of the lymphatic malformation, an acute increase in size that is frequently triggered by infectious diseases may lead to severe functional and aesthetic impairments for the patients as well as life-threatening complications [2]. Lymphatic malformations are equally distributed among genders and races. The incidence in children is estimated to 6% of all benign tumors. The disease is already obvious at the time of birth in 50% of the patients and diagnosed during the first and second year of life in 80–90% of the cases due to clinical symptoms [3]. About 60% of all lymphatic malformations are found in the head and neck region, primarily affecting the tongue and the floor of the mouth [4,5].
The nature of lymphatic malformations is uncertain, some are in continuity with the venous circulation, and rarely can they be associated with an arteriovenous malformation. As reasons for the development of lymphatic malformations a failure of the lymphatic system to connect with or separate from the venous system or an abnormal budding of lymphatic tissue from the cardinal vein are discussed [3]. Contrariwise there is the position that lymphatic malformations develop as a result of sequestration of portions of the primitive lymphatic predisposition [6]. Even traumata, infections, chronic inflammations, and obstructions during the embryonic development are suspected to trigger development of lymphatic malformations. Their infiltrative nature seems to suggest a neoplastic process although their lack of unremitting growth is strongly against it. Lymphatic malformations represent between 1% and 8% of all orbital masses [7]. In contrast to lymphatic malformations in general which have no predilection for sex lymphatic malformations of the orbit are slightly more common in females than in males, with gender ratio of 1.4:1 [8]. They usually present with a slowly progressive proptosis, displacement of the globe, ptosis, diplopia and restriction of eye movements. In the present study we demonstrate two cases of orbital lymphatic malformations which became symptomatic due to an orbital complication of acute rhinosinusitis. 2. Case presentation 2.1. Case 1
* Corresponding author. Tel.: +49 6421 5866478; fax: +49 6421 5866367. E-mail address: [email protected] (A.P. Zimmermann). 0165-5876/$ – see front matter ß 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2009.06.020
The case of a 15-year-old boy who was referred with a left-sided swelling of his eyelid and exophthalmia. He had suffered from
A.P. Zimmermann et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483
rhinosinusitis with fever, rhinorrhoea and headache for 4 days. There was also displacement of the globe, ptosis, and restriction of eye movements based on the acute rhinosinusitis for some days. There were no significant predisponding conditions such as allergy, asthma, or immunoglobulin deficience. Family and medical history was unremarkable (Fig. 1). The rhinological endoscopic examination revealed nasal pus, swelling and redness of the left eyelid as well as pain over all paranasal sinuses. Results of the ophthalmologic examination, including dilated funduscopy, revealed a loss of vision of the left eye to vision 0.4 (40%). The pupils were equally round and reactive to light without a relative afferent pupillary defect. An emergency radiological examination was performed. MRI of the sinuses revealed a supraorbital cystic bulbus displaced tumor and a shadow of the ethmoid sinuses (Fig. 2a and b). A conservative therapy with antibiotics (cefuroxime), corticosteroids, decongestant nasal spray and inhalation was started. Due to worsening of the symptoms with an acute loss of vision the mass was resected via lateral orbitotomy under general anaesthesia on the same day. The preoperative vision of the left side improved from 0.4 (40%) to postoperative 1.0 (100%) after 1 day. The diagnosis of an orbital lymphatic malformation was based on
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Fig. 1. Photography of the 15-year-old boy of case 1 demonstrating a left-sided swelling of his eyelid, exophthalmia, and purulent epiphora.
Fig. 2. (a and b) Contrast-enhanced sagittal (a) and coronar (b) T1-weighted MR images of the orbit of case 1 showing a cystic tumor of the left orbit. The orbital bulb is displaced downward. (c and d) Contrast-enhanced axial (c) and coronar (d) T1-weighted MR images of the orbit of case 2 showing a multicystic tumor of the right orbit. The orbital bulb is displaced forward.
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A.P. Zimmermann et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483
clinical presentation and radiological findings and was confirmed by histopathologic examination. 2.2. Case 2 The case of a 3-year-old girl who was referred with a rightsided progressive exophthalmia and fever and rhinitis for 1 day is reported. In her history there were no comparable medical conditions or a significant eye injury. There were no significant predisposing conditions such as allergy, asthma, or immunoglobulin deficience. Family and medical history was unremarkable. Rhinological examination revealed a displacement of the globe, ptosis, and restriction of eye movements. Additionally in nasal endoscopy there was a septum deviation and purulent rhinorrhoea. Ophthalmological examination showed a loss of vision (vision 0.6 = 60%) and an axial proptosis of the right eye, unremarkable fundi of both sides and regular intraocular eye pressure. MRI of the orbit was performed, which revealed an intraorbital, septated, cystic mass conformable with an infected lymphatic malformation of the right side and a partial obstruction of the ethmoidal and maxillary sinuses (Fig. 2c and d). In the first instance parenteral therapy with antibiotics (cefuroxime), corticosteroids, and local decongestant nose drops was started. Since there was a loss of vision during conservative therapy an operative intervention was performed. The mass was resected via lateral orbitotomy under general anaesthesia. The preoperative vision of the right side improved from 0.6 (60%) to postoperative 1.0 (100%) after 1 day. There was no restriction of eye movements in both eyes. The diagnosis of orbital lymphatic malformation was based on clinical presentation and radiological findings and was confirmed by histopathologic examination.
3. Discussion Acute rhinosinusitis is a common childhood infection. Children may have approximately six to eight viral infections of the upper respiratory tract each year, 5–13% of which may be complicated by a secondary bacterial infection of the paranasal sinuses [7]. The signs and symptoms of acute infection are protracted nasal discharge and congestion often accompanied by a persistent cough. The pathogens of a secondary bacterial infection of a viral sinusitis are usually streptococcus pneumoniae, haemophilus influenza, and moraxella catarrhalis [9,10]. Pediatric complications of acute rhinosinusitis can be divided into two categories, orbital and intracranial complications. One does not necessarily progress to the next, although this certainly can happen. Multiple complications can occur in the same patient [10]. Most of orbital cellulite cases are due to direct extension of acute or chronic bacterial sinusitis. Typically, infection begins in the ethmoid sinuses and can spread into the orbit. Any kind of orbital infection is most prevalent in children and adolescents because of the special anatomic conditions [11]. Spontaneous intraorbital haemorrhage due to the fragility of connecting veins and/or acute infection may cause acute proptosis, compressive optic neuropathy and loss of vision [12]. However, infections can also extend to the orbit haematogenously. Clinical findings of lymphatic malformations of the orbit like swelling of the eyelid and exophthalmia often resemble orbital complications of rhinosinusitis. Lymphatic malformations often show symptoms caused by inflammation. Orbital lymphatic malformations are typically multilocular and multicystic masses with well-defined margins that contain lymphatic fluid or haemorrhaged blood.
When considering the management of a child with a periorbital infection, features from the history and examination such as trauma, medical co-morbidities and ophthalmic signs will guide management and delineate the indications for early CT scan or MRI. In the absence of acute visual compromise or other signs of disease progression, initial management with intravenous antibiotics, corticosteroids and nasal decongestant should be considered before surgical intervention is contemplated. An ophthalmologist should perform a thorough ocular evaluation to rule out the visual compromise. If there is no improvement of the symptoms or a loss of vision an operative intervention has to be performed. Orbital complications are a therapeutic challenge to the practicing ENT surgeon. Immediate attention is critical. Interdisciplinary approach is necessary to achieve an optimal outcome for the patient. A multi-team approach is essential to obtain the best outcome for the child [13]. Acute orbital problems represent a demanding challenge for an interdisciplinary cooperation between ENT specialists, pediatricians, radiologists, and ophthalmologists. Early imaging is critical to establish diagnosis, and if available, MRI should be used owing to its superior sensitivity in differentiating the origin of orbital inflammation [14]. The two cases of the present study demonstrate that lateral orbitotomy constitutes a safe and fast surgical procedure for tumors of the lateral part of the orbit. If the lymphatic malformation is located in the medial part of the orbit the treatment of choice is an endonasal approach. There are also some reports in literature which demonstrate good results for macrocystic extraorbital lymphatic malformations for example in eyelid without acute complications treated with the injection of the sclerosing OK-432, a lyophilized mixture of streptococcus pyogenes which was incubated with penicillin G [15]. Since OK432 causes intense swelling during the first days after injection it should never be used for intraorbital lesions because of the risk of blindness. In conclusion, in spite of the clinical findings and outward aspect, the existence of a lymphatic malformation should be considered in every orbital complication of rhinosinusitis in children. Therefore MRI has to be performed in those cases to exclude a lymphatic malformation [16]. References [1] D.C. Bloom, J.A. Perkins, S.C. Manning, Management of lymphatic malformations, Curr. Opin. Otolaryngol. Head Neck Surg. 12 (2004) 500–504. [2] S. Wiegand, B. Eivazi, P.J. Barth, D. Berens von Rautenfeld, B.J. Folz, R. Mandic, et al., Pathogenesis of lymphatic malformations, Virchows Arch. 453 (2008) 1–8. [3] S. Wiegand, B. Eivazi, S. Sel, H. Renz, J.A. Werner, B.J. Folz, Analysis of cytokine levels in human lymphatic malformations, In Vivo 22 (2) (2008) 253–256. [4] B. Eivazi, M. Ardelean, W. Ba¨umler, H.P. Berlien, H. Cremer, R. Elluru, et al., Update on hemangiomas and vascular malformations of the head and neck, Eur. Arch. Otorhinolaryngol. 266 (2) (2009) 187–197. [5] J.A. Werner, A.A. Du¨nne, B.J. Folz, R. Rochels, S. Bien, A. Ramaswamy, et al., Current concepts in the classification, diagnosis and treatment of hemangiomas and vascular malformation of the head and neck, Eur. Arch. Otorhinolaryngol. 258 (2001) 141–149. [6] K.M. Laurence, Congenital cystic pulmonary lymphangioectasis, J. Pathol. Bacteriol. 70 (1955) 325–333. [7] A.D. Malhotra, M. Parikh, D.C. Garibaldi, S.L. Merbs, N.R. Miller, K. Murphy, Resection of an orbital lymphatic malformation with the aid of an intralesional liquid polymer, Am. J. Neurorad. 26 (2005) 2630–2634. ¨ zu¨m, H. Erdogan, A. Aslan, Orbital lymphatic malformation and its [8] M. Gu¨relik, U. O association with intracranial venous angioma, Br. J. Neurosurg. 18 (2004) 168– 170. [9] W.S. Russel, Chronic sinusitis in children, Clin. Pediatr. 44 (2005) 465–471. [10] N. Bhattacharyya, D.T. Jones, M. Hill, N.L. Shapiro, The diagnostic accuracy of computed tomography in pediatric chronic rhinosinusitis, Arch. Otolaryngol. Head Neck Surg. 130 (2004) 1029–1032. [11] L.T. Bilaniuk, Vascular lesions of the orbit in children, Neuroimaging Clin. N. Am. 15 (2005) 107–120. [12] D. McIntosh, M. Mahadevan, Acute orbital complications of sinusitis: the benefits of magnetic resonance imaging, J. Laryngol. Otol. 2 (2007) 1–3.
A.P. Zimmermann et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483 [13] A.M. Botting, D. McIntosh, M. Mahadevan, Paediatric pre- and post-septal periorbital infections are different diseases, a retrospective review of 262 cases, Int. J. Pediatr. Otorhinolaryngol. 72 (3) (2008) 377–383. [14] G. Hendriksson, K.M. Westrin, J. Kumlien, P. Stierna, A 13-year report on childhood sinusitis: clinical presentations, predisposing factors and possible means of prevention, Rhinology 34 (1996) 171–175.
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[15] S. Ogita, T. Tsuto, K. Tokiwa, T. Takahashi, Intracystic injection of OK-432: a new scleosing therapy for cystic hygroma in children, Br. J. Surg. 74 (1987) 690–691. [16] A. Bisdorff, J.B. Mulliken, J. Carrico, R.L. Robertson, P.E. Burrows, Intracranial vascular anomalies in patients with periorbital lymphatic and lymphaticovenous malformations, Am. J. Neurorad. 28 (2007) 335–341.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case report
Orbital lymphatic malformation showing the symptoms of orbital complications of acute rhinosinusitis in children: A report of 2 cases A.P. Zimmermann *, B. Eivazi, S. Wiegand, J.A. Werner, A. Teymoortash Department of Otolaryngology, Head and Neck Surgery, Philipp University, Deutschhausstr. 3, 35037 Marburg, Germany
A R T I C L E I N F O
A B S T R A C T
Article history: Received 30 January 2009 Received in revised form 23 April 2009 Accepted 29 June 2009 Available online 30 July 2009
Orbital lymphatic malformations are benign cystic malformations of the lymphatic system. The present report shows two cases with symptoms of orbital complications of acute rhinosinusitis with proptosis, compressive optic neuropathy, loss of vision and cellulites in children. Magnetic resonance imaging (MRI) revealed a well-demarcated intraorbital mass with heterogeneous signal conformable with lymphatic malformation in both cases. A tumor extirpation was performed via lateral orbitotomy in both cases. Postoperatively the symptoms and especially the loss of vision improved completely. Histological analysis of the surgical specimens verified lymphatic malformations of the orbit. Orbital lymphatic malformations can mimic the symptoms of orbital complications of acute rhinosinusitis. The existence of lymphatic malformation should be considered in every orbital complication of rhinosinusitis in children. ß 2009 Elsevier Ireland Ltd. All rights reserved.
Keywords: Lymphatic malformation Acute rhinosinusitis Orbital complications
1. Introduction Lymphatic malformations are benign malformations of the lymphatic system. Morphologically, lymphatic malformations are composed of thin-walled, cystically dilated vascular channels lined by inconspicuous endothelial cells and filled with proteinaceous lymph fluid. Traditionally, lymphatic malformations are classified in analog to their histological appearance into capillary, cavernous, and cystic lymphatic malformations. This classification, however, is being replaced by a classification based on morphologic aspects into macrocystic, microcystic and combined lymphatic malformations [1]. Lymphatic malformations often grow proportionally to the patients’ body growth. Depending on the location of the lymphatic malformation, an acute increase in size that is frequently triggered by infectious diseases may lead to severe functional and aesthetic impairments for the patients as well as life-threatening complications [2]. Lymphatic malformations are equally distributed among genders and races. The incidence in children is estimated to 6% of all benign tumors. The disease is already obvious at the time of birth in 50% of the patients and diagnosed during the first and second year of life in 80–90% of the cases due to clinical symptoms [3]. About 60% of all lymphatic malformations are found in the head and neck region, primarily affecting the tongue and the floor of the mouth [4,5].
The nature of lymphatic malformations is uncertain, some are in continuity with the venous circulation, and rarely can they be associated with an arteriovenous malformation. As reasons for the development of lymphatic malformations a failure of the lymphatic system to connect with or separate from the venous system or an abnormal budding of lymphatic tissue from the cardinal vein are discussed [3]. Contrariwise there is the position that lymphatic malformations develop as a result of sequestration of portions of the primitive lymphatic predisposition [6]. Even traumata, infections, chronic inflammations, and obstructions during the embryonic development are suspected to trigger development of lymphatic malformations. Their infiltrative nature seems to suggest a neoplastic process although their lack of unremitting growth is strongly against it. Lymphatic malformations represent between 1% and 8% of all orbital masses [7]. In contrast to lymphatic malformations in general which have no predilection for sex lymphatic malformations of the orbit are slightly more common in females than in males, with gender ratio of 1.4:1 [8]. They usually present with a slowly progressive proptosis, displacement of the globe, ptosis, diplopia and restriction of eye movements. In the present study we demonstrate two cases of orbital lymphatic malformations which became symptomatic due to an orbital complication of acute rhinosinusitis. 2. Case presentation 2.1. Case 1
* Corresponding author. Tel.: +49 6421 5866478; fax: +49 6421 5866367. E-mail address: [email protected] (A.P. Zimmermann). 0165-5876/$ – see front matter ß 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2009.06.020
The case of a 15-year-old boy who was referred with a left-sided swelling of his eyelid and exophthalmia. He had suffered from
A.P. Zimmermann et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483
rhinosinusitis with fever, rhinorrhoea and headache for 4 days. There was also displacement of the globe, ptosis, and restriction of eye movements based on the acute rhinosinusitis for some days. There were no significant predisponding conditions such as allergy, asthma, or immunoglobulin deficience. Family and medical history was unremarkable (Fig. 1). The rhinological endoscopic examination revealed nasal pus, swelling and redness of the left eyelid as well as pain over all paranasal sinuses. Results of the ophthalmologic examination, including dilated funduscopy, revealed a loss of vision of the left eye to vision 0.4 (40%). The pupils were equally round and reactive to light without a relative afferent pupillary defect. An emergency radiological examination was performed. MRI of the sinuses revealed a supraorbital cystic bulbus displaced tumor and a shadow of the ethmoid sinuses (Fig. 2a and b). A conservative therapy with antibiotics (cefuroxime), corticosteroids, decongestant nasal spray and inhalation was started. Due to worsening of the symptoms with an acute loss of vision the mass was resected via lateral orbitotomy under general anaesthesia on the same day. The preoperative vision of the left side improved from 0.4 (40%) to postoperative 1.0 (100%) after 1 day. The diagnosis of an orbital lymphatic malformation was based on
1481
Fig. 1. Photography of the 15-year-old boy of case 1 demonstrating a left-sided swelling of his eyelid, exophthalmia, and purulent epiphora.
Fig. 2. (a and b) Contrast-enhanced sagittal (a) and coronar (b) T1-weighted MR images of the orbit of case 1 showing a cystic tumor of the left orbit. The orbital bulb is displaced downward. (c and d) Contrast-enhanced axial (c) and coronar (d) T1-weighted MR images of the orbit of case 2 showing a multicystic tumor of the right orbit. The orbital bulb is displaced forward.
1482
A.P. Zimmermann et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483
clinical presentation and radiological findings and was confirmed by histopathologic examination. 2.2. Case 2 The case of a 3-year-old girl who was referred with a rightsided progressive exophthalmia and fever and rhinitis for 1 day is reported. In her history there were no comparable medical conditions or a significant eye injury. There were no significant predisposing conditions such as allergy, asthma, or immunoglobulin deficience. Family and medical history was unremarkable. Rhinological examination revealed a displacement of the globe, ptosis, and restriction of eye movements. Additionally in nasal endoscopy there was a septum deviation and purulent rhinorrhoea. Ophthalmological examination showed a loss of vision (vision 0.6 = 60%) and an axial proptosis of the right eye, unremarkable fundi of both sides and regular intraocular eye pressure. MRI of the orbit was performed, which revealed an intraorbital, septated, cystic mass conformable with an infected lymphatic malformation of the right side and a partial obstruction of the ethmoidal and maxillary sinuses (Fig. 2c and d). In the first instance parenteral therapy with antibiotics (cefuroxime), corticosteroids, and local decongestant nose drops was started. Since there was a loss of vision during conservative therapy an operative intervention was performed. The mass was resected via lateral orbitotomy under general anaesthesia. The preoperative vision of the right side improved from 0.6 (60%) to postoperative 1.0 (100%) after 1 day. There was no restriction of eye movements in both eyes. The diagnosis of orbital lymphatic malformation was based on clinical presentation and radiological findings and was confirmed by histopathologic examination.
3. Discussion Acute rhinosinusitis is a common childhood infection. Children may have approximately six to eight viral infections of the upper respiratory tract each year, 5–13% of which may be complicated by a secondary bacterial infection of the paranasal sinuses [7]. The signs and symptoms of acute infection are protracted nasal discharge and congestion often accompanied by a persistent cough. The pathogens of a secondary bacterial infection of a viral sinusitis are usually streptococcus pneumoniae, haemophilus influenza, and moraxella catarrhalis [9,10]. Pediatric complications of acute rhinosinusitis can be divided into two categories, orbital and intracranial complications. One does not necessarily progress to the next, although this certainly can happen. Multiple complications can occur in the same patient [10]. Most of orbital cellulite cases are due to direct extension of acute or chronic bacterial sinusitis. Typically, infection begins in the ethmoid sinuses and can spread into the orbit. Any kind of orbital infection is most prevalent in children and adolescents because of the special anatomic conditions [11]. Spontaneous intraorbital haemorrhage due to the fragility of connecting veins and/or acute infection may cause acute proptosis, compressive optic neuropathy and loss of vision [12]. However, infections can also extend to the orbit haematogenously. Clinical findings of lymphatic malformations of the orbit like swelling of the eyelid and exophthalmia often resemble orbital complications of rhinosinusitis. Lymphatic malformations often show symptoms caused by inflammation. Orbital lymphatic malformations are typically multilocular and multicystic masses with well-defined margins that contain lymphatic fluid or haemorrhaged blood.
When considering the management of a child with a periorbital infection, features from the history and examination such as trauma, medical co-morbidities and ophthalmic signs will guide management and delineate the indications for early CT scan or MRI. In the absence of acute visual compromise or other signs of disease progression, initial management with intravenous antibiotics, corticosteroids and nasal decongestant should be considered before surgical intervention is contemplated. An ophthalmologist should perform a thorough ocular evaluation to rule out the visual compromise. If there is no improvement of the symptoms or a loss of vision an operative intervention has to be performed. Orbital complications are a therapeutic challenge to the practicing ENT surgeon. Immediate attention is critical. Interdisciplinary approach is necessary to achieve an optimal outcome for the patient. A multi-team approach is essential to obtain the best outcome for the child [13]. Acute orbital problems represent a demanding challenge for an interdisciplinary cooperation between ENT specialists, pediatricians, radiologists, and ophthalmologists. Early imaging is critical to establish diagnosis, and if available, MRI should be used owing to its superior sensitivity in differentiating the origin of orbital inflammation [14]. The two cases of the present study demonstrate that lateral orbitotomy constitutes a safe and fast surgical procedure for tumors of the lateral part of the orbit. If the lymphatic malformation is located in the medial part of the orbit the treatment of choice is an endonasal approach. There are also some reports in literature which demonstrate good results for macrocystic extraorbital lymphatic malformations for example in eyelid without acute complications treated with the injection of the sclerosing OK-432, a lyophilized mixture of streptococcus pyogenes which was incubated with penicillin G [15]. Since OK432 causes intense swelling during the first days after injection it should never be used for intraorbital lesions because of the risk of blindness. In conclusion, in spite of the clinical findings and outward aspect, the existence of a lymphatic malformation should be considered in every orbital complication of rhinosinusitis in children. Therefore MRI has to be performed in those cases to exclude a lymphatic malformation [16]. References [1] D.C. Bloom, J.A. Perkins, S.C. Manning, Management of lymphatic malformations, Curr. Opin. Otolaryngol. Head Neck Surg. 12 (2004) 500–504. [2] S. Wiegand, B. Eivazi, P.J. Barth, D. Berens von Rautenfeld, B.J. Folz, R. Mandic, et al., Pathogenesis of lymphatic malformations, Virchows Arch. 453 (2008) 1–8. [3] S. Wiegand, B. Eivazi, S. Sel, H. Renz, J.A. Werner, B.J. Folz, Analysis of cytokine levels in human lymphatic malformations, In Vivo 22 (2) (2008) 253–256. [4] B. Eivazi, M. Ardelean, W. Ba¨umler, H.P. Berlien, H. Cremer, R. Elluru, et al., Update on hemangiomas and vascular malformations of the head and neck, Eur. Arch. Otorhinolaryngol. 266 (2) (2009) 187–197. [5] J.A. Werner, A.A. Du¨nne, B.J. Folz, R. Rochels, S. Bien, A. Ramaswamy, et al., Current concepts in the classification, diagnosis and treatment of hemangiomas and vascular malformation of the head and neck, Eur. Arch. Otorhinolaryngol. 258 (2001) 141–149. [6] K.M. Laurence, Congenital cystic pulmonary lymphangioectasis, J. Pathol. Bacteriol. 70 (1955) 325–333. [7] A.D. Malhotra, M. Parikh, D.C. Garibaldi, S.L. Merbs, N.R. Miller, K. Murphy, Resection of an orbital lymphatic malformation with the aid of an intralesional liquid polymer, Am. J. Neurorad. 26 (2005) 2630–2634. ¨ zu¨m, H. Erdogan, A. Aslan, Orbital lymphatic malformation and its [8] M. Gu¨relik, U. O association with intracranial venous angioma, Br. J. Neurosurg. 18 (2004) 168– 170. [9] W.S. Russel, Chronic sinusitis in children, Clin. Pediatr. 44 (2005) 465–471. [10] N. Bhattacharyya, D.T. Jones, M. Hill, N.L. Shapiro, The diagnostic accuracy of computed tomography in pediatric chronic rhinosinusitis, Arch. Otolaryngol. Head Neck Surg. 130 (2004) 1029–1032. [11] L.T. Bilaniuk, Vascular lesions of the orbit in children, Neuroimaging Clin. N. Am. 15 (2005) 107–120. [12] D. McIntosh, M. Mahadevan, Acute orbital complications of sinusitis: the benefits of magnetic resonance imaging, J. Laryngol. Otol. 2 (2007) 1–3.
A.P. Zimmermann et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1480–1483 [13] A.M. Botting, D. McIntosh, M. Mahadevan, Paediatric pre- and post-septal periorbital infections are different diseases, a retrospective review of 262 cases, Int. J. Pediatr. Otorhinolaryngol. 72 (3) (2008) 377–383. [14] G. Hendriksson, K.M. Westrin, J. Kumlien, P. Stierna, A 13-year report on childhood sinusitis: clinical presentations, predisposing factors and possible means of prevention, Rhinology 34 (1996) 171–175.
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[15] S. Ogita, T. Tsuto, K. Tokiwa, T. Takahashi, Intracystic injection of OK-432: a new scleosing therapy for cystic hygroma in children, Br. J. Surg. 74 (1987) 690–691. [16] A. Bisdorff, J.B. Mulliken, J. Carrico, R.L. Robertson, P.E. Burrows, Intracranial vascular anomalies in patients with periorbital lymphatic and lymphaticovenous malformations, Am. J. Neurorad. 28 (2007) 335–341.