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“Water-lily sign” as a rare finding of spinal hydatidosis
The Spine Journal 12 (2012) e5–e7
Case Report
‘‘Water-lily sign’’ as a rare finding of spinal hydatidosis Sertan Gezgin, MD*, Hatice T. Sanal, MD Department of Radiology, Gulhane Military Medical Academy, Gn.Tevfik Saglam Cad., 06018 Kecioren, Ankara, Turkey Received 24 April 2011; revised 13 September 2011; accepted 17 May 2012
Abstract
Hydatid disease (HD) is a common parasitic infestation in some developing countries. Hydatid disease may affect many organs in the body, but the most affected are the liver and lungs. Hepatic and pulmonary HD is relatively easier to diagnose because of the frequency of pathognomic features and its order in the differential diagnosis list. When HD is located in uncommon sites, it is difficult to recognize even in endemic areas. The ‘‘water-lily sign’’ is a pathognomic sign for HD and mostly described especially after puncture or percutaneous treatment of the cyst. In this case, we observed the water-lily sign in spinal HD, which was not described before for spinal cases but for soft tissues. Ó 2012 Elsevier Inc. All rights reserved.
Keywords:
Hydatid disease; Water-lily sign; Spinal cyst hydatid
Introduction
Case report
Hydatid disease (HD) is an infestation mostly caused by the larvas of Echinococcus granulosus. Hydatid disease is endemic in places such as Central Asia, southern and eastern Mediterranean countries, East Africa, Australia, New Zealand, and South America. The liver and lungs are the most affected organs, but other parts of the body such as kidney, brain, mediastinum, heart, bone, soft tissue, spinal cord, spleen, pleura, adrenal gland, bladder, ovary, scrotum, thyroid gland, and peritoneum were also reported to be affected in descending order [1]. Spinal bony hydatidosis is a rare condition with a poor prognosis that presents diagnostic and therapeutic challenges [2,3]. The difficulty in diagnosis of these lesions lies not only in their being rare but also owing to the lack of descriptive imaging findings. For soft-tissue hydatidosis, particularly for liver, ‘‘water-lily sign’’ is described especially after puncture or percutaneous treatment of the cyst. In this case, we observed the ‘‘water-lily sign’’ in spinal HD, which was not described before for spinal cases but for soft tissues.
A 21-year-old man with an operation report of spinal HD 1 year ago was referred to our department to be investigated for the recurrence of the disease. From his operation report, it was learned that cystic lesions involving the right lamina and body of L2 vertebra and the cysts residing in the right psoas muscle were resected. Magnetic resonance images of his lumbar spine revealed cystic mass in the body, right pedicle, and right inferior articular process of L2 vertebra (Fig. 1). Cysts in the neural foramina between L3 and L4 vertebrae with internal detached membranes were also observed on T2-weighted images (Fig. 2). There were several cystic lesions showing detached membranes posteior to right psoas muscle as well. With these findings and considering the history, the bony and neural foraminal lesions were regarded as recurrent HD.
FDA device/drug status: Approved for these indications (Albendazole, Medendazole). Author disclosures: SG: Nothing to disclose. HTS: Nothing to disclose. * Corresponding author. Department of Radiology, Gulhane Military Medical Academy, Gn.Tevfik Saglam Cad., 06018 Kecioren, Ankara, Turkey. Tel.: (90) 312-304-4701. E-mail address: [email protected] (S. Gezgin) 1529-9430/$ - see front matter Ó 2012 Elsevier Inc. All rights reserved. doi:10.1016/j.spinee.2012.05.013
Discussion Uncommon sites for hydatid cysts cause difficulties in diagnosis even in endemic areas. Furthermore, additional unexpected complications like secondary bacterial infections can change the nature of the cyst and thus lead to further investigation and time loss. Knowing the parasitic etiology of the lesion before surgery or interventional procedure is important in terms of the prevention of local dissemination and anaphylactic reactions because of inappropriate handling. Diagnosis in our patient was straightforward owing to his previous surgery report. Analyzing the lesions
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S. Gezgin and H.T. Sanal / The Spine Journal 12 (2012) e5–e7
Fig. 1. Axial (Left) T1-weighted and (Right) T2-weighted images from the second lumbar vertebra. Cystic lesion expanding the right pedicle is seen with its low and high signal intensity on T1- and T2-weighted images, respectively. The cystic lesion (arrow) involves the right aspect of the vertebra as well causing slight convexity at the border. There is also sclerosis possibly because of the operative management right next to the cyst at the body.
retrospectively made us recognize ‘‘water-lily sign’’ in some of the cystic lesions, especially in the ones settled in the neural foraminae. Water-lily sign is a finding that describes delaminated endocyst floating in the cavity [1,4]. This sign is a pathognomonic finding in pulmonary and hepatic HD and was also described in cerebral, orbital, and intramuscular HD [5–10]. One example of this sign is presented in the liver of a patient being evaluated with contrast-enhanced abdominal computed tomography scan (Fig. 3). This sign in spinal HD has not been reported previously. In our case, we thought that previous surgery might cause a decrease in the pressure of the cysts leading this sign, because surgery toward spinal bone hydatid cysts is difficult because of impossibility to resect all components of the cysts [11,12]. Therefore, being familiar with characteristic and pathogonomic features of hydatidosis is important because of its high recurrence and complication rates. Recurrence must be differentiated from complications like abscess formation and pathologic fractures or osteolysis. Magnetic resonance imaging is the choice of modality to diagnose primary and recurrent spinal HD. Slow growing behavior of the cysts leads to bony remodeling and subsequent enlargement of the bony structures. Cystic fluidfilled lesions with thin walls enlarging the vertebral structures, their continuity with the posterior ribs or spinal
Fig. 2. (Left) Axial and (Right) sagittal T2-weighted images of the spine. Note that the cystic lesions at the neural foraminae have their internal detached membranes (arrows) indicating the so-called sign of ‘‘water-lily.’’
canal, adjacent soft-tissue cystic lesions are most expected signs of spinal HD. ‘‘Water-lily sign,’’ which was described before as a characteristic imaging finding of HD in soft tissues, is observed in spinal bony HD of this case report as well. Gold standard treatment regime of spinal HD is the removal of all the cysts without perforation. On removal, the surgical area may be irrigated with hypertonic saline (3%) to destroy the scoleces [13,14]. Depending on the severity and anatomic site of involvement, posterior, anterior, or dual decompression surgery may be added to the procedure by corpectomy and graft placement [15,16]. Additionally, posterior stabilization may be required to avoid spinal instability [17]. Despite surgical removal, hydatid cysts usually show recurrence, and as a result long-lasting morbidity occurs. Adjuvant administration of medications, such as albendazole or mebendazole, on a long-term basis can improve the prognosis and thereafter stabilize the process longer [18]. Besides serologic tests, patients should be
Fig. 3. Axial contrast-enhanced abdominal computed tomographic scan of a patient through the liver. Cyst located in the posterior segment of the right lobe (arrow) with internal detached membranes (arrowheads) is seen well. This appearance of detached membranes, that is, ‘‘water-lily’’ sign, is a typical finding of hepatic HD, especially after treatment.
S. Gezgin and H.T. Sanal / The Spine Journal 12 (2012) e5–e7
followed by radiologic means periodically for the detection of possible early recurrences [18]. In conclusion, ‘‘water-lily sign,’’ which has not been described for spinal bony HD before, is a useful sign and should be looked for as well. References [1] Polat P, Kantarci M, Alper F, et al. Hydatid disease from head to toe. Radiographics 2003;23:475–94. quiz 477–536. [2] Prabhakar MM, Acharya AJ, Modi DR, Jadav B. Spinal hydatid disease: a case series. J Spinal Cord Med 2005;28:426–31. [3] Torricelli P, Martinelli C, Biagini R, et al. Radiographic and computed tomographic findings in hydatid disease of bone. Skeletal Radiol 1990;19:435–9. [4] Mortele KJ, Segatto E, Ros PR. The infected liver: radiologicpathologic correlation. Radiographics 2004;24:937–55. [5] Gomori JM, Cohen D, Eyd A, Pomeranz S. Water lily sign in CT of cerebral hydatid disease: a case report. Neuroradiology 1988;30:358. [6] Comert RB, Aydingoz U, Ucaner A, Arikan M. Water-lily sign on MR imaging of primary intramuscular hydatidosis of sartorius muscle. Skeletal Radiol 2003;32:420–3. [7] Duygulu F, Karaoglu S, Erdogan N, Yildiz O. Primary hydatid cyst of the thigh: a case report of an unusual localization. Turk J Pediatr 2006;48:256–9.
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[8] Malde HM, Gadkari SS, Chadha D, Gondhalekar N. Water lily sign in an orbital hydatid cyst. J Clin Ultrasound 1993;21:458–9. [9] Vasilevska V, Zafirovski G, Kirjas N, et al. Imaging diagnosis of musculoskeletal hydatid disease. Prilozi 2007;28:199–209. [10] Durakbasa MO, Kose O, Islam NC, Kilicoglu G. A primary hydatid cyst of the gracilis: a case report. J Orthop Surg (Hong Kong) 2007;15:118–20. [11] Fares Y, Khazim R, El Zaatari MM, et al. Spinal hydatid disease and its neurological complications. Scand J Infect Dis 2003;35:394–6. [12] Zlitni M, Ezzaouia K, Lebib H, et al. Hydatid cyst of bone: diagnosis and treatment. World J Surg 2001;25:75–82. [13] Awasthy N, Chand K. Primary hydatid disease of the spine: an unusual case. Br J Neurosurg 2005;19:425–7. [14] Pushparaj K, Sundararajan M, Madeswaran K, Ambalavanan S. Primary spinal intradural hydatid cyst—a short report. Neurol India 2001;49:203–4. [15] Gunecs M, Akdemir H, Tugcu B, et al. Multiple intradural spinal hydatid disease: a case report and review of literature. Spine 2009;34: E346–50. [16] Parvaresh M, Moin H, Miles JB. Dumbbell hydatid cyst of the spine. Br J Neurosurg 1996;10:211–3. [17] Keller TM, Schweitzer JS, Helfend LK, Chappell T. Treatment of progressive cervical spinal instability secondary to hydatid disease. A case report. Spine 1997;22:915–9. [18] Turan Suslu H, Cecen A, Karaaslan A, et al. Primary spinal hydatid disease. Turk Neurosurg 2009;19:186–8.
Case Report
‘‘Water-lily sign’’ as a rare finding of spinal hydatidosis Sertan Gezgin, MD*, Hatice T. Sanal, MD Department of Radiology, Gulhane Military Medical Academy, Gn.Tevfik Saglam Cad., 06018 Kecioren, Ankara, Turkey Received 24 April 2011; revised 13 September 2011; accepted 17 May 2012
Abstract
Hydatid disease (HD) is a common parasitic infestation in some developing countries. Hydatid disease may affect many organs in the body, but the most affected are the liver and lungs. Hepatic and pulmonary HD is relatively easier to diagnose because of the frequency of pathognomic features and its order in the differential diagnosis list. When HD is located in uncommon sites, it is difficult to recognize even in endemic areas. The ‘‘water-lily sign’’ is a pathognomic sign for HD and mostly described especially after puncture or percutaneous treatment of the cyst. In this case, we observed the water-lily sign in spinal HD, which was not described before for spinal cases but for soft tissues. Ó 2012 Elsevier Inc. All rights reserved.
Keywords:
Hydatid disease; Water-lily sign; Spinal cyst hydatid
Introduction
Case report
Hydatid disease (HD) is an infestation mostly caused by the larvas of Echinococcus granulosus. Hydatid disease is endemic in places such as Central Asia, southern and eastern Mediterranean countries, East Africa, Australia, New Zealand, and South America. The liver and lungs are the most affected organs, but other parts of the body such as kidney, brain, mediastinum, heart, bone, soft tissue, spinal cord, spleen, pleura, adrenal gland, bladder, ovary, scrotum, thyroid gland, and peritoneum were also reported to be affected in descending order [1]. Spinal bony hydatidosis is a rare condition with a poor prognosis that presents diagnostic and therapeutic challenges [2,3]. The difficulty in diagnosis of these lesions lies not only in their being rare but also owing to the lack of descriptive imaging findings. For soft-tissue hydatidosis, particularly for liver, ‘‘water-lily sign’’ is described especially after puncture or percutaneous treatment of the cyst. In this case, we observed the ‘‘water-lily sign’’ in spinal HD, which was not described before for spinal cases but for soft tissues.
A 21-year-old man with an operation report of spinal HD 1 year ago was referred to our department to be investigated for the recurrence of the disease. From his operation report, it was learned that cystic lesions involving the right lamina and body of L2 vertebra and the cysts residing in the right psoas muscle were resected. Magnetic resonance images of his lumbar spine revealed cystic mass in the body, right pedicle, and right inferior articular process of L2 vertebra (Fig. 1). Cysts in the neural foramina between L3 and L4 vertebrae with internal detached membranes were also observed on T2-weighted images (Fig. 2). There were several cystic lesions showing detached membranes posteior to right psoas muscle as well. With these findings and considering the history, the bony and neural foraminal lesions were regarded as recurrent HD.
FDA device/drug status: Approved for these indications (Albendazole, Medendazole). Author disclosures: SG: Nothing to disclose. HTS: Nothing to disclose. * Corresponding author. Department of Radiology, Gulhane Military Medical Academy, Gn.Tevfik Saglam Cad., 06018 Kecioren, Ankara, Turkey. Tel.: (90) 312-304-4701. E-mail address: [email protected] (S. Gezgin) 1529-9430/$ - see front matter Ó 2012 Elsevier Inc. All rights reserved. doi:10.1016/j.spinee.2012.05.013
Discussion Uncommon sites for hydatid cysts cause difficulties in diagnosis even in endemic areas. Furthermore, additional unexpected complications like secondary bacterial infections can change the nature of the cyst and thus lead to further investigation and time loss. Knowing the parasitic etiology of the lesion before surgery or interventional procedure is important in terms of the prevention of local dissemination and anaphylactic reactions because of inappropriate handling. Diagnosis in our patient was straightforward owing to his previous surgery report. Analyzing the lesions
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S. Gezgin and H.T. Sanal / The Spine Journal 12 (2012) e5–e7
Fig. 1. Axial (Left) T1-weighted and (Right) T2-weighted images from the second lumbar vertebra. Cystic lesion expanding the right pedicle is seen with its low and high signal intensity on T1- and T2-weighted images, respectively. The cystic lesion (arrow) involves the right aspect of the vertebra as well causing slight convexity at the border. There is also sclerosis possibly because of the operative management right next to the cyst at the body.
retrospectively made us recognize ‘‘water-lily sign’’ in some of the cystic lesions, especially in the ones settled in the neural foraminae. Water-lily sign is a finding that describes delaminated endocyst floating in the cavity [1,4]. This sign is a pathognomonic finding in pulmonary and hepatic HD and was also described in cerebral, orbital, and intramuscular HD [5–10]. One example of this sign is presented in the liver of a patient being evaluated with contrast-enhanced abdominal computed tomography scan (Fig. 3). This sign in spinal HD has not been reported previously. In our case, we thought that previous surgery might cause a decrease in the pressure of the cysts leading this sign, because surgery toward spinal bone hydatid cysts is difficult because of impossibility to resect all components of the cysts [11,12]. Therefore, being familiar with characteristic and pathogonomic features of hydatidosis is important because of its high recurrence and complication rates. Recurrence must be differentiated from complications like abscess formation and pathologic fractures or osteolysis. Magnetic resonance imaging is the choice of modality to diagnose primary and recurrent spinal HD. Slow growing behavior of the cysts leads to bony remodeling and subsequent enlargement of the bony structures. Cystic fluidfilled lesions with thin walls enlarging the vertebral structures, their continuity with the posterior ribs or spinal
Fig. 2. (Left) Axial and (Right) sagittal T2-weighted images of the spine. Note that the cystic lesions at the neural foraminae have their internal detached membranes (arrows) indicating the so-called sign of ‘‘water-lily.’’
canal, adjacent soft-tissue cystic lesions are most expected signs of spinal HD. ‘‘Water-lily sign,’’ which was described before as a characteristic imaging finding of HD in soft tissues, is observed in spinal bony HD of this case report as well. Gold standard treatment regime of spinal HD is the removal of all the cysts without perforation. On removal, the surgical area may be irrigated with hypertonic saline (3%) to destroy the scoleces [13,14]. Depending on the severity and anatomic site of involvement, posterior, anterior, or dual decompression surgery may be added to the procedure by corpectomy and graft placement [15,16]. Additionally, posterior stabilization may be required to avoid spinal instability [17]. Despite surgical removal, hydatid cysts usually show recurrence, and as a result long-lasting morbidity occurs. Adjuvant administration of medications, such as albendazole or mebendazole, on a long-term basis can improve the prognosis and thereafter stabilize the process longer [18]. Besides serologic tests, patients should be
Fig. 3. Axial contrast-enhanced abdominal computed tomographic scan of a patient through the liver. Cyst located in the posterior segment of the right lobe (arrow) with internal detached membranes (arrowheads) is seen well. This appearance of detached membranes, that is, ‘‘water-lily’’ sign, is a typical finding of hepatic HD, especially after treatment.
S. Gezgin and H.T. Sanal / The Spine Journal 12 (2012) e5–e7
followed by radiologic means periodically for the detection of possible early recurrences [18]. In conclusion, ‘‘water-lily sign,’’ which has not been described for spinal bony HD before, is a useful sign and should be looked for as well. References [1] Polat P, Kantarci M, Alper F, et al. Hydatid disease from head to toe. Radiographics 2003;23:475–94. quiz 477–536. [2] Prabhakar MM, Acharya AJ, Modi DR, Jadav B. Spinal hydatid disease: a case series. J Spinal Cord Med 2005;28:426–31. [3] Torricelli P, Martinelli C, Biagini R, et al. Radiographic and computed tomographic findings in hydatid disease of bone. Skeletal Radiol 1990;19:435–9. [4] Mortele KJ, Segatto E, Ros PR. The infected liver: radiologicpathologic correlation. Radiographics 2004;24:937–55. [5] Gomori JM, Cohen D, Eyd A, Pomeranz S. Water lily sign in CT of cerebral hydatid disease: a case report. Neuroradiology 1988;30:358. [6] Comert RB, Aydingoz U, Ucaner A, Arikan M. Water-lily sign on MR imaging of primary intramuscular hydatidosis of sartorius muscle. Skeletal Radiol 2003;32:420–3. [7] Duygulu F, Karaoglu S, Erdogan N, Yildiz O. Primary hydatid cyst of the thigh: a case report of an unusual localization. Turk J Pediatr 2006;48:256–9.
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[8] Malde HM, Gadkari SS, Chadha D, Gondhalekar N. Water lily sign in an orbital hydatid cyst. J Clin Ultrasound 1993;21:458–9. [9] Vasilevska V, Zafirovski G, Kirjas N, et al. Imaging diagnosis of musculoskeletal hydatid disease. Prilozi 2007;28:199–209. [10] Durakbasa MO, Kose O, Islam NC, Kilicoglu G. A primary hydatid cyst of the gracilis: a case report. J Orthop Surg (Hong Kong) 2007;15:118–20. [11] Fares Y, Khazim R, El Zaatari MM, et al. Spinal hydatid disease and its neurological complications. Scand J Infect Dis 2003;35:394–6. [12] Zlitni M, Ezzaouia K, Lebib H, et al. Hydatid cyst of bone: diagnosis and treatment. World J Surg 2001;25:75–82. [13] Awasthy N, Chand K. Primary hydatid disease of the spine: an unusual case. Br J Neurosurg 2005;19:425–7. [14] Pushparaj K, Sundararajan M, Madeswaran K, Ambalavanan S. Primary spinal intradural hydatid cyst—a short report. Neurol India 2001;49:203–4. [15] Gunecs M, Akdemir H, Tugcu B, et al. Multiple intradural spinal hydatid disease: a case report and review of literature. Spine 2009;34: E346–50. [16] Parvaresh M, Moin H, Miles JB. Dumbbell hydatid cyst of the spine. Br J Neurosurg 1996;10:211–3. [17] Keller TM, Schweitzer JS, Helfend LK, Chappell T. Treatment of progressive cervical spinal instability secondary to hydatid disease. A case report. Spine 1997;22:915–9. [18] Turan Suslu H, Cecen A, Karaaslan A, et al. Primary spinal hydatid disease. Turk Neurosurg 2009;19:186–8.