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Intradural cervical inflammatory pseudotumor mimicking epidural hematoma in a pregnant woman: case report and review of the literature
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Surgical Neurology 69 (2008) 302 – 305 www.surgicalneurology-online.com
Spine
Intradural cervical inflammatory pseudotumor mimicking epidural hematoma in a pregnant woman: case report and review of the literature Mahjouba Boutarbouch, MD4, Yasser Arkha, MD, Lobna Rifi, MD, Said Derraz, MD, Abdessamad El Ouahabi, MD, Abdeslam El Khamlichi, MD Department of Neurosurgery, Mohamed Vth University School of Medicine, Hoˆpital des Spe´cialite´s ONO, Rabat 10100, Morocco Received 7 December 2006; accepted 20 December 2006
Abstract
Background: Inflammatory pseudotumors usually affect the lung and the orbit. They occur extremely rarely in the spine. We encountered a case of intradural extramedullary IPT of the cervical spine in a pregnant woman that initially presented as an epidural hematoma in MRI. Case Description: This is a case of a 30-year-old, HIV-negative, full-term pregnant lady presenting with 2-month history of progressive quadriparesis with rapid worsening of power in all 4 limbs for 3 days. Magnetic resonance imaging scanning revealed cervical extramedullary compressive lesion likely to be epidural bleed from its imaging characteristics. Intraoperatively, intradural extramedullary granulomatous lesion was found with dural thickening. Gross total excision was accomplished; histopathologic study assessed an IPT. The patient improved after surgery excision and is doing well at 6-month follow-up with remarkable neurological recovery. Conclusion: Cervical intradural IPT is a very rare issue in the spine. Diagnosis can be confused with a neoplastic lesion like an ben plaque meningiomaQ or as epidural bleed like in the present case. D 2008 Elsevier Inc. All rights reserved.
Keywords:
Inflammatory pseudotumor; Spinal; Pregnancy; Epidural hematoma
1. Introduction Inflammatory pseudotumors are soft-tissue masses of unidentified etiology, which include inflammatory cells, histiocytes, and fibroblasts. These lesions usually affect the lung and the orbit; they are very rare in the spine. We report an interesting case of cervical intradural extramedullary IPT in a 30-year-old lady who was 39 weeks pregnant and was operated upon with a presumptive diagnosis of epidural bleed based on radiological imaging findings. 2. Case description A 30-year-old pregnant woman presented with a history of cervicodorsal pain lasting for 2 months with numbness of Abbreviations: IPT, inflammatory pseudotumor; MRI, magnetic resonance imaging. 4 Corresponding author. Service de Neurochirurgie, Hoˆpital des Spe´cialite´s, BP 6444, Rabat-Instituts, Rabat 10100, Morocco. Tel.: +212 62 72 49 62; fax: +212 37 77 55 73. E-mail address: [email protected] (M. Boutarbouch). 0090-3019/$ – see front matter D 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.surneu.2006.12.056
the upper and lower limbs. She complained of rapid worsening of neurological power for the last 3 days. There was no history of trauma or anticoagulation therapy. On examination, she was detected to have left predominant quadriparesis; power was 3/5 Frankel grade on the right side and 2/5 Frankel grade on the left side with brisk tendon jerks of all 4 limbs; superficial abdominal reflexes were absent and Babinski sign was present bilaterally. A uterine ultrasonography showed a 39–amenorrhea week mature fetus. Results of a chest x-ray and routine laboratory investigations were normal. Magnetic resonance imaging scanning showed an oblong mass located along the C4 to T2 vertebrae; it was hypointense to isointense in T1 sequences (Fig. 1) and hypointense in T2-weighted sequences (Fig. 2), causing significant compression of the spinal cord. Gadolinium could not be injected knowing the patient was pregnant. This picture was highly suggestive of an epidural hematoma. There was no evidence of abnormalities in the adjacent bone, the posterior neural arch was normal, and paraspinal muscles were normal (Fig. 3).
M. Boutarbouch et al. / Surgical Neurology 69 (2008) 302–305
Fig. 1. Sagittal MRI view in T1-weighted sequences showing an oblong C4 to T1 process that appears iso- and hypointense.
Based on the patient’s MRI findings, it was believed that she had cervical epidural hematoma, and was thence scheduled for surgery. At first, the patient was delivered of the infant by performing a caesarean intervention under general anesthesia. The patient was then repositioned, and a cervical C5 to T2 laminectomy was performed; epidural thickening was noted. After durotomy, a well-circumscribed avascular whitish firm mass adhering to the dura without infiltration of the dura or the spinal cord was found. Total removal of both the fibrous mass and the involved dura was performed. There was no bleed or old clotted blood. Duraplasty was performed using adjacent muscle aponevrosis. The intraoperative diagnosis was of an ben plaque meningiomaQ or any other intradural neoplastic lesion. The postoperative course was uneventful. The muscle weakness progressively improved, and the patient recovered 5/5 Frankel grade immediately after intervention. The chest x-ray films were reexamined, and there were no pathologic signs associated with tuberculosis. Mycobacterium tuberculosis could not be cultured from the cerebrospinal fluid. The pathologic findings were diagnostic for IPT; thus, the patient was given no additional drugs. She was discharged on the 10th postoperative day with wearing of the cervical collar for 4 weeks. Histopathologic examination of the surgical specimens showed granulation tissue containing giant cells, abundant hyalinized fibrosis, collagenous deposits, marked infiltration
303
Fig. 2. Sagittal MRI view in T2-weighted sequences showing a hypointense well-circumscribed mass.
mostly by lymphocytes, and fibrohistiocytic proliferation (Fig. 4). Cultures were negative for M tuberculosis, and stains for fungi and acid-fast microorganisms were negative. Immunohistochemical studies showed the infiltrating population to consist of both T and B lymphocytes. The B cells were polyclonal as assessed by light chain expression.
Fig. 3. Axial MRI view in T2-weighted sequences showing a hypointense extramedullary lesion with no evidence of paraspinal abnormalities.
304
M. Boutarbouch et al. / Surgical Neurology 69 (2008) 302–305
Fig. 4. Histopathologic examination showing fibrous trabeculae infiltrated by lymphocytes, plasma cells, and macrophages.
The patient had no complementary treatment because the surgical excision was total. At follow-up examination 6 months postoperatively, the patient had no neurological symptoms. 3. Discussion Inflammatory pseudotumors represent a rare etiology of space-occupying lesions at different localizations. These lesions mostly interest the lung [3], with extrapulmonary localizations including the orbit [5], nasal sinuses [7], liver [7,20], spleen [29], pancreas [25], bowel [31,32], kidney [11], urinary bladder [23], testis [30], heart [8], lymphatic system [13,14], and skin [17]. There is no age, sex, or geographic predilection to their incidence. They are important because of the difficulty in differentiating them from true neoplasms clinically, radiologically, and intraoperatively. The pathogenesis of IPT is unknown. However, it is considered an immunologic host response to infectious agents, microorganisms, neighboring necrotic tissue or chronic inflammation, neoplasms, or foreign bodies. It is also believed to be associated to cocaine consumption [18,19] and Epstein-Barr virus infection [4]. Therefore, diagnosis of IPT is actually made after elimination of detectable fungi or bacteria. However, in most documented cases of IPT, the etiology remains unknown. No inciting factor could be determined in our patient. There is no evidence of documented IPT association with pregnancy; the very few reported cases of IPT in pregnant women [2,21] do not allow any further conclusions concerning the pregnancy implication in the IPT pathogenesis process. In most cases, IPTs are not formally diagnosed until the excised mass is examined histologically and subjected to immunologic staining techniques. This is due to their rarity and nonspecific presentation both clinically and
radiologically. These pseudotumors have diverse histologic patterns; Coffin et al [6] classified basic histologic patterns of IPT into 3 groups: (1) myxoid, vascular, and inflammatory areas resembling nodular fascitis; (2) compact spindle cells with intermingled inflammatory cells resembling fibrous histiocytoma; and (3) dense platelike collagen resembling a desmoid or scar. In our case, the histopathologic findings are likely to be transitional between types 2 and 3. We only found 5 previous cases of intraspinal IPT published in the literature. The first case of intraspinal cervical IPT was reported by Eimoto et al [9] in 1978 about a 37-year-old Japanese man who underwent surgical excision followed by prophylactic radiotherapy before the diagnosis of IPT was made. Gilliard et al [12] reported a second IPT case in 2000 about a 45-year-old man harboring a multifocal fibrosclerosis in whom was diagnosed a cervical epidural IPT extending from C2 to T2; our case is the third reported in the cervical spine. Three other cases of IPT localized in the thoracic spine canal were reported in the literature [26-28]. Intraspinal localization of the lesion can also occur from a pulmonary IPT extension to the neighboring vertebrae [16]. Few cases of IPT of the central nervous system have been reported in the literature. They may actually develop in any site of the brain or the spinal cord, but they mostly arise from the meninges [1]. In other cases, local extension has been reported to occur, as in orbital pseudotumors that can spread along the meningeal coverings into the intracranial cavity [5]. As far as the imaging findings are concerned, characteristic low signal intensity is present in IPTs as in our case; it most likely reflects relative loss of free water and mobile protons within the fibrotic regions [22]. Enhancement after gadolinium injection is not constantly present, particularly when the lesion is predominantly fibrotic. Epidural hematoma in the acute phase can present as a heterogeneous, isointense, or hypointense lesion in T1 sequences with low signal intensity in T2-weighted sequences [10,24] as in our case. The therapeutic approach to IPT in other localizations (eg, lung) may be difficult. Surgical excision is usually mandatory in IPT of the central nervous system because of the emergent need of relieving the mass effect; it is generally curative when total removal is performed [1]. Some patients underwent radiation therapy despite uncertainty about its efficiency [15,33]. Systemic steroids and immunosuppressive drugs are also given in IPT in other sites and lead to mass volume decreasing. 4. Conclusion Although very rare, intraspinal IPTs should be kept in mind in the differential diagnosis of cord compression etiologies. It is a benign lesion with nonspecific clinical and radiological presentation; its total excision is curative.
M. Boutarbouch et al. / Surgical Neurology 69 (2008) 302–305
References [1] Al-Sarraj S, Wasserberg J, Bartlett R, Bridges LR. Inflammatory pseudotumour of the central nervous system: clinicopathological study of one case and review of the literature. Br J Neurosurg 1995; 9:57 - 66. [2] Amir R, Danahey D, Ferrer K, Maffee M. Inflammatory myofibroblastic tumor presenting with tracheal obstruction in a pregnant woman. Am J Otolaryngol 2002;23(6):362 - 7. [3] Anthony PP. Inflammatory pseudotumour (plasma cell granuloma) of the lung, liver and other organs. Histopathol 1993;23:501 - 3. [4] Arber D, Kamel OW, Van De Rjin M, Weiss LM. Frequent presence of the Epstein Barr virus in inflammatory pseudotumor. Hum Pathol 1995;26:1093 - 8. [5] Clifton AG, Borgstein RL, Moseley IF, et al. Intracranial extension of orbital inflammatory pseudotumour. Clin Radiol 1992;45:23 - 6. [6] Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor): a clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 1995;19:859 - 72. [7] De Miguel GF, Bori AMA, Fernandez LR. Inflammatory pseudotumour of the paranasal sinuses. Acta Otorrinolaringol Esp 1990; 41:351 - 3. [8] Dickens P, Lam AK. Sudden death associated with cardiac inflammatory pseudotumour. Forensic Sci Int 1991;49:89 - 93. [9] Eimoto T, Yanaka M, Kurosawa M, et al. Plasma cell granuloma (inflammatory pseudotumour) of the spinal cord meninges: report of a case. Cancer 1978;41:1929 - 36. [10] Fukui MB, Swarnkar AS, Williams RL. Acute spontaneous spinal epidural hematomas. AJNR Am J Neuroradiol 1999;20:1365 - 72. [11] Gelister JS, Jarmulowicz M, et al. Renal inflammatory pseudotumour. Br J Urol 1990;65:548 - 9. [12] Gilliard C, De Coene B, Lahdou JB, Boutsen Y, Noel H, Godfraind C. Cervical epidural pseudotumor and multifocal fibrosclerosis. Case report and review of the literature. J Neurosurg 2000;93(1 Suppl): 152 - 6. [13] Gomez-Morales M, Andujar-Sanchez M, Medina-Cano T, et al. Disseminated inflammatory pseudotumour of the lymph nodes. Med Clin (Barc) 1994;102:581 - 3. [14] Hadley J, Coady AT, Milford CA. Pseudotumour of the maxillary sinus. J Laryngol Otol 1990;104:244 - 7. [15] Hirohata M, Sasaguri Y, Sugita Y, Tokutomi T, Kobayashi S, Minoru M, Shigemori M. Intracranial plasma cell granuloma: a case report. Brain Tumor Pathol 1996;13:133 - 8. [16] Hong HY, Castelli MJ, Walloch JL. Pulmonary plasma cell granuloma (inflammatory pseudotumour) with invasion of thoracic vertebra. Mt Sinai J Med 1990;57:117 - 21.
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[17] Hurt AM, Santa Cruz DJ. Cutaneous inflammatory pseudotumour: lesion resembling inflammatory pseudotumour or plasma cell granuloma of extracutaneous sites. Am J Surg Pathol 1990;14: 764 - 73. [18] Hytiroglou P, Brandwein MS, Strauchen JA, Mirante JP, Urken ML, Biller HF. Inflammatory pseudotumor of the parapharyngeal space: case report and review of the literature. Head Neck 1992;14:230 - 4. [19] Karatsis P, Wyman A, et al. Inflammatory pseudotumour of the liver. Eur J Surg Oncol 1993;19:384 - 7. [20] Lyons TJ, Benbow EW, Taylor PM, et al. Inflammatory pseudotumour of the liver: antecedent causes and clinical experience. J Hepatol 1993;19:273 - 8. [21] Maze GL, Lee M, Schenker S. Inflammatory pseudotumor of the liver and pregnancy. Am J Gastroenterol 1999;94(2):529 - 30. [22] Nakayama K, Inoue Y, Aiba T, Kono K, Wakasa K, Yamada R. Unusual CT and MR findings of inflammatory pseudotumor in the parapharyngeal space: case report. AJNR Am J Neuroradiol 2001; 22:1394 - 7. [23] N’Dow J, Brown PA, McClinton S, et al. Inflammatory pseudotumour of the urinary bladder. Br J Urol 1993;72:379 - 80. [24] Ohayon L, Gorhan C, Soto-Ares G, Reyns N, Pruvo JP. Acute spinal epidural and subdural hematomas. J Radiol 2003;84(1):50 - 3. [25] Page DL. Inflammatory pseudotumour of the pancreas (letter). Histopathology 1994;24:594. [26] Roberts GA, Eldridge PR, Mackenzie JM. Case report: inflammatory pseudotumour of the spine, with literature review. Br J Neurosurg 1997;11(6):570 - 2. [27] Scriven MW, Shandell A, Fitzgerald EJ, et al. Hepatic pseudotumours: an important diagnostic pitfall. Ann R Coll Surg Engl 1993;75:43 - 5. [28] Seol HJ, Kim SS, Kim JE, Lee SH, Won JY. Inflammatory pseudotumor in the epidural space of the thoracic spine: a case report and literature review of MR imaging findings. AJNR Am J Neuroradiol 2005;26(10):2667 - 70. [29] Shah DJ, Royes CA. Inflammatory pseudotumour of the spleen. Report of a case and review of the literature. West Indian Med J 1994; 43:30 - 1. [30] Sharma AK, Morgan RJ, Sadrudin AA. Pseudotumour of the testicular tunica—a trap for the unwary. Br J Clin Pract 1990;44:507. [31] Skaane P. Non-specific inflammatory pseudotumour of the caecum unrelated to the vermiform appendix. Ital J Gastroenterol 1990; 22:357 - 9. [32] Soga J, Saito K, Suzuki N, et al. Plasma cell granuloma of the stomach. A report of a case and review of the literature. Cancer 1970; 25:618 - 25. [33] Tang TT, Segura AD, Oechler HE, Harb JM, Adair SE, Gregg DC, Camitta B.M. Inflammatory myofibrohistiocytic proliferation simulating sarcoma in children. Cancer 1990;65:1626 - 34.
Surgical Neurology 69 (2008) 302 – 305 www.surgicalneurology-online.com
Spine
Intradural cervical inflammatory pseudotumor mimicking epidural hematoma in a pregnant woman: case report and review of the literature Mahjouba Boutarbouch, MD4, Yasser Arkha, MD, Lobna Rifi, MD, Said Derraz, MD, Abdessamad El Ouahabi, MD, Abdeslam El Khamlichi, MD Department of Neurosurgery, Mohamed Vth University School of Medicine, Hoˆpital des Spe´cialite´s ONO, Rabat 10100, Morocco Received 7 December 2006; accepted 20 December 2006
Abstract
Background: Inflammatory pseudotumors usually affect the lung and the orbit. They occur extremely rarely in the spine. We encountered a case of intradural extramedullary IPT of the cervical spine in a pregnant woman that initially presented as an epidural hematoma in MRI. Case Description: This is a case of a 30-year-old, HIV-negative, full-term pregnant lady presenting with 2-month history of progressive quadriparesis with rapid worsening of power in all 4 limbs for 3 days. Magnetic resonance imaging scanning revealed cervical extramedullary compressive lesion likely to be epidural bleed from its imaging characteristics. Intraoperatively, intradural extramedullary granulomatous lesion was found with dural thickening. Gross total excision was accomplished; histopathologic study assessed an IPT. The patient improved after surgery excision and is doing well at 6-month follow-up with remarkable neurological recovery. Conclusion: Cervical intradural IPT is a very rare issue in the spine. Diagnosis can be confused with a neoplastic lesion like an ben plaque meningiomaQ or as epidural bleed like in the present case. D 2008 Elsevier Inc. All rights reserved.
Keywords:
Inflammatory pseudotumor; Spinal; Pregnancy; Epidural hematoma
1. Introduction Inflammatory pseudotumors are soft-tissue masses of unidentified etiology, which include inflammatory cells, histiocytes, and fibroblasts. These lesions usually affect the lung and the orbit; they are very rare in the spine. We report an interesting case of cervical intradural extramedullary IPT in a 30-year-old lady who was 39 weeks pregnant and was operated upon with a presumptive diagnosis of epidural bleed based on radiological imaging findings. 2. Case description A 30-year-old pregnant woman presented with a history of cervicodorsal pain lasting for 2 months with numbness of Abbreviations: IPT, inflammatory pseudotumor; MRI, magnetic resonance imaging. 4 Corresponding author. Service de Neurochirurgie, Hoˆpital des Spe´cialite´s, BP 6444, Rabat-Instituts, Rabat 10100, Morocco. Tel.: +212 62 72 49 62; fax: +212 37 77 55 73. E-mail address: [email protected] (M. Boutarbouch). 0090-3019/$ – see front matter D 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.surneu.2006.12.056
the upper and lower limbs. She complained of rapid worsening of neurological power for the last 3 days. There was no history of trauma or anticoagulation therapy. On examination, she was detected to have left predominant quadriparesis; power was 3/5 Frankel grade on the right side and 2/5 Frankel grade on the left side with brisk tendon jerks of all 4 limbs; superficial abdominal reflexes were absent and Babinski sign was present bilaterally. A uterine ultrasonography showed a 39–amenorrhea week mature fetus. Results of a chest x-ray and routine laboratory investigations were normal. Magnetic resonance imaging scanning showed an oblong mass located along the C4 to T2 vertebrae; it was hypointense to isointense in T1 sequences (Fig. 1) and hypointense in T2-weighted sequences (Fig. 2), causing significant compression of the spinal cord. Gadolinium could not be injected knowing the patient was pregnant. This picture was highly suggestive of an epidural hematoma. There was no evidence of abnormalities in the adjacent bone, the posterior neural arch was normal, and paraspinal muscles were normal (Fig. 3).
M. Boutarbouch et al. / Surgical Neurology 69 (2008) 302–305
Fig. 1. Sagittal MRI view in T1-weighted sequences showing an oblong C4 to T1 process that appears iso- and hypointense.
Based on the patient’s MRI findings, it was believed that she had cervical epidural hematoma, and was thence scheduled for surgery. At first, the patient was delivered of the infant by performing a caesarean intervention under general anesthesia. The patient was then repositioned, and a cervical C5 to T2 laminectomy was performed; epidural thickening was noted. After durotomy, a well-circumscribed avascular whitish firm mass adhering to the dura without infiltration of the dura or the spinal cord was found. Total removal of both the fibrous mass and the involved dura was performed. There was no bleed or old clotted blood. Duraplasty was performed using adjacent muscle aponevrosis. The intraoperative diagnosis was of an ben plaque meningiomaQ or any other intradural neoplastic lesion. The postoperative course was uneventful. The muscle weakness progressively improved, and the patient recovered 5/5 Frankel grade immediately after intervention. The chest x-ray films were reexamined, and there were no pathologic signs associated with tuberculosis. Mycobacterium tuberculosis could not be cultured from the cerebrospinal fluid. The pathologic findings were diagnostic for IPT; thus, the patient was given no additional drugs. She was discharged on the 10th postoperative day with wearing of the cervical collar for 4 weeks. Histopathologic examination of the surgical specimens showed granulation tissue containing giant cells, abundant hyalinized fibrosis, collagenous deposits, marked infiltration
303
Fig. 2. Sagittal MRI view in T2-weighted sequences showing a hypointense well-circumscribed mass.
mostly by lymphocytes, and fibrohistiocytic proliferation (Fig. 4). Cultures were negative for M tuberculosis, and stains for fungi and acid-fast microorganisms were negative. Immunohistochemical studies showed the infiltrating population to consist of both T and B lymphocytes. The B cells were polyclonal as assessed by light chain expression.
Fig. 3. Axial MRI view in T2-weighted sequences showing a hypointense extramedullary lesion with no evidence of paraspinal abnormalities.
304
M. Boutarbouch et al. / Surgical Neurology 69 (2008) 302–305
Fig. 4. Histopathologic examination showing fibrous trabeculae infiltrated by lymphocytes, plasma cells, and macrophages.
The patient had no complementary treatment because the surgical excision was total. At follow-up examination 6 months postoperatively, the patient had no neurological symptoms. 3. Discussion Inflammatory pseudotumors represent a rare etiology of space-occupying lesions at different localizations. These lesions mostly interest the lung [3], with extrapulmonary localizations including the orbit [5], nasal sinuses [7], liver [7,20], spleen [29], pancreas [25], bowel [31,32], kidney [11], urinary bladder [23], testis [30], heart [8], lymphatic system [13,14], and skin [17]. There is no age, sex, or geographic predilection to their incidence. They are important because of the difficulty in differentiating them from true neoplasms clinically, radiologically, and intraoperatively. The pathogenesis of IPT is unknown. However, it is considered an immunologic host response to infectious agents, microorganisms, neighboring necrotic tissue or chronic inflammation, neoplasms, or foreign bodies. It is also believed to be associated to cocaine consumption [18,19] and Epstein-Barr virus infection [4]. Therefore, diagnosis of IPT is actually made after elimination of detectable fungi or bacteria. However, in most documented cases of IPT, the etiology remains unknown. No inciting factor could be determined in our patient. There is no evidence of documented IPT association with pregnancy; the very few reported cases of IPT in pregnant women [2,21] do not allow any further conclusions concerning the pregnancy implication in the IPT pathogenesis process. In most cases, IPTs are not formally diagnosed until the excised mass is examined histologically and subjected to immunologic staining techniques. This is due to their rarity and nonspecific presentation both clinically and
radiologically. These pseudotumors have diverse histologic patterns; Coffin et al [6] classified basic histologic patterns of IPT into 3 groups: (1) myxoid, vascular, and inflammatory areas resembling nodular fascitis; (2) compact spindle cells with intermingled inflammatory cells resembling fibrous histiocytoma; and (3) dense platelike collagen resembling a desmoid or scar. In our case, the histopathologic findings are likely to be transitional between types 2 and 3. We only found 5 previous cases of intraspinal IPT published in the literature. The first case of intraspinal cervical IPT was reported by Eimoto et al [9] in 1978 about a 37-year-old Japanese man who underwent surgical excision followed by prophylactic radiotherapy before the diagnosis of IPT was made. Gilliard et al [12] reported a second IPT case in 2000 about a 45-year-old man harboring a multifocal fibrosclerosis in whom was diagnosed a cervical epidural IPT extending from C2 to T2; our case is the third reported in the cervical spine. Three other cases of IPT localized in the thoracic spine canal were reported in the literature [26-28]. Intraspinal localization of the lesion can also occur from a pulmonary IPT extension to the neighboring vertebrae [16]. Few cases of IPT of the central nervous system have been reported in the literature. They may actually develop in any site of the brain or the spinal cord, but they mostly arise from the meninges [1]. In other cases, local extension has been reported to occur, as in orbital pseudotumors that can spread along the meningeal coverings into the intracranial cavity [5]. As far as the imaging findings are concerned, characteristic low signal intensity is present in IPTs as in our case; it most likely reflects relative loss of free water and mobile protons within the fibrotic regions [22]. Enhancement after gadolinium injection is not constantly present, particularly when the lesion is predominantly fibrotic. Epidural hematoma in the acute phase can present as a heterogeneous, isointense, or hypointense lesion in T1 sequences with low signal intensity in T2-weighted sequences [10,24] as in our case. The therapeutic approach to IPT in other localizations (eg, lung) may be difficult. Surgical excision is usually mandatory in IPT of the central nervous system because of the emergent need of relieving the mass effect; it is generally curative when total removal is performed [1]. Some patients underwent radiation therapy despite uncertainty about its efficiency [15,33]. Systemic steroids and immunosuppressive drugs are also given in IPT in other sites and lead to mass volume decreasing. 4. Conclusion Although very rare, intraspinal IPTs should be kept in mind in the differential diagnosis of cord compression etiologies. It is a benign lesion with nonspecific clinical and radiological presentation; its total excision is curative.
M. Boutarbouch et al. / Surgical Neurology 69 (2008) 302–305
References [1] Al-Sarraj S, Wasserberg J, Bartlett R, Bridges LR. Inflammatory pseudotumour of the central nervous system: clinicopathological study of one case and review of the literature. Br J Neurosurg 1995; 9:57 - 66. [2] Amir R, Danahey D, Ferrer K, Maffee M. Inflammatory myofibroblastic tumor presenting with tracheal obstruction in a pregnant woman. Am J Otolaryngol 2002;23(6):362 - 7. [3] Anthony PP. Inflammatory pseudotumour (plasma cell granuloma) of the lung, liver and other organs. Histopathol 1993;23:501 - 3. [4] Arber D, Kamel OW, Van De Rjin M, Weiss LM. Frequent presence of the Epstein Barr virus in inflammatory pseudotumor. Hum Pathol 1995;26:1093 - 8. [5] Clifton AG, Borgstein RL, Moseley IF, et al. Intracranial extension of orbital inflammatory pseudotumour. Clin Radiol 1992;45:23 - 6. [6] Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor): a clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 1995;19:859 - 72. [7] De Miguel GF, Bori AMA, Fernandez LR. Inflammatory pseudotumour of the paranasal sinuses. Acta Otorrinolaringol Esp 1990; 41:351 - 3. [8] Dickens P, Lam AK. Sudden death associated with cardiac inflammatory pseudotumour. Forensic Sci Int 1991;49:89 - 93. [9] Eimoto T, Yanaka M, Kurosawa M, et al. Plasma cell granuloma (inflammatory pseudotumour) of the spinal cord meninges: report of a case. Cancer 1978;41:1929 - 36. [10] Fukui MB, Swarnkar AS, Williams RL. Acute spontaneous spinal epidural hematomas. AJNR Am J Neuroradiol 1999;20:1365 - 72. [11] Gelister JS, Jarmulowicz M, et al. Renal inflammatory pseudotumour. Br J Urol 1990;65:548 - 9. [12] Gilliard C, De Coene B, Lahdou JB, Boutsen Y, Noel H, Godfraind C. Cervical epidural pseudotumor and multifocal fibrosclerosis. Case report and review of the literature. J Neurosurg 2000;93(1 Suppl): 152 - 6. [13] Gomez-Morales M, Andujar-Sanchez M, Medina-Cano T, et al. Disseminated inflammatory pseudotumour of the lymph nodes. Med Clin (Barc) 1994;102:581 - 3. [14] Hadley J, Coady AT, Milford CA. Pseudotumour of the maxillary sinus. J Laryngol Otol 1990;104:244 - 7. [15] Hirohata M, Sasaguri Y, Sugita Y, Tokutomi T, Kobayashi S, Minoru M, Shigemori M. Intracranial plasma cell granuloma: a case report. Brain Tumor Pathol 1996;13:133 - 8. [16] Hong HY, Castelli MJ, Walloch JL. Pulmonary plasma cell granuloma (inflammatory pseudotumour) with invasion of thoracic vertebra. Mt Sinai J Med 1990;57:117 - 21.
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