Abstract
Plasma cell granuloma (PCG) is an uncommon non-neoplastic mass lesion of unknown etiology. It is characterized by a polyclonal proliferation of chronic inflammatory cells, mostly mature plasma and other mononuclear cells. PCGs arising in the central nervous system are particularly rare. We report two additional cases of intracranial PCG exclusively involving the brain parenchyma. A 47 year-old woman, presenting with partial motor seizures and fluent aphasia, underwent complete excision of a well-demarcated, enhancing left parietal mass. The second patient was a 56 year-old man presenting with headaches and right-sided weakness who underwent stereotactic biopsy of an ill-defined, heterogeneously enhancing lesion in the left basal ganglia. Immunohistochemical analysis of surgical specimens showed polyclonal plasma cells and mature lymphocytes but no etiological agent. A histopathologic diagnosis of intracranial PCG was made in both cases. PCG should be part of the differential diagnosis of enhancing mass lesions of the brain. The etiology and natural history of these tumor-like lesions is not fully understood. Complete surgical excision appears to be curative. Lesions where total resection is not possible may benefit from adjuvant treatment including corticosteroids and possibly radiation therapy.
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Louis DN, Ohgaki H, Wiestler OD, Cavenee WK, Burger PC, Jouvet A, Scheithauer BW, Kleihues P (2007) The 2007 WHO classification of tumours of the central nervous system. Acta Neuropathol 114:97–109
Bahadori M, Liebow AA (1973) Plasma cell granulomas of the lung. Cancer 31:191–208
Adler A, Fimbres A, Marcinak J, Johnson A, Zheng X, Hasegawa S, Shulman ST (2009) Inflammatory pseudotumor of the heart caused by Listeria monocytogenes infection. J Infect 58:161–163
Das N, Lee J, Madden M, Elliot CS, Bateson P, Gilliland R (2006) A rare case of abdominal actinomycosis presenting as an inflammatory pseudotumour. Int J Colorectal Dis 21:483–484
Buccoliero AM, Caldarella A, Santucci M, Ammannati F, Mennonna P, Taddei A, Taddei GL (2003) Plasma cell granuloma—an enigmatic lesion: description of an extensive intracranial case and review of the literature. Arch Pathol Lab Med 127:e220–e223
Kim DJ, Choi YS, Song YJ, Kim KU (2009) Intracranial plasma cell granuloma. J Korean Neurosurg Soc 46:161–164
Arber DA, Kamel OW, van de Rijn M, Davis RE, Medeiros LJ, Jaffe ES, Weiss LM (1995) Frequent presence of the Epstein–Barr virus in inflammatory pseudotumor. Hum Pathol 26:1093–1098
Arber DA, Weiss LM, Chang KL (1998) Detection of Epstein–Barr virus in inflammatory pseudotumor. Semin Diagn Pathol 15:155–160
Gonzalez-Duarte A, Sullivan S, Sips GJ, Naidich T, Kleinman G, Murray J, Morgello S, Germano I, Mullen M, Simpson D (2009) Inflammatory pseudotumor associated with HIV, JCV, and immune reconstitution syndrome. Neurology 72:289–290
Hausler M, Schaade L, Ramaekers VT, Doenges M, Heimann G, Sellhaus B (2003) Inflammatory pseudotumors of the central nervous system: report of 3 cases and a literature review. Hum Pathol 34:253–262
Warter A, Satge D, Roeslin N (1987) Angioinvasive plasma cell granulomas of the lung. Cancer 59:435–443
Coffin CM, Watterson J, Priest JR, Dehner LP (1995) Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 19:859–872
Cerase A, Tarantino A, Gozzetti A, Muccio CF, Gennari P, Monti L, Di Blasi A, Venturi C (2008) Intracranial involvement in plasmacytomas and multiple myeloma: a pictorial essay. Neuroradiology 50:665–674
Gasent Blesa JM, Alberola Candel V, Solano Vercet C, Laforga Canales J, Semler C, Perez Antoli MR, Rodriguez-Galindo C (2008) Langerhans cell histiocytosis. Clin Transl Oncol 10:688–696
Favara BE, Feller AC, Pauli M, Jaffe ES, Weiss LM, Arico M, Bucsky P, Egeler RM, Elinder G, Gadner H, Gresik M, Henter JI, Imashuku S, Janka-Schaub G, Jaffe R, Ladisch S, Nezelof C, Pritchard J (1997) Contemporary classification of histiocytic disorders. The WHO committee on histiocytic/reticulum cell proliferations. Reclassification working group of the histiocyte society. Med Pediatr Oncol 29:157–166
Conley A, Manjila S, Guan H, Guthikonda M, Kupsky WJ, Mittal S (2010) Non-Langerhans cell histiocytosis with isolated CNS involvement: An unusual variant of Erdheim–Chester disease. Neuropathology 30:634–647
Weber MA, Viehoever A, Stieltjes B, Zoubaa S, Wildemann B, Baudendistel K, Kauczor HU (2005) Intracerebral manifestation of an atypical monoclonal plasma cell hyperplasia depicted by MR perfusion and diffusion tensor imaging and MR spectroscopy. AJR Am J Roentgenol 185:784–787
Fukunaga A, Yoshida K, Otani M, Ogawa Y, Horiguchi T, Ishihara M, Toya S, Kawase T (1998) Plasma cell granuloma extending from the extracranial to the intracranial space associated with Epstein–Barr virus infection. Neurol Med Chir (Tokyo) 38:292–296
Morrison A, Gyure KA, Stone J, Wong K, McEvoy P, Koeller K, Mena H (1999) Mycobacterial spindle cell pseudotumor of the brain: a case report and review of the literature. Am J Surg Pathol 23:1294–1299
Kiryu S, Takeuchi K, Shibahara J, Uozaki H, Fukayama M, Tanaka H, Maeda E, Akahane M, Ohtomo K (2009) Epstein–Barr virus-positive inflammatory pseudotumour and inflammatory pseudotumour-like follicular dendritic cell tumour. Br J Radiol 82:e67–e71
Wisser J, Zingman B, Wasik M, Duva-Frissora A, Beazley R, McAneny D (1992) Cytomegalovirus pseudotumor presenting as bowel obstruction in a patient with acquired immunodeficiency syndrome. Am J Gastroenterol 87:771–774
Sato K, Kubota T, Kitai R, Miyamori I (2006) Meningeal plasma cell granuloma with relapsing polychondritis. Case report. J Neurosurg 104:143–146
Chang Y, Horoupian DS, Lane B, Fross RD, Smyth LT Jr, Seiling RJ (1991) Inflammatory pseudotumor of the choroid plexus in Sjogren’s disease. Neurosurgery 29:287–290
Nawashiro H, Omura T, Kobayashi H (2006) Cystic intracranial plasma cell granuloma. J Neurosurg 105:799 author reply 799–800
Qi XL, Chen J, Zheng RL, Li Y, Huang YN, Yuan Y (2009) Tumor-like inflammatory demyelinating diseases of the central nervous system with relapse onset: a case. Beijing Da Xue Xue Bao 41:588–589
Shenoy SN, Raja A (2004) Intracranial plasma cell granuloma. Neurol India 52:262–264
Ozum U, Ozer H, Karadag O, Polat N (2006) Intracranial plasma cell-granuloma with extensive ossification. Br J Neurosurg 20:153–156
Roche PH, Figarella-Branger D, Pellet W (2004) Mixed meningeal and brain plasma-cell granuloma: an example of an unusual evolution. Acta Neurochir (Wien) 146:69–72 discussion 72
Vicuna-Gonzalez RM, Rivera-Salgado MI, Garcia-Velarde PM, de Leon-Bojorge B, Ortiz-Hidalgo C (2008) Multicentric inflammatory pseudotumor with asynchronic presentation in meninges, liver, spleen and lymph nodes in a patient with seronegative spondiloarthropathy. Case report and review of the literature. Rev Neurol 47:464–468
Wong S, Lam WY, Wong WK, Lee KC (2007) Hypophysitis presented as inflammatory pseudotumor in immunoglobulin G4-related systemic disease. Hum Pathol 38:1720–1723
Jung TY, Jung S, Lee MC, Moon KS, Kim IY, Kang SS, Kim SH (2006) Hemorrhagic intracranial inflammatory pseudotumor originating from the trigeminal nerve: a case report. J Neurooncol 76:139–142
Brito CC, Lopes FC, Chimelli L, Gasparetto EL (2010) Intracranial cell plasma granuloma. Arq Neuropsiquiatr 68:127–129
Cannella DM, Prezyna AP, Kapp JP (1988) Primary intracranial plasma-cell granuloma. Case report. J Neurosurg 69:785–788
Figarella-Branger D, Gambarelli D, Perez-Castillo M, Garbe L, Grisoli F (1990) Primary intracerebral plasma cell granuloma: a light, immunocytochemical, and ultrastructural study of one case. Neurosurgery 27:142–147
Gangemi M, Maiuri F, Giamundo A, Donati P, De Chiara A (1989) Intracranial plasma cell granuloma. Neurosurgery 24:591–595
Radi-Bencteux S, Proust F, Vannier JP, Marret S, Laquerriere A (2003) Intracerebral inflammatory pseudotumour in a 16-month-old boy. Neuropediatrics 34:330–333
Wilner HI, Vinas FC, Duffy C, Kupsky WJ, Guthikonda M (1999) Magnetic resonance imaging of intercranial plasmocytic granuloma. Skull Base Surg 9:265–270
Brandsma D, Jansen GH, Spliet W, Van Nielen K, Taphoorn MJ (2003) The diagnostic difficulties of meningeal and intracerebral plasma cell granulomas—presentation of three cases. J Neurol 250:1302–1306
Dettmeyer R, Varchmin-Schultheiss K, Madea B (1998) Intracranial plasma cell granuloma and homocystinuria. Pathol Res Pract 194:205–208
Tresser N, Rolf C, Cohen M (1996) Plasma cell granulomas of the brain: pediatric case presentation and review of the literature. Childs Nerv Syst 12:52–57
Gochman GA, Duffy K, Crandall PH, Vinters HV (1990) Plasma cell granuloma of the brain. Surg Neurol 33:347–352
Greiner C, Rickert CH, Mollmann FT, Rieger B, Semik M, Heindel W, Wassmann H (2003) Plasma cell granuloma involving the brain and the lung. Acta Neurochir (Wien) 145:1127–1131
Lyo IU, Suh JH, Kwon Y (2001) Intracranial plasma cell granuloma: a case report. J Korean Neurosurg Soc 30:774–776
Tang TT, Segura AD, Oechler HW, Harb JM, Adair SE, Gregg DC, Camitta BM, Franciosi RA (1990) Inflammatory myofibrohistiocytic proliferation simulating sarcoma in children. Cancer 65:1626–1634
Chan YF, White J, Brash H (1994) Metachronous pulmonary and cerebral inflammatory pseudotumor in a child. Pediatr Pathol 14:805–815
Malhotra V, Tatke M, Malik R, Gondal R, Beohar PC, Kumar S, Puri V (1991) An unusual case of plasma cell granuloma involving lung and brain. Indian J Cancer 28:223–227
Kilinç M, Ertürk IO, Uysal H, Birler K, Evrenkaya T, Akkalyoncu BB (2002) Multiple plasma cell granuloma of the central nervous system: a unique case with brain and spinal cord involvement. Case report and review of literature. Spinal Cord 40:203–206
Flannery T, Al-Sabah F, Bhangu J, Alderazi Y, Brett F, Pidgeon C (2007) Treatment of subtotally resected intracranial plasma cell granuloma with steroids: a case report. Br J Neurosurg 21:501–503
Shah MD, McClain KL (2005) Intracranial plasma cell granuloma: case report and treatment of recurrence with methotrexate and 6-mercaptopurine. J Pediatr Hematol Oncol 27:599–603
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We would like to thank Dr. Monika Mittal and Mr. Brandon Parker in the Department of Neurosurgery for editorial assistance.
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Puntambekar, P., Santhakumar, S., Kupsky, W.J. et al. Primary intracranial plasma cell granulomas presenting as malignant neoplasms. J Neurooncol 106, 327–337 (2012). https://doi.org/10.1007/s11060-011-0667-5
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DOI: https://doi.org/10.1007/s11060-011-0667-5