Hemophilic Pseudotumor: A Case Report and Review of Literature

Aneesh Pakala, Jimmy Thomas, Philip Comp

Department of Internal Medicine, University of Oklahoma Health Science Center, Oklahoma City, USA..

DOI: 10.4236/ijcm.2012.33046                

Abstract

A hemophilic pseudotumor is one of the rarer complications of hemophilia that results from repetitive bleeding resulting in an encapsulated mass of clotted blood and necrosed tissue. These have become rarer over the years with better treatment modalities for bleeding disorders like factor replacement. In this case report, we describe the natural history and clinical course of a patient with hemophilia who presented with a massive pseudotumor. A review of literature follows which explores all the relevant data to date on the presentation, complications, diagnostic modalities and therapies that have been implemented in the management of this rare complication of a common bleeding disorder.

Keywords

Hemophilia; Pseudotumor

Conflicts of Interest

The authors declare no conflicts of interest.

References

[1]	C. T. A. Brower, “Hemophilia A,” Gene Reviews, 1993.
[2]	C. T. A. Brower, “Hemophilia B,” Gene Reviews, 1993.
[3]	R. Garcia-Perez, G. Torres-Salmeron, F. Sanchez-Bueno, A. Garcia-Lopez and P. Parrilla-Paricio, “Intraabdominal Hemophilic Pseudotumor: Case Report,” Revista Espa?ola de Enfermedades Digestivas, Vol. 102, No. 4, 2010, pp. 275-280. doi:10.4321/S1130-01082010000400009
[4]	W. W. Coon and J. A. Penner, “Management of Abdominal Hemophilic Pseudotumor,” Surgery, Vol. 90, No. 4, 1981, pp. 735-40.
[5]	L. Starker, “Knochenusur Durch ein Hamophiles Subperiostales Hamatom,” Mitteilungen aus den Grenzgebieten der Medizin und Chirurgie, Vol. 31, 1918, pp. 381-415.
[6]	T. R. Lewis, H. R. Webb, J. P. Bell, D. P. Beall and J. R. Fish, “Hemophilic Pseudo-tumor,” The Journal of the Oklahoma State Medical As-sociation, Vol. 98, No. 10, 2005, pp. 485-487.
[7]	F. W. Wright, J. M. Matthews, “Hemophilic Pseudotumor of the Stomach,” Radiology, Vol. 98, No. 3, 1971, pp. 547-549.
[8]	H. Pettersson and A. Ahlberg, “Computed Tomography in Hemophilic Pseudotumor,” Acta Radiologica: Diagno- sis (Stockh), Vol. 23, No. 5, 1982, pp. 453-457.
[9]	M. S. Gilbert, “Characterizing the Hemophilic Pseudotumor,” Annals of the New York Academy of Sciences, Vol. 240, 1975, pp. 311-315. doi:10.1111/j.1749-6632.1975.tb53365.x
[10]	H. Pet-tersson, I. M. Nilsson, U. Hedner, K. Norehn and A. Ahlberg, “Radiologic Evaluation of Prophylaxis in Severe Haemophilia,” Acta Paediatrica, Vol. 70, No. 4, 1981, pp. 565-570. doi:10.1111/j.1651-2227.1981.tb05741.x
[11]	C. A. Lee, E. E. Berntorp and W. K. Hoots, Eds., “Textbook of Hemophilia,” 2nd Edition, Wiley-Blackwell, Hoboken, 2010.
[12]	S. Castro-Boix, J. Pradell-Teigell, R. Boque-Genovard, V. Zanon-Navarro, A. Nadal-Guinard, C. Altisent-Roca, et al., “Hemophilic Pelvic Pseudotumor,” Cirugía Espa?ola, Vol. 81, No. 2, 2007, pp. 102-104. doi:10.1016/S0009-739X(07)71273-3
[13]	J. S. Park and K. N. Ryu, “Hemophilic Pseudotumor Involving the Musculoskeletal System: Spectrum of Radiologic Findings,” American Journal of Roentgenology, Vol. 183, No. 1, 2004, pp. 55-61.
[14]	E. E. Brant and H. H. Jordan, “Radiologic Aspects of Hemophilic Pseudotumors in Bone,” American Journal of Roentgenology, Vol. 115, No. 3, 1972, pp. 525-539.
[15]	S. Bicknell and A. Mason, “Hemophilic Pseudotumor of the Chest,” Journal of Thoracic Imaging, Vol. 16, No. 3, 2001, pp. 188-190. doi:10.1097/00005382-200107000-00010
[16]	H. Grossman, W. E. Berdon and D. H. Baker, “Reversible Gastrointestinal Signs of Hemorrhage and Edema in the Pediatric Age Group,” Radiology, Vol. 84, No. 1, 1965, pp. 33-39.
[17]	J. D. Rothstein, W. R. Sandusky and T. E. Keats, “Hematoma as a Cause of Radiographic Deformity of Stomach,” Radiology, Vol. 90, No. 1, 1968, pp. 116-117.
[18]	W. M. Steel, R. B. Duthie and B. T. O’Connor, “Haemophilic Cysts. Report of Five Cases,” Journal of Bone & Joint Surgery, Vol. 51, No. 4, 1969, pp. 614-626.
[19]	I. M. Nilsson, U. Hedner, A. Ahlberg, S. A. Larsson and S. E. Bergentz, “Surgery of Hemophiliacs—20 Years’ Experience,” World Journal of Surgery, Vol. 1, No. 1, 1977, pp. 55-66. doi:10.1007/BF01654733
[20]	Y. A. Kilic, S. V. Dundar, D. Onat and O. Akhan, “Iliopsoas Hemophilic Pseudo-tumor with Bowel Fistulization,” Bratisl Lek Listy, Vol. 110, No. 11, 2009, pp. 729-731.
[21]	S. Jaovisidha, K. N. Ryu, J. Hodler, M. E. Schweitzer, D. J. Sartoris and D. Resnick, “Hemophilic Pseudotumor: Spectrum of MR Findings,” Skeletal Radiology, Vol. 26, No. 8, 1997, pp. 468-474. doi:10.1007/s002560050268
[22]	M. S. Gilbert, Ed., “Haemophilic Pseudotumor,” Excerpta Medica, Amsterdam, 1975.
[23]	M. Magallon, J. Monteagudo, C. Altisent, A. Ibanez, A. Rodriguez-Perez, J. Riba, et al., “Hemophilic Pseudotumor: Multicenter Experience over a 25-Year Period,” The American Journal of Hematology, Vol. 45, No. 2, 1994, pp. 103-108. doi:10.1002/ajh.2830450202
[24]	R. Hoffman, Ed., “Hematology: Basic Principles and Practice,” 3rd Edition, Churchill Livingstone, New York, 2003.
[25]	M. Sundaram, M. K. Wolverson, J. H. Joist, M. A. Riaz and B. J. Rao, “Case Report 133. Hemophilic Pseudotumor of Ilium and Soft Tissues,” Skeletal Radiology, Vol. 6, No. 1, 1981, pp. 54-57. doi:10.1007/BF00347349
[26]	W. B. Guilford, P. D. Mintz, P. M. Blatt and E.V. Staab, “CT of Hemophilic Pseudotumors of the Pelvis,” American Journal of Roentgenology, Vol. 135, No. 1, 1980, pp. 167-169.
[27]	G. Hermann, H. C. Yeh and M. S. Gilbert, “Computed Tomography and Ultrasonography of the Hemophilic Pseudotumor and Their Use in Surgical Planning,” Skeletal Radiology, Vol. 15, No. 2, 1986, pp. 123-128. doi:10.1007/BF00350205
[28]	J. M. Stafford, T. T. James, A. M. Allen and L. R. Dixon, “Hemophilic pseudotumor: Radiologic-Pathologic Correlation,” Radio-graphics, Vol. 23, No. 4, 2003, pp. 852-856. doi:10.1148/rg.234025154
[29]	T. Morissy and S. L. Weinstein, Eds., “Pediatric Orthopedics,” 6th Edition, Lippincott Williams & Wilkins, Philadelphia, 2006.
[30]	E. Gaary, J. B. Gorlin and D. Jaramillo, “Pseudotumor and Arthropathy in the Knees of a Hemophiliac,” Skeletal Radiology, Vol. 25, No. 1, 1996, pp. 85-87. doi:10.1007/s002560050040
[31]	A. K. Ahlberg, “On the Natural History of Hemophilic Pseudotumor,” Journal of Bone and Joint Surgery, Vol. 57, No. 8, 1975, pp. 1133-6.
[32]	R. L. Rosenthal, J. J. Graham and E. Selirio, “Excision of Pseudotumor with Repair by Bone Graft of Pathological Fracture of Femur in Hemophilia,” Journal of Bone and Joint Surgery, Vol. 55, No. 4, 1973, pp. 827-832.
[33]	J. M. Pisco, V. L. Garcia, J. M. Martins and A. M. Mascarenhas, “Hemophilic Pseudotumor Treated with Transcatheter Arterial Embolization: Case Report,” Angiology, Vol. 41, No. 12, 1990, pp. 1070-1074. doi:10.1177/000331979004101208
[34]	F. Alaniz Camino and E. Lozano, “Hemophilic Pseudotumor Treated with Radiation,” Revista de Investigación Clínica, Vol. 33, No. 3, 1981, pp. 281-283.
[35]	M. W. Hilgartner and W. D. Arnold, “Hemophilic Pseudotumor Treated with Replacement Therapy and Radiation. Report of a Case,” Journal of Bone and Joint Surgery, Vol. 57, No. 8, 1975, pp. 1145-1146.
[36]	M. Issaivanan, M. P. Shrikande, M. Mahapatra and V. P. Choudhry, “Management of Hemophilic Pseudotumor of Thumb in a Child,” Journal of Pediatric Hematology/ Oncology, Vol. 26, No. 2, 2004, pp. 128-132. doi:10.1097/00043426-200402000-00016
[37]	H. S. Reinhold, “The Influence of Radiation on Blood Vessels and Circulation. Chapter IV. Structural Changes in Blood Vessels,” Current Topics in Radiation Research Quarterly, Vol. 10, No. 1, 1974, pp. 58-74.
[38]	L. Meyers and N. Hakami, “Pseudotumor of Hemophilia in the Orbit: The Role of Radiotherapy in Management,” The American Journal of Hematology, Vol. 19, No. 1, 1985, pp. 99-104. doi:10.1002/ajh.2830190113