Bone haemophilic pseudotumour of the ulna: A rare complication of haemophilia in a dog

Summary

Objectives: This case report describes for the first time a bone haemophilic pseudotumour in a dog.

Case description: A seven-month-old German Shepherd male dog was presented with the complaint of a forelimb weight-bearing lameness with major swelling that expanded dramatically after fine needle aspiration. Radiographs showed a large, well-defined ulnar diaphyseal cystic-like osteolytic lesion. Based on prolonged activated partial thromboplastin time (aPTT) and low factor VIII activity, haemophilia A was diagnosed. Bone scintigraphy, computed tomography, magnetic resonance imaging, and histological findings definitely ruled out malignant neoplasia or inflammation and strongly supported a bone haemophilic pseudotumour over an aneurysmal bone cyst. Segmental ulnar resection and replacement by a polymethylmethacrylate spacer combined with perioperative bleeding management resulted in a successful outcome.

Discussion: This case provided evidence that a bone haemophilic pseudotumour may be the sole presenting clinical sign of haemophilia A in dogs. Early diagnosis, based on history and magnetic resonance imaging findings, is imperative for prompt treatment leading to successful outcome. It is challenging as fine needle aspiration or biopsy is contraindicated. As described in humans, surgical excision of the lesion combined with management of severe postoperative bleeding was associated with successful outcome in the present case.

Clinical significance: A bone haemophilic pseudotumour should be considered in the differential diagnosis of expanding mass associated with osteolysis, especially in young male dogs. Perioperative monitoring of the bleeding disorder and subsequent FVIII replacement therapy was of paramount importance in the present case.

Keywords

Diagnostic imaging - factor VIII - haemophilia - bone tumour - dog

• References

• 1 Aslanian ME, Sharp CR, Rozanski EA. et al. Clinical outcome after diagnosis of hemophilia A in dogs. J Am Vet Med Assoc 2014; 245: 677-683.
• 2 Low SF, Sridharan R, Ngiu CS. et al. Osseous haemophilic pseudotumour and concurrent primary hyperparathyroidism: a diagnostic conundrum. BMJ Case Rep 2014: pii: bcr2013203282doi: 10.1136/bcr-2013 -203282.
• 3 Fernandezde V, Spain M, Matthews JM. The haemophilic pseudotumour or haemophilic subperiosteal haematoma. J Bone Joint Surg Br 1965; 47: 256-265.
• 4 Jaovisidha S, Ryu KN, Hodler J. et al. Hemophilic pseudotumor: spectrum of MR findings. Skeletal Radiol 1997; 26: 468-474.
• 5 Magallon M, Monteagudo J, Altisent C. et al. Hemophilic pseudotumor: multicenter experience over a 25-year period. Am J Hematol 1994; 45: 103-108.
• 6 Lim MY, Nielsen B, Ma A. et al. Clinical features and management of haemophilic pseudotumours: a single US centre experience over a 30-year period. Haemophilia 2014; 20: e58-62.
• 7 Panotopoulos J, Ay C, Trieb K. et al. Surgical treatment of the haemophilic pseudotumour: a single centre experience. Int Orthop 2012; 36: 2157-2162.
• 8 Rodriguez-Merchan EC. The haemophilic pseudo-tumour. Haemophilia 2002; 8: 12-16.
• 9 Ishiguro N, Iwahori Y, Kato T. et al. The surgical treatment of a haemophilic pseudotumour in an extremity: a report of three cases with pathological fractures. Haemophilia 1998; 4: 126-131.
• 10 Espandar R, Heidari P, Rodriguez-Merchan EC. Management of haemophilic pseudotumours with special emphasis on radiotherapy and arterial embolization. Haemophilia 2009; 15: 448-457.
• 11 Gilbert MS. The hemophilic pseudotumor. Prog Clin Biol Res 1990; 324: 257-262.
• 12 Mohanty SS, Bhasme VK, Garg H. et al. The haemophilic pseudotumour – surgical treatment by excision and filling the defect with calcium-phosphate cement granules. Haemophilia 2007; 13: 217-220.
• 13 Stafford JM, James TT, Allen AM. et al. Hemophilic pseudotumor: radiologic-pathologic correlation. Radiographics 2003; 23: 852-856.
• 14 Pruzansky JS, Gilbert MS, Garcia RA. et al. Intraosseous pseudotumor of the distal radius in a patient with hemophilia: case report. J Hand Surg Am 2012; 37: 532-537.
• 15 O’Donnell PG. Cystic lesions of bone. In Davies MA, Sundaram M, James SJ. editors Imaging of Bone Tumors and Tumor-Like Lesions: Techniques and Applications. Berlin: Springer Verlag; 2009: 425
• 16 Nomura K, Sato K. Pelvic aneurysmal bone cyst in a dog. J Vet Med Sci 1997; 59: 1027-1030.
• 17 Barnhart MD. Malignant transformation of an aneurysmal bone cyst in a dog. Vet Surg 2002; 31: 519-524.
• 18 Benamou J, Lussier B, Alexander K. et al. Use of magnetic resonance imaging and histopathologic findings for diagnosis of an aneurysmal bone cyst in the scapula of a cat. J Am Vet Med Assoc 2012; 240: 69-74.
• 19 Jaganathan S, Gamanagatti S, Goyal A. Musculoskeletal manifestations of hemophilia: imaging features. Curr Probl Diagn Radiol 2011; 40: 191-197.
• 20 Hudson TM. Scintigraphy of aneurysmal bone cysts. AJR Am J Roentgenol 1984; 142: 761-765.
• 21 Gilbert MS. Characterizing the hemophilic pseudotumor. Ann N Y Acad Sci 1975; 240: 311-315.
• 22 Fang J, Feng X, Lin C. et al. Haemophilic pseudo-tumour with fistulae formation: a retrospective report of five infected cases. Haemophilia 2016; 22: e54-56.
• 23 Caviglia H, Candela M, Landro ME. et al. Haemophilia pseudotumours in patients with inhibitors. Haemophilia 2015; 21: 681-685.
• 24 Allen DJ, Goddard NJ, Mann HA. et al. Primary malignancies mistaken for pseudotumours in haemophilic patients. Haemophilia 2007; 13: 383-386.
• 25 Park JS, Ryu KN. Hemophilic pseudotumor involving the musculoskeletal system: spectrum of radiologic findings. AJR Am J Roentgenol 2004; 183: 55-61.
• 26 Hermann G, Yeh HC, Gilbert MS. Computed tomography and ultrasonography of the hemophilic pseudotumor and their use in surgical planning. Skeletal Radiol 1986; 15: 123-128.
• 27 Thomas ML, Walters HL. The angiographic findings in a haemophilic pseudotumour of bone. Australas Radiol 1977; 21: 346-349.
• 28 Sevilla J, Alvarez MT, Hernandez D. et al. Therapeutic embolization and surgical excision of haemophilic pseudotumour. Haemophilia 1999; 5: 360-363.