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Massive pelvic osteolysis in the Gorham-Stout syndrome

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Summary

Massive osteolysis is a very rare tumour-like lesion usually affecting young adults. There are 132 reported cases which we have reviewed, including 31 single cases of pelvic involvement. The term “massive osteolysis” is based on the typical radiological findings, such as increasing translucency and loss of bone density. The diagnosis of the Gorham-Stout syndrome must be confirmed by the microscopic finding of intramedullary vascular structures. Our patient was a 23 year old white Caucasian, who had the full clinical signs of Gorham-Stout syndrome. She had achieved 2 normal pregnancies and deliveries in spite of her massive pelvic osteolysis.

Résumé

L'ostéolyse massive est une lésion pseudotumorale extrêmement rare, qui atteint habituellement les adultes jeunes. Il en existe 132 cas publiés, qui ont été passés en revue, parmi lesquels 31 cas d'atteinte limitée au bassin. Le terme «d'ostéolyse massive» est basé sur les images radiologiques typiques, telles que l'augmentation de la transparence et la diminution de la densité de l'os. Le diagnostic de syndrome de Gorham-Stout doit être étayé par des constatations microscopiques concernant les structures vasculaires intra-médullaires. Notre malade est une européenne blanche de 23 ans qui présente le tableau complet du syndrome de Gorham-Stout. Elle a eu deux grossesses et deux accouchements normaux malgrè son ostéolyse massive du bassin.

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References

  1. Abel MS, Smith GR (1974) The case of disappearing pelvis. Radiology 111: 105–106

    Google Scholar 

  2. Abrahams J, Ganick D, Gilbert E, Wolfson J (1980) Massive osteolyse in an infant. Am J Radiol 135: 1084–1086

    Google Scholar 

  3. Bickel WH, Broders AC (1947) Primary lymphangioma of the ileum. J Bone Joint Surg 29-A: 517–523

    Google Scholar 

  4. Branco F, da Silva Horta J (1958) Notes of a rare case of essential osteolysis. J Bone Joint Surg 40-B: 519–527

    Google Scholar 

  5. Bullough PG (1971) Massive osteolysis. NY State J Med 71: 2267–2278

    Google Scholar 

  6. Butler RW, McCance RA, Barrett AM (1958) Unexplained destruction of the shaft of the femur in a child. J Bone Joint Surg 40-B: 487–493

    Google Scholar 

  7. Cannon SR (1986) Massive osteolysis — A review of seven cases. J Bone Joint Surg 68-B: 24–28

    Google Scholar 

  8. Campbell J, Almond HGA, Johnson R (1975) Massive osteolysis of the humerus with spontaneous recovery — Report of a case. J Bone Joint Surg 57-B: 238–240

    Google Scholar 

  9. Castleman B, McNeely BU (1964) Case records of the Massachusetts General Hospital — Case 16–1964. N Engl J Med 270: 731–736

    Google Scholar 

  10. Catalan A, Ignacio AC (1978) Vanishing bone disease: A case report. Hawaii Med J 37: 302–304

    Google Scholar 

  11. Coley BL (1949) Neoplasms of bone and related conditions. Their etiology, pathogenesis, diagnosis, and treatment. P. B. Hoeber, Inc., Medical Book Department of Harper & Brothers, New York, pp 688–698

    Google Scholar 

  12. Donaldson JR, Marshall CE (1962) Massive osteolysis. Indian J Surg 24: 136–140

    Google Scholar 

  13. Edwards WH Jr, Thompson RC, Varsa EW (1983) Lymphangiomatosis and massive osteolysis of the cervical spine. A case report and review of the literature. Clin Orthop 177: 222–229

    Google Scholar 

  14. El-Mofty S (1971) Atrophy of the mandible (massive osteolysis). Oral Surg 31: 690–700

    Google Scholar 

  15. Falkmer S, Tilling G (1957) Primary lymphangioma of bone. Acta Orthop Scand 26: 99–110

    Google Scholar 

  16. Feigl D, Seidel L, Marmor A (1981) Gorham's disease of the clavicle with bilateral pleural effusions. Chest 79: 242–244

    Google Scholar 

  17. Fornasier VL (1970) Haemangiomatosis with massive osteolysis. J Bone Joint Surg 52-B: 444–451

    Google Scholar 

  18. Gorham LW, Stout AP (1954) Hemangiomatosis and its relation to massive osteolysis. Trans Ass Am Phys 67: 302–307

    Google Scholar 

  19. Gorham LW, Stout AP (1955) Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone). J Bone Joint Surg 37-A: 985–1004

    Google Scholar 

  20. Gorham LW, Wright AW, Schultz HH, Maxon FC Jr (1954) Disappearing bones: a rare form of massive osteolysis. Report of two cases, one with autopsy findings. Am J Med 17: 674–682

    Google Scholar 

  21. Haas JP, Reichelt A (1966) Die isolierte Knochenlymphangiomatose — eine familiäre Erkrankung? Röntgenforum 105: 733–755

    Google Scholar 

  22. Haferkamp O (1961) Über das Syndrome: Generalisierte maligne Hämangiomatose mit Osteolyse. Z Krebsforsch 64: 418 ff

    Google Scholar 

  23. Halliday DR, Dahlin DC, Pugh DG, Young HH (1964) Massive osteolysis and angiomatosis. Radiology 82: 637–644

    Google Scholar 

  24. Hambach R, Pujman J, Maly V (1958) Massive osteolysis due to hemangiomatosis. Report of a case of Gorham's disease with autopsy. Radiology 71: 43–47

    Google Scholar 

  25. Hartmann G (1947) Osteophthisis pelvis et femorum. Zugleich ein Beitrag zur Frage der spontanen Rückbildung maligner Tumoren. Maudrich, Wien

    Google Scholar 

  26. Hejgaard N, Olsen PR (1987) Massive Gorham osteolysis of the right hemipelvis complicated by chylothorax: Report of a case in a 9-year-old boy successfully treated by pleurodesis. J Pediatr Orthop 7: 96–99

    Google Scholar 

  27. Henry AN (1984) see Picault et al. (1984)

    Google Scholar 

  28. Heyden G, Kinblom L-G, Möller Nielsen J (1977) Disappearing bone disease. A clinical and histological study. J Bone Joint Surg 59-A: 57–61

    Google Scholar 

  29. Holland C, Werner H (1966) Über Osteolysen. Arch Orthop Unfall-Chir 60: 317–339

    Google Scholar 

  30. Horst M, Zsernaviczky J, Delling G (1979) Ein seltener Fall von sogenannter ideopathischer Osteolyse, assoziiert mit einem Lymphangiom der Fibula. Z Orthop 117: 89–95

    Google Scholar 

  31. Jackson JBS (1838) A singular case of absorption of bone (a boneless arm). Boston Med Surg J 18: 368–369

    Google Scholar 

  32. Jackson JBS (1872) Absorption of the humerus after fracture. Report to the Boston Society of Medical Improvement, by the various gentlemen under whose observation the case had fallen. Boston Med Surg J 10: 245–247

    Google Scholar 

  33. Jacobs P (1964) Post-traumatic osteolysis of the outer end of the clavicle. J Bone Joint Surg 46-B: 705–707

    Google Scholar 

  34. Jesserer H (1963) Atlas der Knochen- und Gelenkkrankheiter. E Merck AG, Darmstadt, pp 38–39

    Google Scholar 

  35. Jesserer H (1971) Knochenkrankheiten. Urban Schwarzenberg, München Berlin Wien, pp 141–143

    Google Scholar 

  36. Johnson PM, McClure JG (1958) Observations on massive osteolysis. Radiology 71: 28–42

    Google Scholar 

  37. Jones GB, Midgley RL, Smith GS (1958) Massive osteolysis-disappearing bones. J Bone Joint Surg 40-B: 494–501

    Google Scholar 

  38. Imbert JC, Picault C (1974) Ostéolyse massive ideopatique ou maladie de Jackson-Gorham. A propos d'une nouvelle observation. Rev Chir Orthop 60: 73–80

    Google Scholar 

  39. Kery L, Wouters HW (1970) Massive osteolysis. Report of two cases. J Bone Joint Surg 52-B: 452–459

    Google Scholar 

  40. King DJ (1946) A case resembling hemangiomatosis of the lower extremity. J Bone Joint Surg 28: 623–628

    Google Scholar 

  41. Knoch H-G (1963) Die Gorhamsche Krankheit aus klinischer Sicht. Zentralbl Chir 18: 674–683

    Google Scholar 

  42. Knoch H-G (1964) Die Osteolysen im Bereich des Schultergürtels. Beitr Orthop 11: 169–170

    Google Scholar 

  43. Krikler DM (1955) The case of the vanishing toes. A Afr Med J 29: 1050–1052

    Google Scholar 

  44. Kulenkampff H-A, Adler CP (1987) Radiologische und Pathologische Befunde beim Gorham-Stout-Syndrom (Massive Osteolyse). Verh Dtsch Ges Pathol 71: 574

    Google Scholar 

  45. Kulenkampff H-A, Richter GM, Adler C-P, Haase WE (1989) Massive Osteolyse (Gorham-Stout-Syndrom) — Klinik, Diagnostik, Therapie und Prognose. Vortrag zur Jahrestagung der Deutschen Gesellschaft für Osteologie e. V., Göttingen

    Google Scholar 

  46. Kyllonen AS (1967) Disappearing ribs. Ann Thorac Surg 4: 559–563

    Google Scholar 

  47. Leger L, Ducroquet R, Leger H (1949) Ostéolyse dite essentielle. In: Maladies du Squelette. Masson et Cie, Paris, pp 190–207

    Google Scholar 

  48. Leriche R (1937) A propos des ostéolyses d'orgine indéterinée. Mem de l'Acad de Chir 63: 419–421

    Google Scholar 

  49. Louyot P, Gaucher A, Benoît P, Combebias JF (1966) Ostéolyse massive par hémangiome capillaire dysgénétique. J Radiol Électrol de Méd Nucl 47: 458–461

    Google Scholar 

  50. Milner SM, Baker SL (1958) Disappearing bones. J Bone Joint Surg 40-B: 502–513

    Google Scholar 

  51. Mouchet A, Rouvillois H (1937) Ostéolyse du bassin d'origine indéterminée. Mém Acad de Chir 63: 277–286

    Google Scholar 

  52. Nicod L (1945) Osteolyse. Helv Chir Acta 12: 331–340

    Google Scholar 

  53. Nicod L (1946) Un cas rare d'ostéolyse posttraumatique de l'avant-bras. Schweiz Med Wochenschr 76: 59

    Google Scholar 

  54. Pastakia B, Horvath K, Lack EE (1987) Seventeen year follow-up and autopsy findings in a rare case of massive osteolysis. Skelet Radiol 16: 291–297

    Google Scholar 

  55. Patrick JH (1976) Massive osteolysis complicated by chylothorax successfully treated by pleurodesis. J Bone Joint Surg 58-B: 347–349

    Google Scholar 

  56. Phemister DB (1948) The pathology of vascular tumors of bone. In: The American Academy of Orthopeadic Surgeons: instructional course lectures. J. W. Edwards Co, Ann Arbor Vol 5, pp 19–26

    Google Scholar 

  57. Phillips RM, Bush OB Jr, Hall HD (1972) Massive osteolysis (phantom bone, disappearing bone). Report of a case with mandibular involvement. Oral Surg 34: 886–896

    Google Scholar 

  58. Picault C, Comtet JJ, Imbert JC, Boyer JM (1984) Surgical repair of extensive ideopathic osteolysis of the pelvic girdle (Jackson-Gorham Disease). J Bone Joint Surg 66-B: 148–149

    Google Scholar 

  59. Poilleux F, Lievre J-A, Codron Ph (1957) Sur un cas de résorption osseuse par ostéolyse massive régionale. Mém Acad Chir 83: 966–974

    Google Scholar 

  60. Resnick D, Niwayama G (1981) Osteolysis und Chondrolysis. In: Diagnosis of bone and joint disorders (D. Resnick and G. Niwayama eds). WB Saunders Comp, Philadelphia, London, Toronto, pp 3017–3046

    Google Scholar 

  61. Poirer H (1968) Massive osteolysis of the humerus treated by resection and prosthetic replacement. J Bone Joint Surg 50 B: 158–160

    Google Scholar 

  62. Ross JL, Schinella R, Shenkman L (1978) Massive osteolysis — an unusual cause of bone destruction. Am J Med 65: 367–372

    Google Scholar 

  63. Sacristan HD, Portal LF, Castresana FG, Pena DR (1977) Massive osteolysis of the scapula and ribs. J Bone Joint Surg 59-A: 405–406

    Google Scholar 

  64. Simpson BS (1937) An unusual case of posttraumatic decalcification of the bones of the foot. J Bone Joint Surg 19: 223–227

    Google Scholar 

  65. Schmitt O, Schmitt E, Biehl G (1982) Die essentielle Osteolyse. Klinik und Verlauf eines seltenen Krankheitsbildes. Z Orthop 120: 160–164

    Google Scholar 

  66. Schneider V, Schimke K (1967) Progressive Osteolyse bei hämangiomatösen Knochen- und Weichteilprozessen. Röntgenforum 106: 584–589

    Google Scholar 

  67. Schwarzweller F (1975) Die primären und sekundären Osteolysen. Ein Beitrag zur Systematik, Morphologie, Therapie und Klinik. ML-Verlagsgesellschaft, Uelzen

    Google Scholar 

  68. Stubbe LDJ (1962) Vanishing bones. J Bone Joint Surg 44-B: 955

    Google Scholar 

  69. Tändler P, Träger D, Adler CP (1984) Spontane ideopathische Osteolyse (“Gorham-Syndrom”) Z Orthop 122: 635–638

    Google Scholar 

  70. Thoma KH (1933) A case of progressive atrophy of the facial bones with complete atrophy of the mandible. J Bone Joint Surg XV: 494–501

    Google Scholar 

  71. Thompson JS, Schurman DJ (1974) Massive osteolysis. Case report and review of the literature. Clin Orthop 103: 206–211

    Google Scholar 

  72. Torg JS, Steel HH (1969) Sequential roetgenographic changes occuring in the massive osteolysis. J Bone Joint Surg 51-A: 1649–1655

    Google Scholar 

  73. Vanecek R, Stryhal F (1960) Massivi Osteolyza Gorham-Stout. Acta Chir Orthop Traum Che 27: 89–95

    Google Scholar 

  74. Weiss K (1960) Osteophthise-Osteolyse. Radiol Austriaca 11: 1–12

    Google Scholar 

  75. Wenz W, Reichelt A, Rau WS, Adler CP (1984) Lymphographischer Nachweis eines Wirbellymphangioms. Radiologe 24: 381–388

    Google Scholar 

  76. Woodward HR, Chan DPK, Lee J (1981) Massive osteolysis of the cervical spine. A case report of bone graft failure. Spine 6: 545–549

    Google Scholar 

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Kulenkampff, HA., Richter, G.M., Hasse, W.E. et al. Massive pelvic osteolysis in the Gorham-Stout syndrome. International Orthopaedics 14, 361–366 (1990). https://doi.org/10.1007/BF00182645

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