Skip to main content
Top
Published in: Japanese Journal of Radiology 10/2017

01-10-2017 | Original Article

MR imaging findings of vertebral involvement in Gorham–Stout disease, generalized lymphatic anomaly, and kaposiform lymphangiomatosis

Authors: Hiroki Kato, Michio Ozeki, Toshiyuki Fukao, Masayuki Matsuo

Published in: Japanese Journal of Radiology | Issue 10/2017

Login to get access

Abstract

Purpose

To assess the MR imaging findings of vertebral involvement in Gorham–Stout disease (GSD), generalized lymphatic anomaly (GLA), and kaposiform lymphangiomatosis (KLA).

Methods

Nine patients (three patients with GSD, three with GLA, and three with KLA) who underwent whole-spine MR examinations were included. MR findings of fatty marrow replacement and prolonged T1 and T2 lesions of the vertebrae were retrospectively assessed. Prolonged T1 and T2 lesions were defined as well-defined and round-, oval-, or geographic-shaped areas.

Results

Six patients [one (33%) patient with GSD, two (67%) with GLA, and three (100%) with KLA] showed both fatty marrow replacement and prolonged T1 and T2 lesions. Fatty marrow replacement was seen in the cervical spine of two (33%) patients, thoracic spine of six (100%), lumbar spine of six (100%), and sacral spine of two (33%). Prolonged T1 and T2 lesions were seen in the cervical spine of three (50%) patients, thoracic spine of three (50%), lumbar spine of six (100%), and sacral spine of three (50%).

Conclusion

Both fatty marrow replacement and prolonged T1 and T2 lesions of the vertebrae could be observed in GSD, GLA, and KLA. The most commonly affected site was the lumbar spine, followed by the thoracic spine.
Literature
1.
go back to reference Wassef M, Blei F, Adams D, Alomari A, Baselga E, Berenstein A, et al. Vascular anomalies classification: recommendations from the International Society for the Study of Vascular Anomalies. Pediatrics. 2015;136:e203–14.CrossRefPubMed Wassef M, Blei F, Adams D, Alomari A, Baselga E, Berenstein A, et al. Vascular anomalies classification: recommendations from the International Society for the Study of Vascular Anomalies. Pediatrics. 2015;136:e203–14.CrossRefPubMed
2.
go back to reference Gorham LW, Wright AW, Shultz HH, Maxon FC Jr. Disappearing bones: a rare form of massive osteolysis; report of two cases, one with autopsy findings. Am J Med. 1954;17:674–82.CrossRefPubMed Gorham LW, Wright AW, Shultz HH, Maxon FC Jr. Disappearing bones: a rare form of massive osteolysis; report of two cases, one with autopsy findings. Am J Med. 1954;17:674–82.CrossRefPubMed
3.
go back to reference Gorham LW, Stout AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis. J Bone Jt Surg Am. 1955;37-A:985–1004.CrossRef Gorham LW, Stout AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis. J Bone Jt Surg Am. 1955;37-A:985–1004.CrossRef
4.
go back to reference Johnson PM, Mc CJ. Observations on massive osteolysis; a review of the literature and report of a case. Radiology. 1958;71:28–42.CrossRefPubMed Johnson PM, Mc CJ. Observations on massive osteolysis; a review of the literature and report of a case. Radiology. 1958;71:28–42.CrossRefPubMed
5.
go back to reference Pastakia B, Horvath K, Lack EE. Seventeen-year follow-up and autopsy findings in a case of massive osteolysis. Skelet Radiol. 1987;16:291–7.CrossRef Pastakia B, Horvath K, Lack EE. Seventeen-year follow-up and autopsy findings in a case of massive osteolysis. Skelet Radiol. 1987;16:291–7.CrossRef
6.
go back to reference Radhakrishnan K, Rockson SG. Gorham’s disease: an osseous disease of lymphangiogenesis? Ann N Y Acad Sci. 2008;1131:203–5.CrossRefPubMed Radhakrishnan K, Rockson SG. Gorham’s disease: an osseous disease of lymphangiogenesis? Ann N Y Acad Sci. 2008;1131:203–5.CrossRefPubMed
7.
go back to reference Dellinger MT, Garg N, Olsen BR. Viewpoints on vessels and vanishing bones in Gorham–Stout disease. Bone. 2014;63:47–52.CrossRefPubMed Dellinger MT, Garg N, Olsen BR. Viewpoints on vessels and vanishing bones in Gorham–Stout disease. Bone. 2014;63:47–52.CrossRefPubMed
8.
go back to reference Rossler J, Saueressig U, Kayser G, von Winterfeld M, Klement GL. Personalized therapy for generalized lymphatic anomaly/Gorham–Stout disease with a combination of sunitinib and taxol. J Pediatr Hematol Oncol. 2015;37:e481–5.CrossRefPubMedPubMedCentral Rossler J, Saueressig U, Kayser G, von Winterfeld M, Klement GL. Personalized therapy for generalized lymphatic anomaly/Gorham–Stout disease with a combination of sunitinib and taxol. J Pediatr Hematol Oncol. 2015;37:e481–5.CrossRefPubMedPubMedCentral
9.
go back to reference Alvarez OA, Kjellin I, Zuppan CW. Thoracic lymphangiomatosis in a child. J Pediatr Hematol Oncol. 2004;26:136–41.CrossRefPubMed Alvarez OA, Kjellin I, Zuppan CW. Thoracic lymphangiomatosis in a child. J Pediatr Hematol Oncol. 2004;26:136–41.CrossRefPubMed
10.
go back to reference Croteau SE, Kozakewich HP, Perez-Atayde AR, Fishman SJ, Alomari AI, Chaudry G, et al. Kaposiform lymphangiomatosis: a distinct aggressive lymphatic anomaly. J Pediatr. 2014;164:383–8.CrossRefPubMed Croteau SE, Kozakewich HP, Perez-Atayde AR, Fishman SJ, Alomari AI, Chaudry G, et al. Kaposiform lymphangiomatosis: a distinct aggressive lymphatic anomaly. J Pediatr. 2014;164:383–8.CrossRefPubMed
11.
go back to reference Goyal P, Alomari AI, Kozakewich HP, Trenor CC 3rd, Perez-Atayde AR, Fishman SJ, et al. Imaging features of kaposiform lymphangiomatosis. Pediatr Radiol. 2016;46:1282–90.CrossRefPubMed Goyal P, Alomari AI, Kozakewich HP, Trenor CC 3rd, Perez-Atayde AR, Fishman SJ, et al. Imaging features of kaposiform lymphangiomatosis. Pediatr Radiol. 2016;46:1282–90.CrossRefPubMed
12.
go back to reference Ozeki M, Fujino A, Matsuoka K, Nosaka S, Kuroda T, Fukao T. Clinical features and prognosis of generalized lymphatic anomaly, kaposiform lymphangiomatosis, and Gorham–Stout disease. Pediatr Blood Cancer. 2016;63:832–8.CrossRefPubMed Ozeki M, Fujino A, Matsuoka K, Nosaka S, Kuroda T, Fukao T. Clinical features and prognosis of generalized lymphatic anomaly, kaposiform lymphangiomatosis, and Gorham–Stout disease. Pediatr Blood Cancer. 2016;63:832–8.CrossRefPubMed
13.
go back to reference Lala S, Mulliken JB, Alomari AI, Fishman SJ, Kozakewich HP, Chaudry G. Gorham–Stout disease and generalized lymphatic anomaly–clinical, radiologic, and histologic differentiation. Skelet Radiol. 2013;42:917–24.CrossRef Lala S, Mulliken JB, Alomari AI, Fishman SJ, Kozakewich HP, Chaudry G. Gorham–Stout disease and generalized lymphatic anomaly–clinical, radiologic, and histologic differentiation. Skelet Radiol. 2013;42:917–24.CrossRef
14.
go back to reference Bode-Lesniewska B, von Hochstetter A, Exner GU, Hodler J. Gorham–Stout disease of the shoulder girdle and cervico-thoracic spine: fatal course in a 65-year-old woman. Skelet Radiol. 2002;31:724–9.CrossRef Bode-Lesniewska B, von Hochstetter A, Exner GU, Hodler J. Gorham–Stout disease of the shoulder girdle and cervico-thoracic spine: fatal course in a 65-year-old woman. Skelet Radiol. 2002;31:724–9.CrossRef
15.
go back to reference Ceroni D, De Coulon G, Regusci M, Kaelin A. Gorham–Stout disease of costo-vertebral localization: radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings. Acta Radiol. 2004;45:464–8.CrossRefPubMed Ceroni D, De Coulon G, Regusci M, Kaelin A. Gorham–Stout disease of costo-vertebral localization: radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings. Acta Radiol. 2004;45:464–8.CrossRefPubMed
16.
go back to reference Dominguez R, Washowich TL. Gorham’s disease or vanishing bone disease: plain film, CT, and MRI findings of two cases. Pediatr Radiol. 1994;24:316–8.CrossRefPubMed Dominguez R, Washowich TL. Gorham’s disease or vanishing bone disease: plain film, CT, and MRI findings of two cases. Pediatr Radiol. 1994;24:316–8.CrossRefPubMed
17.
go back to reference Livesley PJ, Saifuddin A, Webb PJ, Mitchell N, Ramani P. Gorham’s disease of the spine. Skelet Radiol. 1996;25:403–5.CrossRef Livesley PJ, Saifuddin A, Webb PJ, Mitchell N, Ramani P. Gorham’s disease of the spine. Skelet Radiol. 1996;25:403–5.CrossRef
18.
go back to reference Safi F, Gupta A, Adams D, Anandan V, McCormack FX, Assaly R. Kaposiform lymphangiomatosis, a newly characterized vascular anomaly presenting with hemoptysis in an adult woman. Ann Am Thorac Soc. 2014;11:92–5.CrossRefPubMed Safi F, Gupta A, Adams D, Anandan V, McCormack FX, Assaly R. Kaposiform lymphangiomatosis, a newly characterized vascular anomaly presenting with hemoptysis in an adult woman. Ann Am Thorac Soc. 2014;11:92–5.CrossRefPubMed
19.
go back to reference Luks VL, Kamitaki N, Vivero MP, Uller W, Rab R, Bovee JV, et al. Lymphatic and other vascular malformative/overgrowth disorders are caused by somatic mutations in PIK3CA. J Pediatr. 2015;166(1048–54):e1–5. Luks VL, Kamitaki N, Vivero MP, Uller W, Rab R, Bovee JV, et al. Lymphatic and other vascular malformative/overgrowth disorders are caused by somatic mutations in PIK3CA. J Pediatr. 2015;166(1048–54):e1–5.
20.
21.
go back to reference Adams DM, Trenor CC 3rd, Hammill AM, Vinks AA, Patel MN, Chaudry G, et al. Efficacy and safety of sirolimus in the treatment of complicated vascular anomalies. Pediatrics. 2016;137:e20153257.CrossRefPubMedPubMedCentral Adams DM, Trenor CC 3rd, Hammill AM, Vinks AA, Patel MN, Chaudry G, et al. Efficacy and safety of sirolimus in the treatment of complicated vascular anomalies. Pediatrics. 2016;137:e20153257.CrossRefPubMedPubMedCentral
23.
go back to reference Shah LM, Hanrahan CJ. MRI of spinal bone marrow: Part I. Techniques and normal age-related appearances. AJR Am J Roentgenol. 2011;197:1298–308.CrossRefPubMed Shah LM, Hanrahan CJ. MRI of spinal bone marrow: Part I. Techniques and normal age-related appearances. AJR Am J Roentgenol. 2011;197:1298–308.CrossRefPubMed
24.
go back to reference Hanrahan CJ, Shah LM. MRI of spinal bone marrow: Part 2. T1-weighted imaging-based differential diagnosis. AJR Am J Roentgenol. 2011;197:1309–21.CrossRefPubMed Hanrahan CJ, Shah LM. MRI of spinal bone marrow: Part 2. T1-weighted imaging-based differential diagnosis. AJR Am J Roentgenol. 2011;197:1309–21.CrossRefPubMed
Metadata
Title
MR imaging findings of vertebral involvement in Gorham–Stout disease, generalized lymphatic anomaly, and kaposiform lymphangiomatosis
Authors
Hiroki Kato
Michio Ozeki
Toshiyuki Fukao
Masayuki Matsuo
Publication date
01-10-2017
Publisher
Springer Japan
Published in
Japanese Journal of Radiology / Issue 10/2017
Print ISSN: 1867-1071
Electronic ISSN: 1867-108X
DOI
https://doi.org/10.1007/s11604-017-0674-3

Other articles of this Issue 10/2017

Japanese Journal of Radiology 10/2017 Go to the issue