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Gorham–Stout disease of the temporal bone involving the temporomandibular joint

Published online by Cambridge University Press:  24 January 2018

D Mulvihill ,
R S Kumar ,
J Muzaffar  and
R Irving
D Mulvihill*
Affiliation:
Department of Otology, Queen Elizabeth University Hospital Birmingham, UK
R S Kumar
Affiliation:
Department of Otology, Queen Elizabeth University Hospital Birmingham, UK
J Muzaffar
Affiliation:
Department of Otology, Queen Elizabeth University Hospital Birmingham, UK
R Irving
Affiliation:
Department of Otology, Queen Elizabeth University Hospital Birmingham, UK
*
Address for correspondence: Mr Daniel Mulvihill, 31 West Heath Road, Birmingham B31 3TN, UK E-mail: danmulvihill@doctors.org.uk
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Abstract

Background:

Gorham–Stout disease of the skull is a very rare entity. It presents with gradual bone resorption, and proliferation of lymphoid and vascular channels within the bony matrix. This is often a diagnosis of exclusion confirmed with serial imaging and based on radiological evidence.

Case report:

A case of Gorham–Stout disease of the temporal bone involving the temporomandibular joint, and presenting with sensorineural hearing loss and recurrent temporomandibular joint dislocation, is reported. The findings are presented and the literature on this condition is reviewed.

Conclusion:

ENT and maxillofacial surgeons should be aware of this extremely rare cause of temporomandibular joint dislocation and ear symptoms. Imaging comprising computed tomography and magnetic resonance imaging is crucial to achieving a diagnosis, which may only become evident after repeated imaging follow up. Symptomatic treatment is advised, with the option of anti-osteoclastic medication and radiotherapy indicated for advanced cases. Surgery is only recommended for complications including involvement of neurovascular structures.

Keywords

Gorham-Stout DiseaseVanishing Bone DiseaseTemporal BoneTemporomandibular Joint
Type
Clinical Record
Information
The Journal of Laryngology & Otology , Volume 132 , Issue 3 , March 2018 , pp. 279 - 281
Copyright
Copyright © JLO (1984) Limited 2018 

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References

1 Gorham, LW, Stout, AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis. J Bone Joint Surg Am 1955;37:985–1004.Google Scholar
2 Ruggieri, P, Montalti, M, Angelina, A, Alberghini, M, Mercuri, M. Gorham-Stout disease: the experience of the Rizzoli Institute and review of the literature. Skeletal Radiol 2011;40:1391–7.Google Scholar
3 Morimoto, N, Ogiwara, H, Miyazaki, O, Kitamuara, M, Nishina, S, Nakazawa, A et al. Gorham-Stout syndrome affecting the temporal bone with cerebrospinal fluid leakage. Int J Pediatr Otorhinolaryngol 2013;77:1596–600.Google Scholar
4 Cushing, SL, Ishak, G, Perkins, JA, Rubinstein, JT. Gorham-Stout syndrome of the petrous apex causing chronic cerebrospinal fluid leak. Otol Neurotol 2010;31:789–92.CrossRefGoogle ScholarPubMed
5 Plontke, S, Koitschev, A, Ernemann, U, Pressler, H, Zimmermann, R, Plasswilm, L. Massive Gorham-Stout osteolysis of the temporal bone and the craniocervical transition [in German]. HNO 2002;50:354–7.CrossRefGoogle ScholarPubMed
6 Nozawa, A, Ozeki, M, Kuze, B, Asano, T, Matsuoka, K, Fukao, T. Gorham-Stout disease of the skull base with hearing loss: dramatic recovery and antiangiogenic therapy. Pediatr Blood Cancer 2016;63:931–4.CrossRefGoogle ScholarPubMed
7 Mowry, S, Canalis, R. Gorham-Stout disease of the temporal bone. Laryngoscope 2010;120:598–600.Google Scholar
8 Oujilal, A, Lazrak, A, Benhalima, H, Boulaich, M, Amarti, A, Saidi, A et al. Massive lytic osteodystrophy or Gorham-Stout disease of the craniomaxillofacial area [In French]. Rev Laryngol Otol Rhinol (Bord) 2000;121:255–60.Google ScholarPubMed
9 Choma, ND, Biscotti, CV, Bauer, TW, Mehta, AC, Licata, AA. Gorham's syndrome: a case report and review of the literature. Am J Med 1987;83:1151–6.Google Scholar
10 Hagberg, H, Lamberg, K, Aström, G. Alpha-2b interferon and oral clodronate for Gorham's disease. Lancet 1997;350:1822–3.Google Scholar
11 Dunbar, SF, Rosenberg, A, Mankin, H, Rosenthal, D, Suit, HD. Gorham's massive osteolysis: the role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys 1993;26:491–7.CrossRefGoogle Scholar