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Nasal septal perforation and antiphospholipid syndrome (Hughes syndrome)

Published online by Cambridge University Press:  07 June 2007

S S Banerjee ,
K Lammin  and
J de Carpentier
S S Banerjee*
Affiliation:
Department of Otorhinolaryngology, Royal Preston Hospital, Preston, UK.
K Lammin
Affiliation:
Department of Otorhinolaryngology, Royal Preston Hospital, Preston, UK.
J de Carpentier
Affiliation:
Department of Otorhinolaryngology, Royal Preston Hospital, Preston, UK.
*
Address for correspondence: Miss Sudipta Banerjee, 11 Langham Court, Mersey Road, West Didsbury, Manchester M20 2PX, UK.Fax: 0044 1422 341801 E-mail: sudipta@banerjee.fsnet.co.uk
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Abstract

We present the case of a 19-year-old woman who developed a nasal septal perforation and atrophic rhinitis following septal surgery. During the subsequent five years, she also experienced intermittent episodes of swelling and ischaemia of the left foot, as well as livedo reticularis. Ultimately, haematological investigations revealed the presence of lupus anticoagulant and elevated antiphospholipid immunoglobulin M levels. A diagnosis of antiphospholipid syndrome (Hughes syndrome) was made.

A search of the literature revealed that nasal septal perforation has been described in up to 0.8 per cent of patients with antiphospholipid syndrome. However, it is undescribed in the otorhinolaryngological literature. The impact of this syndrome on otorhinolaryngology practice is examined.

Keywords

Antiphospholipid SyndromeComplications, SurgeryOtorhinolaryngology
Type
Clinical Records
Information
The Journal of Laryngology & Otology , Volume 121 , Issue 12 , December 2007 , pp. 1197 - 1200
Copyright
Copyright © JLO (1984) Limited 2007

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References

1 Cervera, R, Piette, JC, Font, J, Khamashta, MA, Shoenfeld, Y, Camps, MT et al. , for the Euro-phospholipid Project Group. Antiphospholipid syndrome: clinical and immunological manifestations and patterns of disease expression in a cohort of 1000 patients. Arthritis Rheum 2002;46:1019–27CrossRefGoogle Scholar
2 O'Gradaigh, D, Scott, DGI. Hughes' ears: an unusual presentation of antiphospholipid syndrome. Ann Rheum Dis 1999;58:65–6CrossRefGoogle ScholarPubMed
3 Mouadeb, DA, Ruckenstein, MJ. Antiphospholipid inner ear syndrome. Laryngoscope 2005;115:879–83CrossRefGoogle ScholarPubMed
4 Moine, A, Mohcini, M, Beal, C, Guillevin, L, Frachet, B. Deafness and auto-immunity: what is the role of antiphospholipid syndrome? Apropos of 35 cases [French]. Ann Otolaryngol Chir Cervicofac 1994;111;363–70Google ScholarPubMed
5 Green, L, Miller, EB. Sudden sensori-neural hearing loss as a first manifestation of systemic lupus erythematosus: association with cardiolipin antibodies. Clin Rheum 2001;20:220–2CrossRefGoogle Scholar
6 Compadretti, GC, Brandolini, C, Tasca, I. Sudden sensorineural hearing loss in lupus erythematosus associated with antiphospholipid syndrome: case report and review. Ann Otol Rhinol Laryngol 2005;114:214–18CrossRefGoogle ScholarPubMed
7 Vyse, T, Luxon, LM, Walport, MJ. Audiovestibular manifestations of the antiphospholipid syndrome. J Laryngol Otol 1994;108:57–9CrossRefGoogle ScholarPubMed
8 Piette, JC, Papo, T, Godeau, B, Wechsler, B, Frances, C, Du-Boutin, LTH et al. Nasal septal perforation: a new manifestation associated with anti-phospholipid antibodies (abstract 130). Lupus 1994;3:345Google Scholar
9 Egan, RM, Munn, RK. Catastrophic antiphospholipid antibody syndrome presenting with multiple thromboses and sites of avascular necrosis. J Rheumatol 1994;21:2376–9Google ScholarPubMed
10 Bladt, V, Steengaard-Pedersen, K, Poulsen, LH, Petersen, OB, Laursen, B, d'Amore, F. Late puerperal thrombohemorrhagic complications in a patient with antiphospholipid syndrome. Eur J Haematol 2004;73:437–40CrossRefGoogle Scholar
11 Asherson, RA, Cervera, R, Piette, JC, Sheonfeld, Y, Espinosa, G, Petri, MA et al. Catastrophic antiphospholipid syndrome: clues to the pathogenesis from a series of 80 patients. Medicine 2001;80:355–77CrossRefGoogle Scholar
12 Kale, US, Wight, RG. Primary presentation of spontaneous jugular vein thrombosis to the otolaryngologist – in three different pathologies. J Laryngol Otol 1998;112:888–90CrossRefGoogle Scholar
13 Holland, AJ, Childs, PA, Beilin, J, Baker, RI. Spontaneous thrombosis of the internal jugular vein as the initial presentation of the primary antiphospholipid syndrome. Aust N Z J Surg 1996;66:854–5CrossRefGoogle ScholarPubMed
14 Munday, DN, Jones, WR. Pregnancy complicated by the antiphospholipid syndrome. Aus N Z J Obs Gynae 1993;33:255–8CrossRefGoogle ScholarPubMed
15 Alcazar-Ramirez, JD, Fernandez-Nebro, A, Abarca, M, Salgado, F, De, Haro-Liger M, Rodriguez-Andreu, J et al. Central vein thrombosis in the antiphospholipid syndrome. Lupus 1997;6:549–51CrossRefGoogle ScholarPubMed
16 Papaioannou, G, Speletas, M, Kaloutsi, V, Pavlitou-Tsiontsi, A. Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease) associated with antiphospholipid syndrome: case report and literature review. Ann Haematology 2002;81:732–5CrossRefGoogle ScholarPubMed
17 Emir, M, Ozisik, K, Cagli, K, Bakuy, V, Ozisik, P, Sener, E. Dural sinus thrombosis after cardiopulmonary bypass. Perfusion 2004;19:133–5CrossRefGoogle ScholarPubMed
18 Champion, BL, Choy, F, Schrieber, L, Roche, J, Rowe, DB. Isolated fascicular oculomotor nerve palsy as the initial presentation of the antiphospholipid syndrome. J Clin Neurosci 2002;9:691–4CrossRefGoogle ScholarPubMed
19 Erkan, D, Asherson, RA, Espinosa, G, Cervera, R, Font, J, Piette, JC et al. , for the Catastrophic Antiphospholipid Syndrome Registry Project Group. Long term outcome of catastrophic antiphospholipid syndrome survivors. Ann Rheum Dis 2003;62:530–3CrossRefGoogle ScholarPubMed
20 Asherson, RA. The catastrophic antiphospholipid syndrome. J Rheumatol 1992;19:508–12Google ScholarPubMed
21 Wilson, WA, Gharavi, AE, Koike, T, Lockshin, MD, Branch, DW, Piette, JC et al. International consensus statement on preliminary classification criteria for definite antiphospholipid syndrome. Arth Rheum 1999;42:1309–113.0.CO;2-F>CrossRefGoogle ScholarPubMed