A 48-year-old woman presented with isolated sixth cranial nerve palsy. She subsequently developed systemic lupus erythematosus (SLE) based on clinical and laboratory parameters. Three years later, she presented again with sixth cranial nerve palsy affecting the contralateral eye. Within 2 weeks of steroid initiation, complete recovery occurred. The unusual rare presentation of SLE in the current patient, as well as the pathogenesis and treatment of cranial neuropathy in SLE are discussed. Lupus (2010) 19, 201—205.
Hochberg MCUpdating the American College of Rheumatology Revised criteria for the classification of systemic lupus erythematosus. Arth Rheum1997; 40: 1725.
2.
The American College of Rheumatology nomenclature and case definitions for neuropsychiatric lupus syndromes. Arth Rheum1999; 42: 599-608.
3.
Bruns A., Meyer O.Neuropsychiatric manifestations of systemic lupus erythematosus. Joint Bone Spine2006; 73: 639-645.
4.
Teoh SC, Yap EY, Au Eong KGNeuro-ophthalmological manifestations of systemic lupus erythematosus in Asian patients. Clin Exp Ophthalmol2001; 29: 213-216.
5.
Sivaraj RR, Durrani OM, Denniston AK, Murray PI, Gordon C.Ocular manifestations of systemic lupus erythematosus. Rheumatology (Oxford)2007; 46: 1757-1762.
6.
Caldarelli DD , Rejowski JE, Corey JPSensorineural hearing loss in lupus erythematosus . Am J Otol1986; 7: 210-213.
7.
Hamblin TJ, Muftin GJ, Bracewell A.A severe deafness in systemic lupus erythematosus: Its immediate relief by plasma exchange. Br Med J (Clin Res Ed)1982; 284: 1374.
8.
Bowman CA, Linthicum FH Jr, Nelson RA, Mikami K., Quismorio F.Sensorineural hearing loss associated with systemic lupus erythematosus. Otolaryngol Head Neck Surg1986; 94: 197-204.
9.
Kobayashi S. , Fujishiro N., Sugiyama K.Systemic lupus erythematosus with sensorineural hearing loss and improvement after plasmapheresis using the double filtration method. Intern Med1992; 31: 778-781.
10.
Vyse T., Luxon LMAudiovestibular manifestations of the antiphospholipid syndrome. J Laryngol Otol1994; 108: 57-59.
11.
Kataoka H., Takeda T.Sensorineural hearing loss of suspected autoimmune etiology: a report of three cases. Auris Nasus Larynx1995; 22: 53-58.
12.
Naarendorp M., Spiera H.Sudden sensorineural hearing loss in patients with systemic lupus erythematosus or lupus-like syndromes and antiphospholipid antibodies. J Rheumatol1998; 25: 589-592.
13.
Green L., Miller EBSudden sensorineural hearing loss as a first manifestation of systemic lupus erythematosus: Association with anticardiolipin antibodies. Clin Rheumatol2001; 20: 220-222.
14.
Peeva E., Barland P.Sensorineural hearing loss in conjunction with aortic insufficiency in systemic lupus erythematosus. Scand J Rheumatol2001 ; 30: 45-47.
15.
Kastanioudakis I., Ziavra N., Voulgari PV, Exarchakos G., Skevas A., Drosos AAEar involvement in systemic lupus erythematosus patients: a comparative study. J Laryngol Otol2002; 116: 103-107.
16.
Compadretti GC, Brandolini C.Sudden sensorineural hearing loss in lupus associated with antiphospholipid syndrome: case report and review. Ann Otol Rhinol Laryngol2005; 114: 214-218.
17.
Roverano S. , Cassano G.Asymptomatic sensorineural hearing loss in patients with systemic lupus erythematosus. J Clin Rheumatol2006; 12: 217-220.
18.
Gomides AP, do Rosario EJ, Borges HM, Gomides HH, de Padua PM, Sampaio-Barros PDSensorineural dysacusis in patients with systemic lupus erythematosus. Lupus2007; 16: 987-990.
19.
Karatas E., Onat AMAudiovestibular disturbance in patients with systemic lupus erythematosus . Otolaryngol Head Neck Surg2007; 136: 82-86.
20.
Khalidi NA, Rebello R., Robertson DDSensorineural hearing loss in systemic lupus erythematosus: case report and literature review. J Laryngol Otol2008; 122: 1371-1376.
21.
Asherson RA , Mackworth-Young CGPulmonary hypertension in systemic lupus erythematosus . Br Med J1983; 287: 1024-1025.
22.
Asherson RA , Hughes GRVocal cord paralysis in systemic lupus erythematosus complicated by pulmonary hypertension. J Rheumatol1985; 12: 1029-1030.
23.
Asherson RA , Hackett D.Pulmonary hypertension in systemic lupus erythematosus: a report of three cases. J Rheumatol1986; 13: 416-420.
24.
Aszkenasy OM , Clarke TJSystemic lupus erythematosus, pulmonary hypertension, and left recurrent laryngeal nerve palsy. Ann Rheum Dis1987; 46: 246-247.
25.
Sharma A., Singh RRVocal cord palsy in a patient of systemic lupus erythematosus. J Assoc Physicians India1988; 36: 674-675.
Gordon T., Dann ECSystemic lupus erythematosus and right recurrent laryngeal nerve palsy. Br J Rheumatol1990; 29: 308-309.
28.
Kraus A., Guerra-Bautista G.Laryngeal involvement as a presenting symptom of systemic lupus erythematosus . Ann Rheum Dis1990; 4: 421.
29.
Espana A., Gutierrez JMRecurrent laryngeal palsy in systemic lupus erythematosus. Neurology1990; 40: 1143-1144.
30.
Asherson RA , Higenbottam TWPulmonary hypertension in a lupus clinic: experience with twenty-four patients. J Rheumatol1990; 17: 1292-1298.
31.
Vaile JH, Davis P.Isolated unilateral vagus nerve palsy in systemic lupus erythematosus. J Rheumatol1998; 25: 2287-2288.
32.
Toubi E.Vocal cord paralysis in a patient with active systemic lupus erythematosus . Isr Med Assoc J2000; 2: 243-244.
33.
Imauchi Y.Left vocal cord paralysis in cases of systemic lupus erythematosus. ORL2001; 63: 53-55.
34.
Yu K-H. , Yang C-H.Swallowing disturbance due to isolated vagus nerve involvement in systemic lupus erythematosus. Lupus2007; 16: 746-749.
35.
Lee JH, Sung IYRecurrent laryngeal neuropathy in a systemic lupus erythematosus (SLE) patient . Am J Phys Rehabil2008; 87: 68-70.
36.
Johnson RT, Richardson EPThe neurological manifestations of systemic lupus erythematosus: a clinical-pathologic study of 24 cases and review of the literature. Medicine1968; 47: 337-369.
37.
Lanham JG, Elkon KBPtosis in systemic lupus erythematosus. Postgrad Med J1982; 58: 688-689.
38.
Rosenstein ED, Sobelman J.Isolated, pupil-sparing third nerve palsy as initial manifestation of systemic lupus erythematosus. J Clin Neuro Ophtalmol1989; 4: 285-288.
39.
Keane JREye movement abnormalities in systemic lupus erythematosus. Arch Neurol1995; 52: 1145-1149.
40.
Lee JSIsolated oculomotor nerve palsy with pupillary abnormality in systemic lupus erythematosus. J Pediatr Ophthalmol Strabismus2000; 37: 241-243.
41.
Genevay S., Hayem G.Oculomotor palsy in six patients with systemic lupus erythematosus. A possible role of antiphospholipid syndrome. Lupus2002 ; 11: 313-316.
42.
Evans OB, Lexow SSPainful ophtalmoplegia in systemic lupus erythematosus. Ann Neurol1978; 4: 584-585.
McDonald E. , Marino C.Painful ophtalmoplegia in a patient with SLE . Hosp Pract (Off Ed)1992; 27: 41-44.
45.
Friedman AS , Folkert V.Recurrence of systemic lupus erythematosus in a hemodialysis patient presenting as unilateral abducens nerve palsy. Clin Nephrol1995; 44: 338-339.
46.
Kwok SK, Seo SH, Ju JH, et al. Cryptococcal meningitis presenting with isolated sixth cranial nerve palsy in a patient with systemic lupus erythematosus. J Korean Med Sci2008; 23: 153-155.
47.
Peters GB III , Bakri SJ , Krohel GBCause and prognosis of non-traumatic sixth nerve palsies in young adults. Ophthalmology2002; 109: 1925-1928.
48.
Arbuckle MR , McClain MTDevolpment of autoantibodies before the clinical onset of systemic lupus erythematosus. N Engl J Med2003; 349: 1526-1533.
49.
Sanna G., Bertolaccini ML, Cuadrado MJNeuropsychiatric manifestations in systemic lupus erythematosus: prevalence and association with antiphospholipid antibodies. J Rheumatol2003; 30: 985-992.
50.
Balestrieri G., Doria A. III.European Conference on Systemic Lupus Erythematosus. Conference summary. Clin Exp Rheumol1996; 14(Suppl 16): S3-S16.
51.
Galindo-Rodriguez G., Avina-Zubieta JACyclophosphamide pulse therapy in optic neuritis due to systemic lupus erythematosus: an open trial. Am J Med1999; 106: 65.
52.
Miyakis S., Lockshin MD, Atsumi T., et al. International consensus statement on an update of the classification criteria for definite antiphospholipid syndrome (APS). J Thromb Haemost2006; 4: 295-306.
