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Gaze palsy, hypogeusia and a probable association with miscarriage of pregnancy--the expanding clinical spectrum of non-opticospinal neuromyelitis optica spectrum disorders: a case report.
BMC Res Notes 2015; 8:36BR

Abstract

BACKGROUND

Neuromyelitis optica is characterised by optic neuritis, longitudinally-extensive transverse myelitis and presence of anti-aquaporin-4 antibodies in the serum. However, non-opticospinal central nervous system manifestations have been increasingly recognised. Awareness of the widening clinical spectrum of neuromyelitis optica (unified within the nosology of 'neuromyelitis optica spectrum disorders') is key to earlier diagnosis and appropriate therapy. We report 2 patients to illustrate the varied clinical manifestations of neuromyelitis optica spectrum disorders while postulating an effect of anti-aquaporin-4 antibodies on the miscarriage of pregnancy. This is the first report of horizontal gaze palsy as a presenting symptom of neuromyelitis optica spectrum disorders.

CASE PRESENTATION

Patient 1: A 17-year-old Sri Lankan female presented with hypersomnolence, lateral gaze palsy and loss of taste of 1 week duration. Two years previously she had presented with intractable hiccups and vomiting followed by a brainstem syndrome. Magnetic resonance imaging showed a lesion in the left cerebellum extending into the pons while lesions in bilateral hypothalami and medulla noted 2 years ago had resolved. Autoimmune, vasculitis and infection screens were negative. Anti-aquaporin-4 antibodies were detected in serum. All her symptoms resolved with immunosuppressive therapy. Patient 2: A 47-Year-old Sri Lankan female presented with persistent vomiting lasting over 3 weeks. Three years previously, at 25-weeks of her 4(th) pregnancy, she had presented with quadriparesis and was found to have a longitudinally extensive transverse myelitis from C2 to T2 vertebral levels, which gradually improved following intravenous steroid therapy. Magnetic resonance imaging showed a hyper-intense lesion in the area postrema and longitudinally extensive atrophy of the cord corresponding to her previous myelitis. Autoimmune, vasculitis and infection screens were negative. Anti-aquaporin-4 antibodies were detected in serum. Her vomiting subsided with immunosuppressive therapy. Her second pregnancy had resulted in a first-trimester miscarriage.

CONCLUSION

The clinical spectrum of neuromyelitis optica spectrum disorders has expanded beyond optic neuritis and myelitis to include non-opticospinal syndromes involving the diencephalon, brainstem and cerebrum. Our report highlights the varied central nervous system manifestations of neuromyelitis optica spectrum disorders and miscarriage of pregnancy possibly related to anti-aquaporin-4 antibodies.

Authors+Show Affiliations

Department of Clinical Medicine, Faculty of Medicine, University of Colombo, 25, Kynsey Road, Colombo, 08, Sri Lanka. thashichang@gmail.com. University Medical Unit, National Hospital of Sri Lanka, Colombo, Sri Lanka. thashichang@gmail.com.University Medical Unit, National Hospital of Sri Lanka, Colombo, Sri Lanka. milishan2003@gmail.com.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

25888897

Citation

Chang, Thashi, and Milinda Withana. "Gaze Palsy, Hypogeusia and a Probable Association With Miscarriage of Pregnancy--the Expanding Clinical Spectrum of Non-opticospinal Neuromyelitis Optica Spectrum Disorders: a Case Report." BMC Research Notes, vol. 8, 2015, p. 36.
Chang T, Withana M. Gaze palsy, hypogeusia and a probable association with miscarriage of pregnancy--the expanding clinical spectrum of non-opticospinal neuromyelitis optica spectrum disorders: a case report. BMC Res Notes. 2015;8:36.
Chang, T., & Withana, M. (2015). Gaze palsy, hypogeusia and a probable association with miscarriage of pregnancy--the expanding clinical spectrum of non-opticospinal neuromyelitis optica spectrum disorders: a case report. BMC Research Notes, 8, p. 36. doi:10.1186/s13104-015-0991-5.
Chang T, Withana M. Gaze Palsy, Hypogeusia and a Probable Association With Miscarriage of Pregnancy--the Expanding Clinical Spectrum of Non-opticospinal Neuromyelitis Optica Spectrum Disorders: a Case Report. BMC Res Notes. 2015 Feb 10;8:36. PubMed PMID: 25888897.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Gaze palsy, hypogeusia and a probable association with miscarriage of pregnancy--the expanding clinical spectrum of non-opticospinal neuromyelitis optica spectrum disorders: a case report. AU - Chang,Thashi, AU - Withana,Milinda, Y1 - 2015/02/10/ PY - 2014/10/28/received PY - 2015/01/23/accepted PY - 2015/4/19/entrez PY - 2015/4/19/pubmed PY - 2015/12/15/medline SP - 36 EP - 36 JF - BMC research notes JO - BMC Res Notes VL - 8 N2 - BACKGROUND: Neuromyelitis optica is characterised by optic neuritis, longitudinally-extensive transverse myelitis and presence of anti-aquaporin-4 antibodies in the serum. However, non-opticospinal central nervous system manifestations have been increasingly recognised. Awareness of the widening clinical spectrum of neuromyelitis optica (unified within the nosology of 'neuromyelitis optica spectrum disorders') is key to earlier diagnosis and appropriate therapy. We report 2 patients to illustrate the varied clinical manifestations of neuromyelitis optica spectrum disorders while postulating an effect of anti-aquaporin-4 antibodies on the miscarriage of pregnancy. This is the first report of horizontal gaze palsy as a presenting symptom of neuromyelitis optica spectrum disorders. CASE PRESENTATION: Patient 1: A 17-year-old Sri Lankan female presented with hypersomnolence, lateral gaze palsy and loss of taste of 1 week duration. Two years previously she had presented with intractable hiccups and vomiting followed by a brainstem syndrome. Magnetic resonance imaging showed a lesion in the left cerebellum extending into the pons while lesions in bilateral hypothalami and medulla noted 2 years ago had resolved. Autoimmune, vasculitis and infection screens were negative. Anti-aquaporin-4 antibodies were detected in serum. All her symptoms resolved with immunosuppressive therapy. Patient 2: A 47-Year-old Sri Lankan female presented with persistent vomiting lasting over 3 weeks. Three years previously, at 25-weeks of her 4(th) pregnancy, she had presented with quadriparesis and was found to have a longitudinally extensive transverse myelitis from C2 to T2 vertebral levels, which gradually improved following intravenous steroid therapy. Magnetic resonance imaging showed a hyper-intense lesion in the area postrema and longitudinally extensive atrophy of the cord corresponding to her previous myelitis. Autoimmune, vasculitis and infection screens were negative. Anti-aquaporin-4 antibodies were detected in serum. Her vomiting subsided with immunosuppressive therapy. Her second pregnancy had resulted in a first-trimester miscarriage. CONCLUSION: The clinical spectrum of neuromyelitis optica spectrum disorders has expanded beyond optic neuritis and myelitis to include non-opticospinal syndromes involving the diencephalon, brainstem and cerebrum. Our report highlights the varied central nervous system manifestations of neuromyelitis optica spectrum disorders and miscarriage of pregnancy possibly related to anti-aquaporin-4 antibodies. SN - 1756-0500 UR - https://www.unboundmedicine.com/medline/citation/25888897/Gaze_palsy_hypogeusia_and_a_probable_association_with_miscarriage_of_pregnancy__the_expanding_clinical_spectrum_of_non_opticospinal_neuromyelitis_optica_spectrum_disorders:_a_case_report_ L2 - https://bmcresnotes.biomedcentral.com/articles/10.1186/s13104-015-0991-5 DB - PRIME DP - Unbound Medicine ER -