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Toxoplasmic pachymeningitis with visual field impairment in a single-eyed patient and a literature review.
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MedLine Citation:
PMID:  22557877     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
To describe the clinical features of a patient with toxoplasmic pachymeningitis and provide a review of the recent cases of pachymeningitis in the literature.Retrospective, observational case report. A one-eyed patient who presented to our institution with blurred vision, floaters, and visual field shrinkage. Review of clinical, laboratory, perimetric, and radiologic records of the patient and description of the efficacy of antibiotic therapy. The signs of ocular impairment, including visual acuity, visual field, and magnetic resonance imaging (MRI) changes. A one-eyed patient who complained of blurred vision and unexplained visual field shrinkage was evaluated. The diagnosis of toxoplasmic pachymeningitis was confirmed by existence of a toxoplasmic seroconversion, cerebrospinal fluid abnormalities, radiological features, absence of other diagnoses, improvement of symptoms, and resolution of pachymeningitis in MRI with oral trimethoprim-sulfamethoxazole combination.Pachymeningitis is a rare disease and toxoplasmic pachymeningitis has never been described. Among the various infectious causes, screening for toxoplasmosis must be performed because it can lead to pachymeningitis and sequelae. Variable features may lead to misdiagnosis and delay in the treatment of this sight-threatening and potentially fatal disease.
Authors:
Nicolas Bonnin; Frédéric Chiambaretta; Miguel Ulla; Frédéric Taithe; Emmanuel Chabert; Béatrice Claise; Franck Bacin
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Oman journal of ophthalmology     Volume:  5     ISSN:  0974-7842     ISO Abbreviation:  Oman J Ophthalmol     Publication Date:  2012 Jan 
Date Detail:
Created Date:  2012-05-04     Completed Date:  2012-10-02     Revised Date:  2013-05-29    
Medline Journal Info:
Nlm Unique ID:  101519430     Medline TA:  Oman J Ophthalmol     Country:  India    
Other Details:
Languages:  eng     Pagination:  46-50     Citation Subset:  -    
Affiliation:
CHU Clermont-Ferrand, Pôle Médecine Interne-Ophtalmologie-ORL, Service d'Ophtalmologie, Clermont-Ferrand, France.
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Journal Information
Journal ID (nlm-ta): Oman J Ophthalmol
Journal ID (iso-abbrev): Oman J Ophthalmol
Journal ID (publisher-id): OJO
ISSN: 0974-620X
ISSN: 0974-7842
Publisher: Medknow Publications & Media Pvt Ltd, India
Article Information
Copyright: © 2012 Bonnin N, et al
open-access:
Print publication date: Season: Jan-Apr Year: 2012
Volume: 5 Issue: 1
First Page: 46 Last Page: 50
ID: 3339675
PubMed Id: 22557877
Publisher Id: OJO-5-46
DOI: 10.4103/0974-620X.94768

Toxoplasmic pachymeningitis with visual field impairment in a single-eyed patient and a literature review
Nicolas Bonninaff1
Frédéric Chiambarettaaff1
Miguel Ulla1
Frédéric Taithe1
Emmanuel Chabert2
Béatrice Claise2
Franck Bacinaff1
CHU Clermont-Ferrand, Pôle Médecine Interne-Ophtalmologie-ORL, Service d’Ophtalmologie, Clermont-Ferrand, France
1CHU Clermont-Ferrand, Pôle Rhumatologie, Médecine Physique et de Réadaptation Neurologie et Neurochirurgie, Centre d’Evaluation et Traitement de la Douleur, Service de Neurologie, Clermont-Ferrand, France
2CHU Clermont-Ferrand, Pôle Radiologie, Service de Neuroradiologie, F-63003, Clermont-Ferrand, France
Correspondence: Correspondence: Dr. Nicolas Bonnin, Service d’Ophtalmologie, F-63003 Clermont-Ferrand, France. E-mail: nbonnin@chu clermontferrand.fr

Introduction

Cranial pachymeningitis remains a poorly understood syndrome defined by meningeal thickening and typical gadolinium enhancement on magnetic resonance imaging (MRI). It is a rare disease with protean manifestations including many ophthalmological symptoms.

It can be caused by infections, neoplasia, or autoimmune disorders.[1] Nevertheless, evaluation often fails to reveal a cause so it is supposed to be idiopathic.[2]


Case Report

In December 2008, a 70-year-old man presented to our institution with blurred vision, floaters, and visual field shrinkage. His medical history included right eye evisceration in 1997 due to retinal detachment complications, hyperuricemia, arterial hypertension, open-angle glaucoma, and prostatic resection in December 2008. His usual treatments were carteolol eye drops, captopril–hydrochlorothiazide combination, amlodipine, acetylsalicylic acid, ketoprofene, and colchicine.

Being monocular, this man had always had careful ophthalmological follow-up and regular glaucoma assessments showing a stable normal visual field.

Ophthalmological examination in January 2009 showed a decreased visual acuity of 20/32 in the left eye, while it was 20/20 in November 2008. Slit-lamp examination, ocular motility, and fundus were normal. Pupillary examination showed a decreased myosis to light. Goldman visual field showed generalized constriction to 30 degrees [Figure 1a]. Brain MRI was performed in which a diffuse meningeal thickening with gadolinium enhancement was observed, suggesting the diagnosis of pachymeningitis [Figures 24]. Fortuitously, an aneurysm located in the anterior communicant artery and measuring 9 mm was discovered.

A complete clinical, biological, radiological, inflammatory, and infectious assessment was performed in March 2009. The complete blood count showed: 9.75 Giga. L-1 leucocytes, 2% lymphocytes being of a large hyperbasophil form. Sedimentation rate was 16 mm after an hour; C-reactive protein was 5.5 mg. L-1. Infectious assessment showed significant rates of anti-toxoplasmosis IgM and IgG antibodies which increased between two blood samples spaced out of a month. The infectious and inflammatory assessment including herpes viridae serology, TPHA-VDRL, Lyme disease serology, HIV test, quantiferon test, angiotensin converting enzyme, antineutrophil cytoplasmic antibodies, and antinuclear antibodies cyclic citrullinated peptide antibodies was normal.

Before the lumbar puncture, as the patient recently received peridural anesthesia for prostatic resection, a cerebrospinal MRI searching arguments for intracranial hypotension was performed, but it did not show any cerebrospinal leakage.

The lumbar puncture showed an increased protein level; chloride and glucose levels were normal. There were 24 red blood cells mm-3 and 10 leucocytes mm-3, but the cell analysis could not be performed because of the insufficient quantity of cerebrospinal fluid. Toxoplasmosis serology and PCR, cytology for carcinomatous cells in cerebrospinal fluid were normal. The opening pressure on lumbar puncture was normal at 13 cm H2O. Serum and cerebrospinal fluid immune electrophoresis showed IgM and IgG antibody oligoclonal bands in both samples.

As seroconversion for toxoplasmosis was the only relevant result, oral trimethoprim-sulfamethoxazole combination 12 g/day was administrated during a month (April 2009), but the occurrence of a medicamentous hepatitis led to cessation of treatment 3 days earlier. The visual acuity increased to 20/25 in his left eye, but the Goldman visual field poorly improved and remained constricted. MRI brain imaging performed in May 2009 [Figure 5] showed the resolution of the pachymeningitis, but the optical coherence tomography imaging showed an atrophy of the optic disc in the left eye [Figure 6]. Aneurysm embolization was performed a few months later with no modification of the visual field.


Discussion

Cranial pachymeningitis is a very rare disease with only a few cases described. Different etiologies are known: [Table 1] essentially rheumatoid polyarthritis,[35] sarcoidosis,[16] tuberculosis,[79] intracranial hypotension,[10] and idiopathic pachymeningitis.[7]

Wegener granulomatosis[2] or systemic lupus erythematosus have also been described.[11] Several authors report that Borrelia infections,[12, 13] syphilis, or fungal infections can present with similar clinical and radiological features.[7] A case of Listeria has been described.[14] Pachymeningitis can be due to HIV: Either HIV virus or opportunist infections occurring in HIV.[15, 16] It can be related to neoplasia too: Solid cancers or hemopathies.[1719] A negative exhaustive assessment leads to diagnosis of idiopathic pachymeningitis.[7]

Isolated toxoplasmic pachymeningitis has never been reported except in case of congenital toxoplasmosis with generalized systemic locations.[20]

By definition, an abnormal dural enhancement is detected on MRI.[2] Pachymeningeal thickening can be diffuse or localized.[7] The enhancement pattern can be nodular, linear, or both.[2] Thickening of the dura mater can involve cranial bones, pericranial soft tissue, and, rarely, the cerebral parenchyma.[7] Brain MRI shows iso- or hypointense thickened dura in T1-weighted images with characteristic gadolinium enhancement [Figure 2]. T2-weighted images can give informations about the inflammation severity and the prognosis: Indeed, classical T2-weighted images show a central hypointense area which represents the fibrous reacting tissue, while a peripheral hyperintense border represents the degree of inflammation.[7, 21, 22]

In our patient, gadolinium enhancement surrounding the optic nerve gain in Fatsat T1-weighted images [Figure 3] confirms that visual loss and visual field shrinkage can be attributed to pachymeningitis. In T2-weighted images [Figure 4], the hyperintense peripheral area being important and inhomogeneous, it suggests an intense inflammation with risks of definitive visual damages.[7]

In Kupersmith et al., a series of 12 patients with idiopathic hypertrophic pachymeningitis, headache was the most frequent symptom, occurring in 92% of patients; unilateral or bilateral vision loss linked to optic neuropathy was the next most frequent symptom, occurring in 58% cases, from 20/30 to complete blindness. 66% had ophthalmoparesis, 13% had papilledema, and 13% presented ataxia.[2]

Multiple cranial nerve involvement can be explained by their compression at skull base by thickened pachymeninges.[23] Neurological deficits may appear due to cortical irritation, mass effect of nodular pachymeningitis,[7] or compressive vessel occlusion by stenosis at the skull base,[24] affecting either arteries or venous sinuses.[2, 7] Rare cases of sudden death have been described in the literature.[25, 26]

In our case, the absence of complete visual field resolution resulted from ischemia due to optic nerve compression which was objectified by evolution toward atrophy [Figure 6].

Assessment of pachymeningitis requires a clinical examination, blood and cerebrospinal fluid samples, chest X-ray or computed tomography, and brain MRI. When all results are negative or when symptoms worsen despite treatment, a meningeal biopsy can be performed.[2]

In this case, the toxoplasmic seroconversion was strongly suspected to be responsible for the pachymeningitis even if serologic tests and PCR were negative in cerebrospinal fluid: In patients with known toxoplasmic central nervous system lesions, only 28% show positive antibody in both serum and cerebrospinal fluid, this complex relationship can be explained by the blood-brain barrier,[27] and PCR is rarely useful for diagnosis in immunocompetent patients.[28]

The aneurysm could not be responsible for the condition: It classically leads to visual amputation more than constriction. Furthermore, the aneurysm embolization did not modify the visual field.

Hence treatment was initiated and another MRI was performed. Clinical improvement and favorable imaging evolution [Figure 5] favored the diagnosis of toxoplasmosis implication.


Conclusion

Even if it is a rare disease, ophthalmologists should think about pachymeningitis in case of unexplained visual loss or visual field shrinkage. A detailed assessment is needed to identify the etiology but idiopathic forms exist. Among the various infectious causes, toxoplasmosis must be screened for, because it can lead to pachymeningitis and its severe ultimate damages such as blindness, neurological deficits or even death.


Notes

Source of Support: Nil

Conflict of Interest: No.

References
1. Pareja-Esteban J,Gutierrez-Solana S,Cedazo M,Sanchez-Cordon B,Gamo-Gallego M,Teus MA. [Hypertrophic pachymeningitis and ophthalmological disturbances: Description of two case reports]Arch Soc Esp OftalmolYear: 20088349750018661447
2. Kupersmith MJ,Martin V,Heller G,Shah A,Mitnick HJ. Idiopathic hypertrophic pachymeningitisNeurologyYear: 2004626869415007115
3. Cianfoni A,Falcone C,Faustini F,Lauriola L,Imbesi S,Della Marca G,et al. Rheumatoid leptomeningitis: Magnetic resonance imaging and pathologic findings–A case reportJ NeuroimagingYear: 201020192419309435
4. Kurne A,Karabudak R,Karadag O,Yalcin-Cakmakli G,Karli-Oguz K,Yavuz K,et al. An unusual central nervous system involvement in rheumatoid arthritis: Combination of pachymeningitis and cerebral vasculitisRheumatol IntYear: 20092913495319093117
5. Ii Y,Kuzuhara S. Rheumatoid cranial pachymeningitis successfully treated with long-term corticosteroidRheumatol IntYear: 200929583518807253
6. Teirstein A. Neuromuscular sarcoidosisSemin Respir Crit Care MedYear: 2002235051216088646
7. D’Andrea G,Trillo G,Celli P,Roperto R,Crispo F,Ferrante L. Idiopathic intracranial hypertrophic pachymeningitis: Two case reports and review of the literatureNeurosurg RevYear: 20042719920415007702
8. Bernaerts A,Vanhoenacker FM,Parizel PM,Van Goethem JW,Van Altena R,Laridon A,et al. Tuberculosis of the central nervous system: Overview of neuroradiological findingsEur RadiolYear: 20031318769012942288
9. Boukobza M,Tamer I,Guichard JP,Brunereau L,Polivka M,Leibinger F,et al. [Tuberculosis of the central nervous system.MRI features and clinical course in 12 cases]J NeuroradiolYear: 1999261728110655673
10. Vankan Y,Lambrechts I,Janssen L. Pachymeningitis in intracranial liquor hypotension syndromeJBR-BTRYear: 20058824716302336
11. Coskun O,Atasoy HT,Tunc T,Yildiz H,Inan LE. Aseptic leptomeningitis in systemic lupus erythematosus.A case reportMed Princ PractYear: 20041351314657621
12. Hildenbrand P,Craven DE,Jones R,Nemeskal P. Lyme neuroborreliosis: Manifestations of a rapidly emerging zoonosisAJNR Am J NeuroradiolYear: 20093010798719346313
13. Mantienne C,Albucher JF,Catalaa I,Sevely A,Cognard C,Manelfe C. MRI in Lyme disease of the spinal cordNeuroradiologyYear: 200143485811465762
14. Lysiak Z,Litwin T,Baranska-Gieruszczak M,Poniatowska R. [Neural listeriosis presenting as leptomeningitis: A case report]Neurol Neurochir PolYear: 2002361221612715698
15. Gray F. [Lesions of the central nervous system in the early stages of human immunodeficiency virus infection]Rev Neurol (Paris)Year: 1997153629409686250
16. Morgello S,Estanislao L,Ryan E,Gerits P,Simpson D,Verma S,et al. Effects of hepatic function and hepatitis C virus on the nervous system assessment of advanced-stage HIV-infected individualsAIDSYear: 200519Suppl 3S1162216251806
17. Babiuch M,Sarzynska-Dlugosz I,Litwin T,Baranska-Gieruszczak M. [Leptomeningitis carcinomatosa with progressive involvement of the cranial nerves–Report of two cases]Neurol Neurochir PolYear: 200236581612185813
18. Okumura H,Natsugoe S,Yokomakura N,Mataki Y,Haraguchi Y,Sakita H,et al. A case of leptomeningeal carcinomatosis from esophageal basaloid carcinoma diagnosed by quantitative reverse transcription-polymerase chain reaction for carcinoembryonic antigenJ GastroenterolYear: 200540879315692794
19. Mazzeo A,Granata F,Vinci S,Musolino C,Nastasi V,Autunno M,et al. Osteosclerotic myeloma with spinal leptomeningitis and severe polyneuropathy: A case reportJ Comput Assist TomogrYear: 2006306495216845298
20. Begic-Janev A,Lastic-Maletic S,Janev A,Radojkovic Z. [Morphological study of 2 cases of congenital toxoplasmosis]Jugosl Ginekol OpstetYear: 197616243501004011
21. Hatano N,Behari S,Nagatani T,Kimura M,Ooka K,Saito K,et al. Idiopathic hypertrophic cranial pachymeningitis: Clinicoradiological spectrum and therapeutic optionsNeurosurgeryYear: 199945133642 discussion 42-4. 10598701
22. Bang OY,Kim DI,Yoon SR,Choi IS. Idiopathic hypertrophic pachymeningeal lesions: Correlation between clinical patterns and neuroimaging characteristicsEur NeurolYear: 19983949569476724
23. Goyal M,Malik A,Mishra NK,Gaikwad SB. Idiopathic hypertrophic pachymeningitis: Spectrum of the diseaseNeuroradiologyYear: 199739619239335058
24. Willing SJ,Broghamer W. Internal carotid artery occlusion due to idiopathic cranial pachymeningitisAJNR Am J NeuroradiolYear: 199213159461442436
25. Matturri L,Ottaviani G,Ramos SG,Biondo B,Rossi L. Discrete T-lymphocytic leptomeningitis of the ventral medullary surface in a case of Sudden Unexpected Infant DeathAdv Clin PathYear: 199823136
26. Chute DJ,Reiber K. Three unusual neuropathologic-related causes of sudden deathJ Forensic SciYear: 200853734818471225
27. Choi WY,Nam HW,Youn JH,Kim DJ,Kong Y,Kang SY,et al. Detection of antibodies in serum and cerebrospinal fluid to Toxoplasma gondii by indirect latex agglutination test and enzyme-linked immunosorbent assayKisaengchunghak ChapchiYear: 19923083901627507
28. Montoya JG. Laboratory diagnosis of Toxoplasma gondii infection and toxoplasmosisJ Infect DisYear: 2002185Suppl 1S738211865443

Figures

[Figure ID: F1]
Figure 1 

Kinetic perimetry of the left eye: (a) before treatment: Generalized constriction to 30 degrees; (b) after treatment: Goldman visual field poorly improved and remained constricted



[Figure ID: F2]
Figure 2 

Magnetic resonance imaging (a and b) T1-weighted images, (c and d) Gadolinium T1-weighted images: Hypo-intense thickened dura in T1-weighted images with characteristic Gadolinium enhancement



[Figure ID: F3]
Figure 3 

Magnetic resonance imaging Fatsat T1-weighted image: Gadolinium enhancement surrounding the optic nerve gain



[Figure ID: F4]
Figure 4 

Magnetic resonance imaging T2-weighted images: the hyperintense peripheral area being important and inhomogeneous, it suggests an intense inflammation with risks of definitive visual damages



[Figure ID: F5]
Figure 5 

Magnetic resonance imaging (a and b) T1-weighted images, (c and d) Gadolinium T1-weighted images: Favorable imaging evolution after treatment



[Figure ID: F6]
Figure 6 

Optical coherence tomography imaging of the optic disc in the left eye: The absence of complete visual field resolution resulted in ischemia due to optic nerve compression which resulted in resulted in optic nerve fibers loss



Tables
[TableWrap ID: T1] Table 1 

Pachymeningitis etiologies described in the literature




Article Categories:
  • Case Report

Keywords: Brain, central nervous system infection, meninges, pachymeningitis, toxoplasma, toxoplasmosis, trimethoprim–sulfamethoxazole combination.

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