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Case Report Severe Headache with Eye Involvement from Herpes Zoster Ophthalmicus, Trigeminal Tract, and Brainstem Nuclei Sasitorn Siritho, 1 Wadchara Pumpradit, 1 Wiboon Suriyajakryuththana, 1 and Krit Pongpirul 1,2,3 1 Bumrungrad International Hospital, Bangkok 10110, ailand 2 Department of Preventive and Social Medicine, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, ailand 3 Department of International Health, Johns Hopkins Bloomberg School of Public Health, Baltimore, MD 21205, USA Correspondence should be addressed to Krit Pongpirul; [email protected] Received 18 December 2014; Revised 4 February 2015; Accepted 19 March 2015 Academic Editor: Bruce J. Barron Copyright © 2015 Sasitorn Siritho et al. is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. A 43-year-old female presented with severe sharp stabbing right-sided periorbital and retroorbital area headache, dull-aching unilateral jaw pain, eyelid swelling, ptosis, and tearing of the right eye but no rash. e pain episodes lasted five minutes to one hour and occurred 10–15 times per day with unremitting milder pain between the attacks. She later developed an erythematous maculopapular rash over the right forehead and therefore was treated with antivirals. MRI performed one month aſter the onset revealed small hypersignal-T2 in the right dorsolateral mid-pons and from the right dorsolateral aspect of the pontomedullary region to the right dorsolateral aspect of the upper cervical cord, along the course of the principal sensory nucleus and spinal nucleus of the right trigeminal nerve. No definite contrast enhancement of the right brain stem/upper cervical cord was seen. Orbital imaging showed no abnormality of bilateral optic nerves/chiasm, extraocular muscles, and globes. Slight enhancement of the right V1, V2, and the cisterna right trigeminal nerve was detected. Our findings support the hypothesis of direct involvement by virus theory, reflecting rostral viral transmission along the gasserian ganglion to the trigeminal nuclei at brainstem and caudal spreading along the descending tract of CN V. A 43-year-old Canadian female presented with a two-day history of severe stabbing right-sided periorbital and retroor- bital area headache, along with dull-aching unilateral jaw pain. e pain was initially described as “pins and needles” but subsequently became stabbing in nature. e pain episodes lasted five minutes to one hour and occurred 10–15 times per day with unremitting milder pain between the attacks. She also had eyelid swelling, ptosis, and tearing of the right eye but no rash. She had history of chicken pox in childhood. An initial brain MRI was unremarkable and she was diagnosed with trigeminal cephalalgia and treated symp- tomatically. Two weeks later, she returned with persistent but somewhat improved pain. Her autonomic symptoms, photo- phobia, tearing, and eyelid swelling over the right eye with no corneal involvement were noted along with rash without vesicle over her nose. Oral valacyclovir, topical acyclovir, and antibiotics were prescribed. She returned six days later with severe pain and possible keratitis and was transferred to our hospital. Physical examination revealed maculopapular rash in the right forehead without typical vesicles over right periorbital area, keratitis, photophobia, tearing, mildly dilated pupil with no reaction to light with visual acuity of 20/40, normal fundus, minimal eyelid drooping, decreased corneal reflex, and a mild cellular infiltrate in the anterior chamber of the right side. No proptosis was observed. She complained of electrical shock-like pain involving her neck, right cheek, and right periorbital and retroorbital area and jaw. No temporal artery tenderness or nodularity, no weakness of muscles of mastication, and no definite motor weakness were seen else- where. e diagnosis of Herpes Zoster Ophthalmicus with uveitis was made. Hindawi Publishing Corporation Case Reports in Radiology Volume 2015, Article ID 402015, 4 pages http://dx.doi.org/10.1155/2015/402015

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Case ReportSevere Headache with Eye Involvement from Herpes ZosterOphthalmicus, Trigeminal Tract, and Brainstem Nuclei

Sasitorn Siritho,1 Wadchara Pumpradit,1

Wiboon Suriyajakryuththana,1 and Krit Pongpirul1,2,3

1Bumrungrad International Hospital, Bangkok 10110, Thailand2Department of Preventive and Social Medicine, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, Thailand3Department of International Health, Johns Hopkins Bloomberg School of Public Health, Baltimore, MD 21205, USA

Correspondence should be addressed to Krit Pongpirul; [email protected]

Received 18 December 2014; Revised 4 February 2015; Accepted 19 March 2015

Academic Editor: Bruce J. Barron

Copyright © 2015 Sasitorn Siritho et al.This is an open access article distributed under the Creative Commons Attribution License,which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

A 43-year-old female presented with severe sharp stabbing right-sided periorbital and retroorbital area headache, dull-achingunilateral jaw pain, eyelid swelling, ptosis, and tearing of the right eye but no rash. The pain episodes lasted five minutes to onehour and occurred 10–15 times per day with unremitting milder pain between the attacks. She later developed an erythematousmaculopapular rash over the right forehead and therefore was treated with antivirals. MRI performed one month after the onsetrevealed small hypersignal-T2 in the right dorsolateral mid-pons and from the right dorsolateral aspect of the pontomedullaryregion to the right dorsolateral aspect of the upper cervical cord, along the course of the principal sensory nucleus and spinalnucleus of the right trigeminal nerve. No definite contrast enhancement of the right brain stem/upper cervical cord was seen.Orbital imaging showed no abnormality of bilateral optic nerves/chiasm, extraocular muscles, and globes. Slight enhancement ofthe right V1, V2, and the cisterna right trigeminal nerve was detected. Our findings support the hypothesis of direct involvementby virus theory, reflecting rostral viral transmission along the gasserian ganglion to the trigeminal nuclei at brainstem and caudalspreading along the descending tract of CN V.

A 43-year-old Canadian female presented with a two-dayhistory of severe stabbing right-sided periorbital and retroor-bital area headache, along with dull-aching unilateral jaw pain.The pain was initially described as “pins and needles” butsubsequently became stabbing in nature. The pain episodeslasted five minutes to one hour and occurred 10–15 times perday with unremitting milder pain between the attacks. Shealso had eyelid swelling, ptosis, and tearing of the right eyebut no rash. She had history of chicken pox in childhood.

An initial brain MRI was unremarkable and she wasdiagnosed with trigeminal cephalalgia and treated symp-tomatically. Two weeks later, she returned with persistent butsomewhat improved pain. Her autonomic symptoms, photo-phobia, tearing, and eyelid swelling over the right eye withno corneal involvement were noted along with rash withoutvesicle over her nose. Oral valacyclovir, topical acyclovir, and

antibiotics were prescribed. She returned six days later withsevere pain and possible keratitis and was transferred to ourhospital.

Physical examination revealed maculopapular rash in theright forehead without typical vesicles over right periorbitalarea, keratitis, photophobia, tearing,mildly dilated pupil withno reaction to light with visual acuity of 20/40, normalfundus, minimal eyelid drooping, decreased corneal reflex,and a mild cellular infiltrate in the anterior chamber of theright side. No proptosis was observed. She complained ofelectrical shock-like pain involving her neck, right cheek, andright periorbital and retroorbital area and jaw. No temporalartery tenderness or nodularity, no weakness of muscles ofmastication, and no definite motor weakness were seen else-where. The diagnosis of Herpes Zoster Ophthalmicus withuveitis was made.

Hindawi Publishing CorporationCase Reports in RadiologyVolume 2015, Article ID 402015, 4 pageshttp://dx.doi.org/10.1155/2015/402015

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2 Case Reports in Radiology

(a) (a4)

(a3)

(a2)

(a1)

(b1) (b2)

(b3) (b4)

Figure 1: (a) Coronal and axial MRI sections of the brainstem to spinal cord showed small hypersignal-T2 in the right dorsolateral mid-pons(a1) and from the right dorsolateral aspect of the pontomedullary region (a2) to the right dorsolateral aspect of the upper cervical cord (a3-a4),along the course of the principle sensory nucleus and spinal nucleus of the right trigeminal nerve. Sagittal (b1), coronal (b2), and axial (b3-b4)spinal MRI showed enhancement of the nerve roots along the spinal cord.

Because of her severe headache and pain, involving mul-tiple cranial nerves, the patient underwent MRI of the brainand orbit one month after the onset, to exclude spreadinginfection, which revealed small hypersignal-T2 in the rightdorsolateral mid-pons and from the right dorsolateral aspectof the pontomedullary region to the right dorsolateral aspectof the upper cervical cord, along the course of the principalsensory nucleus and spinal nucleus of the right trigemi-nal nerve. No definite contrast enhancement of the rightbrain stem/upper cervical cord was seen. Orbital imagingshowedno abnormal signal intensity or definite enhancementof bilateral optic nerves/chiasm, extraocular muscles, andglobes. Slight enhancement of the right V1, V2, and thecisterna right trigeminal nerve was detected (Figures 1 and 2).

She was given intravenous and topical acyclovir for threeweeks, along with antibiotics and other medications forpostherpetic neuralgia. Her headache and eye symptomsresponded dramatically to the treatment; the skin of the rightcheek became less severely hypersensitive. BrainMRI showeda slight decrease in the enhancement of the lesions previouslyseen.

Dermatomal pain with eye involvement without typicalpapulovesicular skin lesion was suggestive of OphthalmicZoster Sine Herpete [1], which can be accompanied by signsand symptoms of CN III paresis [2]. In this case, patient hadbeen partially treated with antivirals prior to the rash thatappeared so this may conceal her symptoms and signs as wellas MRI enhancement.

Herpetic involvement of cranial nerves has been mostlikely underestimated. Even more difficult to find is imagingevidence that demonstrates the CN V connection pathway tothe nuclear lesion at brainstem (rhombencephalitis) [3–5].

Similar to our findings, M. Aribandi and L. Aribandidemonstrated abnormal hypersignal in the left lower ponsand medulla posteriorly and laterally near the fourth ven-tricle, extending into the upper cervical cord, correspondingto the location of the spinal trigeminal nucleus and tract[3]. Haanpaa et al. described focal nonenhancing brainstemhyperintense lesions in T2-weighted images in patients withtrigeminal zoster without extraocular muscle involvement[4]. Our MRI findings demonstrated an even longer spread-ing of the abnormal hyperintensity signal on T2W compared

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Case Reports in Radiology 3

(a1) (a2)

(a4)(a3)

(b1) (b2)

Figure 2: AxialMRI sections of the brainstem at the pontine level demonstrated fusiform enlargement of the fifth cranial nerve at the cisternalevel on the T1-weighted images (a1). Postcontrast, fat-suppressed T1 images showed slight enhancement that is disclosed on cisterna levelof the fifth cranial nerve (a2) and enlargement of the maxillary division of the fifth cranial nerve with slight enhancement while it passedforamen rotundum (a3). Also coronal MRI section at the level of the fifth cranial nerve showed slight enhancement (a4). Axial MRI sectionsshowed brain stem at the level of facial and vestibulocochlear nerves in precontrast image (b1) and slight enhancement in postcontrast study(b2).

to a case report by Tsai et al. which described a long enhanc-ing lesion from upper cervical cord extending to the lowerthoracic spinal cord [6].

Interestingly we found ptosis and pupillary dilatation,which might be from the involvement of the third nerve.Similar to our study, Hu et al. reported a patient with brain-stem and multiple cranial nerves involvement including pto-sis but no pupillary involvement in immunocompetent host[7]. Our MRI findings may support the hypothesis of directinvolvement by virus that VZV migrated transaxonally ortransynaptically into the brain stem parenchyma and mayspread to multiple cranial nerve, reflecting rostral viral trans-mission along the gasserian ganglion to the trigeminal nucleiat brainstem and caudal spreading along the descending tractof CN V.

Consent

Written consent to publishing was obtained.

Conflict of Interests

The authors declare no conflict of interests.

Authors’ Contribution

Sasitorn Siritho and Wadchara Pumpradit managed thepatient. Sasitorn Siritho prepared the case summary.WiboonSuriyajakryuththana, Wadchara Pumpradit, and Krit Pong-pirul helped to prepare and edited the report. All authors readand approved the final paper.

References

[1] D. Gilden, R. J. Cohrs, R. Mahalingam, and M. A. Nagel, “Neu-rological disease produced by varicella zoster virus reactivationwithout rash,” Current Topics in Microbiology and Immunology,vol. 342, no. 1, pp. 243–253, 2010.

[2] T. Parkinson, “Rarer manifestations of herpes zoster; a reporton three cases,” British Medical Journal, vol. 1, no. 4539, pp. 8–10, 1948.

[3] M. Aribandi and L. Aribandi, “MRI of trigeminal zoster,” Neu-rology, vol. 65, no. 11, p. 1812, 2005.

[4] M.Haanpaa, P. Dastidar, A.Weinberg et al., “CSF andMRI find-ings in patients with acute herpes zoster,”Neurology, vol. 51, no.5, pp. 1405–1411, 1998.

[5] R. D. Tien, G. J. Felsberg, and A. K. Osumi, “Herpesvirus infec-tions of the CNS: MR findings,”The American Journal of Roent-genology, vol. 161, no. 1, pp. 167–176, 1993.

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4 Case Reports in Radiology

[6] J. Tsai, R. J. Bert, and D. Gilden, “Zoster paresis: AsymptomaticMRI lesions far beyond the site of rash and focal weakness,”Journal of the Neurological Sciences, vol. 330, no. 1-2, pp. 119–120,2013.

[7] S. Hu,M.Walker, T. Czartoski et al., “Acyclovir responsive brainstem disease after the Ramsay Hunt syndrome,” Journal of theNeurological Sciences, vol. 217, no. 1, pp. 111–113, 2004.

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