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Unusual Case of Orbital Neurocysticercosis Involving Lateral Rectus, Cerebellum and Brain Stem

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Kalpana Sharma, Kulbhushan Prakash Chaudhary, Ravinder Kumar Gupta
Dept. of Ophthalmology, Indira Gandhi Medical College, Shimla

Corresponding Author:

Kalpana Sharma
Department of Ophthalmology,
Indira Gandhi Medical College, Shimla
E-mail: doctorkalpana.84@gmail.com

Published Online: 01-MAR-2015
DOI:http://dx.doi.org/10.7869/djo.110

Abstract

Keywords :

Dear Editor
 
Cysticercosis is a zoonotic parasitic infection caused by the larval stage (Cysticercus cellulosae) of the pig tapeworm Taenia solium. This is the most common helminth to produce CNS infection in human beings (neurocysticercosis).[1] Ocular cysticercosis may be extraocular (in the sub-conjunctival or orbital tissues) or intraocular (in the vitreous, sub-retinal space, or anterior chamber).[2] Ocular manifestations of cysticercosis vary from asymptomatic to painful blind eye and may be associated with neurological symptoms such as headache and fits. The most common site of localisation reported in western studies is posterior segment and in Indian literature the ocular adnedxa.[3] We report a case of orbital neurocysticercosis to document the characteristic involvement of orbit and CNS. A 17 years old patient reported to us in eye OPD with headache for 3 months, diminution of vision and double vision since 15 days. On thorough clinical evaluation in central nervous system examination the patient had positive cerebellar signs of ataxia, past pointing, intention tremors and dysdiadochokinesia. The patient also had motor system involvement leading to bilateral lower limbs weakness consistent with brain stem lesions. On ocular examination the patient had 6/6 vision in right eye and 6/24 in left eye. Ocular motility testing showed left abduction deficit with horizontal diplopia on levoversion (Figure a,b,c). The anterior and posterior segments including the fundus examination of both the eyes were normal. Blood investigations in the form of complete blood count, blood sugar (fasting and postprandial) were normal. X ray chest (PA view) was within normal limits. On CT scan multiple ring enhancing lesions involving bilateral cerebral hemisphere, cerebellum and brain stem with ring enhancing lesion in lateral rectus of left eye (Figure 1-4).








  
Discussion

 Humans are the only definitive hosts for the parasite. When people ingest the undercooked infected meat, enzymes lyse the outer shell of parasite cyst leaving scolex behind. The scolex gets attached to the intestinal wall, proliferates and become adult tapeworm over 2 months which produce eggs (proglottids) which mature and become gravid and are shed in stools.When pigs ingest the eggs the cycle begins.[4] The eggs lysed in stomach release oncosphere in small intestine and penetrate the bowel mucosa thereby entering blood stream to various tissue brain, subcutaneous tissue, eyes, muscles.[5] Normally pigs are intermediate host in life cycle of this cysticercus. However rarely man act as intermediate host and manifest as cysticercosis.[6] Clinical presentations of cysticercosis in human may involve the CNS, eye, or other viscera. Ocular involvements include cysts in eyelids, extraocular muscles, orbit, conjunctiva, anterior chamber, uvea, retina-vitreous, and optic nerve.[7] Although there are various reports of isolated ocular and intraocular cysticercosis in the literature, there are few reports of cysticercosis involving the eye and brain.[8] In India most common site of localization is orbit, whereas posterior segment involvement is more common in western people. Intraocularly, cysticerci occur in vitreous body and sub retinal but some may be found in the anterior chamber and subconjunctival. The most damaging location is intravitreal and subretinal location which leads to blindness in 3 to 5 years unless the parasite is removed.[9] One of the primary symptoms of extraocular cysticercosis is double vision (diplopia) from misalignment of the visual axes, and the pattern of image separation is the key to diagnosing which particular cranial nerve (and extraocular muscle) is involved.

 Surgical excision of an extraocular muscle cyst had been described. In contrast to medical treatment, surgical excision is technically difficult due to the attachment of the cysts to underlying orbital structures, the amorphous consistency of degenerating cysticerci and the risk to neurovascular structures in the posterior orbit.[10] There is also a likelihood of postoperative restrictive myopathy arising from the ?brotic response in surgical excisions of large cysts or in cysts requiring extensive dissection from the underlying muscle. The potential risk of damage to adjacent tissue and adhesion from surgical exploration should not be taken lightly, particularly when effective medical therapy is available. Albendazol and prednisone is the best choice taken by most of the authors and recommended duration of treatment varies from a few days to up to 6 weeks. The treatment of ocular cysticercosis is conflicting. In our case patient was treated with systemic steroids and cysticidal therapy and the response was dramatic.

The common differential diagnoses of orbital cysticercosis are endocrine orbitopathy (Grave’s disease) and orbital pseudotumor. Grave’s disease is found in majority of patients and is usually bilateral with involvement of more than one muscle along with lid retraction. On CT and MRI imaging extraocular muscles are bulky predominantly involving the belly of the muscles with sparing of tendinous attachment along with inflammatory changes in the intraorbital fat. The orbital pseudotumor is usually unilateral and presents with pain, enlargement of extraocular muscles including its tendinous insertion. On MRI, pseudotumor appears as hypointense lesion both on T1- and T2-weighted images (probably due to fibrotic changes) and shows marked gadolinium enhancement. A prompt response to steroid treatment, in conjunction with the radiological findings, supports the diagnosis of a pseudotumor. Orbital hydatid cyst is rare entity and they are often larger in size and mostly require surgical excision. The other differential diagnosis can be lipomas, neurofibromas , epidermal cysts and tuberculomas.

 It is important to carry out a detailed medical evaluation in every case, in order to exclude the presence of the parasite at other sites. It must take into account other conditions that mimic the orbital cysticercosis in their differential diagnosis. Although as a rare disease, it is a challenge for health professionals to manage these patients.

Financial & competing interest disclosure

 The authors do not have any competing interests in any product/procedure mentioned in this study. The authors do not have any financial interests in any product / procedure mentioned

References
  1. H Foyaca-Sibat, M Salazar-Campos, L Ibanez-Valdes. Cysticercosis Of The Extraocular Muscles. Our Experience And Review Of The Medical Literature. The Internet Journal of Neurology. 2012 Volume 14 Number 1.
  2. Sawhney IM, Singh G, Lekhra OP, Mathuriya SN, Parihar PS, Prabhakar S. Uncommon presentations of neurocysticercosis. J Neurol Sci. 1998; 154:94-100.
  3. Malik SRK, Gupta AK,Choudhry S. Ocular cysticercosis. Am J Ophthalmol 1968; 66:1168.
  4. Yamshita P, Kelsey J, Henderson S O. Subcutaneous cysticercosis. J Emerg Med 1998; 16:583-86.
  5. Despommier DD. Tapeworm infection: The long and short of it. N Engl J Med 1992; 327:727-28.
  6. Horton J. Biology of tapeworm disease (letter). Lancet 1996 ;348:486.
  7. Sharma T, Sinha S, Shah N, Gopal L, Shanmugam MP, Bhende P, et al. Intraocular cysticercosis: Clinical characteristics and visual outcome after vitreoretinal surgery. Ophthalmology 2003; 110:996-1004.
  8. Das D, Deka S, Islam S, Deuri N, Deka P, Deka AC, et al. Neuro and intraocular cysticercosis: A clinicopathological case report. Eye and Brain 2010; 2:39-42.
  9. Mohan Reddy C C, Gupta V P, Sarada P, Prabhakar V, Reddy D L, Anjaneyulu C. Ocular cysticercosis. Indian J Ophthalmol 1980; 28:69-72.
  10. Rath S, Honavar SG, Naik M, Anand R, Agarwal B, Krishnaiah S et al. Orbital cysticercosis: clinical manifestations, diagnosis, management, and outcome. Ophthalmology 2010; 117:600-5.

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Kalpana Sharma, Kulbhushan Prakash Chaudhary, Ravinder Kumar GuptaUnusual Case of Orbital Neurocysticercosis Involving Lateral Rectus, Cerebellum and Brain Stem.DJO 2015;25:209-211

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Kalpana Sharma, Kulbhushan Prakash Chaudhary, Ravinder Kumar GuptaUnusual Case of Orbital Neurocysticercosis Involving Lateral Rectus, Cerebellum and Brain Stem.DJO [serial online] 2015[cited 2022 Aug 7];25:209-211. Available from: https://www.djo.org.in/articles/25/3/unusual-case-of-orbital.html