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|LETTER TO EDITOR
|Year : 2015 | Volume
| Issue : 4 | Page : 615-616
Asymmetric lumbosacral radiculoplexopathy: Rare presentation of sarcoidosis
Mandeep Singh Ghuman1, Savinay Kapur1, Sameer Vyas1, Sanat Bhatkar2, Manoj K Goyal2, Niranjan Khandelwal1
1 Department of Radiodiagnosis, Post Graduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Neurology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
|Date of Web Publication||4-Aug-2015|
Department of Radiodiagnosis, Post Graduate Institute of Medical Education and Research, Chandigarh
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Ghuman MS, Kapur S, Vyas S, Bhatkar S, Goyal MK, Khandelwal N. Asymmetric lumbosacral radiculoplexopathy: Rare presentation of sarcoidosis. Neurol India 2015;63:615-6
|How to cite this URL:|
Ghuman MS, Kapur S, Vyas S, Bhatkar S, Goyal MK, Khandelwal N. Asymmetric lumbosacral radiculoplexopathy: Rare presentation of sarcoidosis. Neurol India [serial online] 2015 [cited 2019 Aug 21];63:615-6. Available from: http://www.neurologyindia.com/text.asp?2015/63/4/615/162089
Sarcoidosis is a multisystemic inflammatory disorder of unknown etiology. Though the lungs are the most consistently affected, sarcoidosis is known to affect the lymph nodes, skin, liver, brain, periperhal nerves, and almost every organ system of the body. Nervous system involvement is seen in approximately 5-10% of patients with sarcoidosis.  Neurosarcoidosis can have a highly varied clinical presentation and is difficult to diagnose, especially in patients lacking pulmonary or systemic manifestations.  We describe clinicoradiological features of a rare case of neurosarcoidosis where the initial manifestation was in the form of peripheral nervous system involvement presenting as asymmetric lumbosacral radiculoplexopathy.
A 56-year-old lady, nondiabetic, presented with a history of low backache, and weakness of left lower limb since 2½ months in the form of difficulty in getting up and in gripping slippers. She also complained of sensory disturbances in the form of tingling and paresthesias over the left thigh since 2 months. On general examination, she was found to be having bilateral parotid swelling. Neurological examination revealed decreased power of left hip movements (4/5) and decreased sensation over anterior aspect of the left thigh in L2, L3 distribution associated with pins and needles sensation. Her cranial nerve examination was normal. The tone and gait were normal. Laboratory examination revealed serum calcium in high normal range; however, angiotensin-converting enzyme levels were mildly raised. Biopsy from the parotid gland was performed which showed presence of noncaseating granulomas consistent with sarcoidosis. The magnetic resonance imaging of the lumbosacral spine and lumbosacral plexus was performed. Thickening and abnormal enhancement of bilateral lumbar (L2-L5) and sacral (S1, S2) nerve roots, dorsal root ganglia, and exiting nerves were found with a more pronounced involvement on the left side [Figure 1]. Contrast-enhanced computed tomography of the chest, abdomen, and pelvis was done, which showed nodular fissural thickening, subpleural and peribronchovascular nodules in bilateral lungs [Figure 2]. Few enlarged mediastinal and hilar lymph nodes were also found.
|Figure 1: Coronal T1-weighted postcontrast fat-saturated lumbar spine magnetic resonance imaging shows diffuse thickening and abnormal enhancement of bilateral lumbar nerve roots and exiting nerves (arrows), with asymmetric involvement on the left side|
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|Figure 2: Contrast-enhanced computed tomography chest lung window image shows subpleural nodules (arrows) in bilateral lungs along with nodular thickening of bilateral major fissures (arrowheads)|
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Winkler was the first to describe the pathological affliction of nerves in sarcoidosis in 1905.  The incidence of peripheral neuropathy within the subset of patients with neurosarcoidosis has been reported to be 8.5%. The spectrum of noncranial peripheral neuropathy has typically been described as a chronic sensorimotor neuropathy. Atypical patterns of involvement include mononeuritis multiplex, acute Guillain-Barré syndrome, pure sensory neuropathy, pure motor neuropathy, as well as well as lumbar plexopathy. These atypical neuropathies are known to become symptomatic before any other manifestations of the multi-system disease become evident and have been reported to precede the systemic symptoms by up to 18 years.  Though different authors have reported different incidences of the various noncranial peripheral neural manifestations, interestingly two of the largest case series found that subacute or chronic polyneuropathy was consistently associated with the absence of cranial nerve involvement. ,
Although rare, sarcoidosis can present with an asymmetric lumbosacral plexopathy associated with chronic sensory motor neuropathy.  Electrophysiological studies are usually consistent with a generalized axonal sensorimotor neuropathy. , Pulmonary lesions should be specifically sought by appropriate imaging investigations in every patient with clinically manifesting isolated radiculoplexus neuropathies. Chest lesions, especially mediastinal lymph nodes, are seen in most of the sarcoid patients diagnosed with peripheral neuropathy. These can, however, remain clinically silent.  The diagnosis rests on demonstration of noncaseating inflammatory granulomas with multisystem involvement; however, sarcoid granulomas are seldom found in nerve biopsy specimens.  Treatment for neurosarcoidosis is similar to sarcoidosis in any other part of the body with corticosteroids forming the first line of management. In case of contraindication to steroid therapy or in case of relapse of symptoms, cytotoxic drugs such as methotrexate, azathioprine, and cyclosporine have been used with fairly good clinical outcomes, although no controlled studies are available to document their benefit. 
The lack of consistent diagnostic criteria, as well as any specific and sensitive tests, has meant that the diagnosis of sarcoidosis remains enigmatic. Nonetheless, we would like to highlight that sarcoidosis should be considered as an important differential diagnosis in inflammatory lumbosacral plexopathies in addition to the other common entities causing diabetic and nondiabetic lumbosacral radiculoplexus neuropathy. 
| » References|| |
Miller R, Sheron N, Semple S. Sarcoidosis presenting with an acute Guillain-Barré syndrome. Postgrad Med J 1989;65:765-7.
Said G. Sarcoidosis of the peripheral nervous system. In: Said G, Krarup C, editors. Handbook of Clinical Neurology. Vol. 115, Ch. 27. Edinburgh Elsevier; 2013. p. 485-95.
Zuniga G, Ropper AH, Frank J. Sarcoid peripheral neuropathy. Neurology 1991;41:1558-61.
Said G, Lacroix C, Planté-Bordeneuve V, Le Page L, Pico F, Presles O, et al.
Nerve granulomas and vasculitis in sarcoid peripheral neuropathy: A clinicopathological study of 11 patients. Brain 2002;125:264-75.
Laughlin RS, Dyck PJ. Electrodiagnostic testing in lumbosacral plexopathies. Phys Med Rehabil Clin N Am 2013;24:93-105.
[Figure 1], [Figure 2]