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Spinal Cord Syphilitic Gumma Presenting with Brown-Séquard

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Spinal Cord Syphilitic Gumma Presenting with Brown-Séquard Available online at www.annclinlabsci.org Annals of Clinical & Laboratory Science, vol. 49, no. 2, 2019 265 Spinal Cord Syphilitic Gumma Presenting with Brown- Séquard Syndrome: A Case Report and Literature Review Yin-Hui Huang1,* , Qing-Xiao Shi1,*, Mian-Mian Xu1,*, Chuan-Zhen Chen1,*, Mei-Li Yang2,*, Jun-jing Li3,*, Ya-Fang Chen2,*, Zhi-Qiang Lin1,*, and You-Yu Lin4 1Department of Neurology, Jinjiang Municipal Hospital, Jinjiang, 2Department of Neurology, Second Affiliated Hospital of Fujian Medical University, Quanzhou, 3Department of Neurology, Quanzhou First Hospital, Quanzhou, and 4Department of Neurosurgery, Jinjiang Municipal Hospital, Jinjiang, Fujian, China Abstract. Background. Spinal neurosyphilis manifesting as a solitary syphilitic gumma is exceedingly rare. There are non-specific imaging findings and challenges in the diagnosis of spinal syphilitic gumma, which could be easily misdiagnosed as tumor lesions and require surgical resection or biopsy. Clinical Presenta- tion. We report the case of a 45-year-old female patient who was diagnosed with Spinal syphilitic gumma. Our case is the first reported case of spinal cord syphilitic gumma with intradural-extramedullary and intra- medullary involvement. Conclusion. Spinal syphilitic gumma exhibits diverse clinical manifestations, lacks specific imaging features, accompanied by the patient's history deliberately concealed. Since clinicians do not have sufficient knowledge about such rare cases, misdiagnosis and missed diagnosis will be likely. When there is clinical suspicion for spinal syphilitic gumma, clinicians should pay close attention to relevant medical history, carry out a comprehensive physical examination and specific serological tests and cerebro- spinal fluid (CSF) analysis. In summary, in cases with stable neurologic conditions, a trial administration of intravenous penicillin with follow-up imaging may be the optimal treatment option, and in cases with rapid progression or acute exacerbation, a surgical resection together with systemic antibiotic treatment for syphilis after surgery may be the best treatment strategy. Key words: neurosyphilis, Spinal cord gumma, spinal cord. Introduction Case Report Neurosyphilis is a clinical syndrome caused by A 45-year-old woman was admitted to the Neurology Treponema pallidum (syphilis) infection of the department with complaints of right lower limb weak- nervous system (which involve either the brain) the ness, left lower limb numbness for 10 days in October th spinal cord or meningeal blood vessels. Spinal neu- 26 , 2015. She had no history of hypertension, diabetes mellitus or coronary artery disease, also of tuberculosis rosyphilis manifesting as a solitary syphilitic gum- or AIDS or sick contacts, and denied feculent sexual life ma is exceedingly rare; Only about 8 cases have history. Neurologic examination revealed muscle been described worldwidely [1-8]. There are non- strength of grade 3/5 in right lower extremities and loss specific imaging findings and challenges in the di- of superficial sensation of grade 3/10 below the umbili- agnosis of spinal syphilitic gumma, which could be cus on the left side, Grade 3 ankle and knee hyperreflexia easily misdiagnosed as tumor lesions and require on the right side, positive Babinski sign and Chaddock surgical resection or biopsy [9]. Therefore, we re- sign. Syphilis serology showed a rapid plasma reagin port a case of spinal syphilitic gumma and review (RPR) test positive with a titer of 1:2 and the T. pallidum the previous relevant published literature to in- particle agglutination test positive with a titer of 1:43.89. crease awareness of the disease. Tests for HIV and hepatitis B and other routine blood tests were negative. A CSF examination was not per- formed due to the patient's lumbar puncture refusal. *These authors contributed equally to this work. Address correspondence to Mrs. Ya-Fang Chen and Mr. Mei-Li Yang; Spinal MRI showed an irregular nodule at the T8 level Department of Neurology, Second Affiliated Hospital of Fujian combined intradural-extramedullary and intramedullary Medical University, Quanzhou, Fujian, China, No.34, Zhongshan North Road, Quanzhou City, Fujian Province, China; phone: +86 involvement. The nodule had a slightly hyperintense sig- 0595 85659153; fax:+86 0595 85659153; e mail:251045413@ nal on T1-weighted imaging and a heterogeneously hy- qq.com. perintense signal on T2-weighted imaging, which was 0091-7370/19/0200-265. © 2019 by the Association of Clinical Scientists, Inc. 266 Annals of Clinical & Laboratory Science, vol. 49, no. 2, 2019 A preoperative diagnosis of space occupying lesion in thoracic spinal canal was made, and a surgical lesionec- tomy was performed via a posterior midline approach. The lesion was found to be a solid mass at the level of T8-T9 combined intradural-extramedullary (about 3×1.5×0.5cm in size) and intramedullary (about 1.8×1.3×0.6cm in size) involvement. The lesion didn’t have a clear borderline and coating, with medium hard- ness, had medium hardness and not so rich blood sup- ply. It lay at the lateral side of spinal canal. The lesion was located in the spinal canal laterally, bound at the nerve root with severe adhesions. Lesion debulking was performed. A diagnosis of the lesion could not be made on the intraoperative frozen section, which showed spindle cells. 5 days later, pathologic examination of paraffin section revealed granulomatous inflammation with small areas of caseous necrosis, and multinucleat- ed giant cells infiltration, surrounded by large numbers of lymphocytes and small numbers of neutrophils. It Figure 1. The nodule had a slightly hyperintense signal on also showed swelling and hyperplasia of some vascular T1-weighted imaging and a heterogeneously hyperintense endothelial cells with massive infiltration of lympho- signal on T2-weighted imaging, was significantly and ho- cytes and plasma cells around the blood vessels. A sus- mogeneously enhanced on Gadolinium-enhanced MRI pected diagnosis of syphilis infection was made based (Gd-MRI). on patient history. Immunohistochemical analysis re- vealed immunopositivity with glial fibrillary acidic pro- tein, myelin basic protein, neurofilament protein, CD3, CD45RO, and CD68 but was negative for peri- odicacid Schiff and CD56. Acid-fast staining was nega- tive; further Warthin Starry staining confirmed spiro- chete infection. A definitive diagnosis of spinal intramedullary gumma was made (Figure 2). And the treatment for syphilis was administered with intrave- nous penicillin G (19.6 million U/day, and 4.9 million U QID) for 14 days, then intramuscular benzathine penicillin G (2.4 million U QW) for 21 days. After the treatment, the patient’s right lower limb weakness and left lower limb numbness were improved. Figure 2. Pathological examination of paraffin section re- After discharge, the muscle strength of her legs im- vealed granulomatous inflammation with small areas of ca- proved to grade 4/5. Three months later, she has com- seous necrosis, and multinucleated giant cells infiltration, pletely recovered from her illness. surrounding by large numbers of lymphocytes and small numbers of neutrophil. It also showed swelling and hyper- plasia of some vascular endothelial cells with a massive infil- Literature Review tration of lymphocytes and plasma cells around the blood vessels. PubMed and Embase searches were performed be- tween the years of 1960 and 2017 based on the significantly and homogeneously enhanced on following keywords: 1. (spine OR spinal) AND Gadolinium-enhanced MRI (Gd-MRI). The nodule size (approx) was 1.8×0.7×0.9cm. Adjacent meninges had (gumma OR syphilitic granuloma); 2. (spine OR cord-like enhanced, and contiguous spinal cord became spinal) AND (syphilis OR syphilitic). Seven case thicken with a striped slightly hyperintense signal on T2- reports of spinal syphilitic gumma were identified, weighted imaging. A radiological diagnosis of spinal cord including 3 English case reports [1-3] and 5 non- vascular malformations or space-occupying lesions with English case reports in Spanish, French, rich blood supply at the T8 level was made (Figure 1). Portuguese, and Chinese [4-8] (Tables 1-3). Table 1. Clinical and Imaging Features in Cases with Intraspinal Gumma. Case Lan Age Sex Onset Symptoms Duration LocationImaging Features guage (years) Yang et al,20161 English 65 F Neck, shoulder, and back pain, 2 months Spinal intramedullary lesion T1WI, isointensity tetraplegia, sensory disturbance at C5 level T2WI,hyperintensity with isointense center, perilesional edema Contrast T1WI, peripheral contrast enhancement Dhasmana et al, English 40 M Headache, nausea, photophobia, 3 months 1 lesion in right cingulate gyrus; T1WI, intracranial, N/A; spinal, 20132 lower backache, paraplegia 3 lesions in spinal cord (details N/A) isointensity T2WI, intracranial, signal characteristics N/A, perilesional edema; spinal, isointensity Contrast T1WI, intracranial, ring enhancement; spinal, N/A Zhou et al,20143 English 49 F Paraplegia, sensory disturbance 10 years Spinal intradural extramedullary T1WI, isointensity lesion at T2-3 level T2WI, hyperintensity with hypointense middle portion Contrast T1WI, bright and homogeneous contrast enhancement Molina-Olier Spanish 47 F Shoulder pain, paraplegia, 2 days Spinal extradural lesion at CT, isodensity T1WI, isointensity et al,20124 sphincteric disturbance T6-8 level T2WI, N/A Contrast T1WI, peripheral contrast enhancement El Quessar French 25 F Brown-Séquard syndrome 2 years Spinal intramedullary
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