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J Korean Neurosurg Soc. 2010 July; 48(1): 85–87.
Published online 2010 July 31. doi:  10.3340/jkns.2010.48.1.85
PMCID: PMC2916157

A Solitary Skull Lesion of Syphilitic Osteomyelitis

Suk Hyung Kang, M.D.,1 Seung Won Park, M.D.,corresponding author1 Ki Young Kwon, M.D.,2 and Won Jin Hong, M.D.3

Abstract

We experienced a rare case of solitary syphilitic osteomyelitis of the skull without any other clinical signs or symptoms of syphilis. A 20-year-old man was referred due to intermittent headache and mild tenderness at the right parietal area of the skull with a palpable coin-sized lesion of softened cortical bone. On radiological studies, the lesion was a radiolucent well enhanced mass (17 mm in diameter). The erythrocyte sedimentation rate (52 mm/h) and C-reactive protein (2.24 mg/dL) were elevated on admission. Serum venereal disease research laboratory (VDRL) and Treponema pallidum haemagglutination assay (TPHA) tests were positive. There were no clinical signs or symptoms of syphilis. After treatment with benzathine penicillin, we removed the lesion and performed cranioplasty. The pathologic finding of the skull lesion was fibrous proliferation with lymphoplasmocytic infiltration forming an osteolytic lesion. In addition, a spirochete was identified using the Warthin-starry stain. The polymerase chain reaction study showed a positive band for Treponema pallidum. Solitary osteomyelitis of the skull can be the initial presenting pathological lesion of syphilis.

Keywords: Infectious osteomyelitis, Syphilis, Skull, Treponema pallidum

INTRODUCTION

The history of syphilitic diseases7), includes syphilitic osteomyelitis of the skull as a common finding5). However, there are only few reports of a solitary skull lesion due to syphilitic osteomyelitis without any other clinical manifestations of Treponema pallidum infection. We report a case of early syphilis presenting with syphilitic osteomyelitis of the skull that was the only pathological lesion identified in the patient.

CASE REPORT

A 20-year-old man was referred to our hospital due to intermittent headache and mild tenderness of a small coin-sized and slightly bulging lesion with softened cortical bone at the right parietal skull area. A small round radiolucent lesion was found on plain skull X-ray (Fig. 1A). The computed tomography (CT) and magnetic resonance imaging (MRI) revealed a 1.7 cm-sized, osteolytic and well enhanced lesion involving the whole thickness of skull bone; a solitary bone tumor was suspected (Fig. 1B, C). Bone scintigraphy demonstrated hot-uptake at the lesion (Fig. 1D). Preoperative laboratory tests showed evidence of syphilis; the erythrocyte sedimentation rate was 52 mm/h, the C-reactive protein (CRP) was 2.24 mg/dL, the venereal disease research laboratory (VDRL) test was positive, and Treponema pallidum haemagglutination assay (TPHA) was above 5,120 IU/L. However, the patient did not have any signs or symptoms of syphilis. The serology for the human immunodeficiency virus (HIV) was negative. There was no evidence of congenital syphilis or familial history of venereal diseases. The patient had a sexual encounter with a prostitute, which was the first and only sexual activity reported, 7 months previously. After three weeks of treatment with benzathine penicillin (intramuscular injection, 2,400,000 IU once a week), a resection of the lesion and cranioplasty with bone cement were performed simultaneously.

Fig. 1
The Results of the imaging study. A : Skull lateral x-ray shows a 17 mm radiolucent skull lesion (arrow) of parietal bone. B : Computer tomography shows a 17 mm osteolytic skull lesion (arrow) involving whole thickness of the right parietal bone. C : ...

Hematoxylin and eosin staining showed fibrous proliferation with lymphoplasmocytic infiltration forming an osteolytic defect suggesting non-specific chronic inflammatory changes. A spirochete was identified with the Warthin-starry staining (Fig. 2). The result of the polymerase chain reaction (PCR) with skull pathology showed a positive band for Treponema pallidum1). Cerebrospinal fluid (CSF) examinations, performed after confirming syphilitic osteomyelitis, were positive for VDRL testing without any evidence of inflammation.

Fig. 2
Identification of spirochetes by Warthin-starry staining (×1,000). Arrow indicates the Trepreponema pallidum.

DISCUSSION

Although there was no evidence by serologic test or pathological evaluations, T. pallidum infection is reported to have existed prior to AD 3007). Syphilitic osteomyelitis of the skull is a destructive bone disease that occurs as a complication of syphilis; however, it is an unusual complication of early stage (primary or secondary) syphilis9,10,14). In this case, the patient presented only with a syphilitic osteomyelitis of the skull even though he had early stage syphilis according to his clinical, familial, and social histories.

Recent epidemiological data from the US National Institutes of Health showed increasing prevalence of syphilis after 2002, and osteomyelitis lesions of the skull in early stage syphilis have been reported in HIV infected patients2,6,9,10). The situation is similar in the Republic of Korea12). Just like tuberculosis is increasing in HIV infected patients; syphilis also has been on the rise3,13,15). There have been many reports of syphilitic osteomyelitis not associated with HIV, and likely many unreported cases4,7,11). Syphilitic osteomyelitis can involve various bones including skull, clavicle, tibia, humerus, ulna, and the radius4). Syphilitic osteomyelitis can be treated with antibiotic therapy, benzathine penicillin or ceftriaxone4,16). We performed surgery on this patient with the initial impression of a skull tumor; osteomyelitis of skull as the sole and initial presenting finding of syphilis is very rare in patients without an immunological disorder, especially in this country. Radiologically, it is difficult to distinguish a syphilitic osteomyelitis from other radiolucent lesions of the skull, such as, eosinophilic granuloma, multiple myeloma, or cystic fibrous dysplasia8). This case illustrates that syphilis should be included in the differential diagnosis and that a bone biopsy for pathological examination and PCR and/or VDRL test should be performed.

CONCLUSION

Syphilitic osteomyelitis of the skull as the sole initial presenting lesion is very rare, and can be diagnosed by identification of spirochetes in the lesion combined with a positive PCR or VDRL of affected bone.

References

1. Behrhof W, Springer E, Bräuninger W, Kirkpatrick CJ, Weber A. PCR testing for treponema pallidum in paraffin-embedded skin biopsy specimens : test design and impact on the diagnosis of syphilis. J Clin Pathol. 2008;61:390–395. [PubMed]
2. Centers for Disease Control and Prevention (CDC) Primary and secondary syphilis-Jefferson county, Alabama, 2002-2007. MMWR Morb Mortal Wkly Rep. 2009;58:463–467. [PubMed]
3. Crampin AC, Glynn JR, Traore H, Yates MD, Mwaungulu L, Mwenebabu M, et al. Tuberculosis transmission attributable to close contacts and HIV status, Malawi. Emerg Infect Dis. 2006;12:729–735. [PubMed]
4. Dismukes WE, Delgado DG, Mallernee SV, Myers TC. Destructive bone disease in early syphilis. JAMA. 1976;236:2646–2648. [PubMed]
5. Ehrlich R, Kricun ME. Radiographic findings in early acquired syphilis : case report and cirtical review. AJR Am J Roentgenol. 1976;127:789–792. [PubMed]
6. Fenton KA, Breban R, Vardavas R, Okano JT, Martin T, Aral S, et al. Infectious syphilis in high-income settings in the 21st century. Lancet Infect Dis. 2008;8:244–253. [PubMed]
7. Gerszten PC, Gerszten E, Allison MJ. Diseases of the skull in pre-Columbian South American mummies. Neurosurgery. 1998;42:1145–1151. discussion 1151-1152. [PubMed]
8. Greenberg MS. Handbook of neurosurgery. ed 5th. New York: Thieme; 2001. pp. 890–891.
9. Gurland IA, Korn L, Edelman L, Wallach F. An unusual manifestation of acquired syphilis. Clin Infect Dis. 2001;32:667–669. [PubMed]
10. Huang I, Leach JL, Fichtenbaum CJ, Narayan RK. Osteomyelitis of the skull in early-acquired syphilis : evaluation by MR imaging and CT. AJNR Am J Neuroradiol. 2007;28:307–308. [PubMed]
11. Lee CW, Lim MJ, Son D, Lee JS, Cheong MH, Park IS, et al. A case of cerebral gumma presenting as brain tumor in a human immunodeficiency virus (HIV)-negative patient. Yonsei Med J. 2009;50:284–288. [PMC free article] [PubMed]
12. Lee IS. Historical changes and the present situation of sexually transmitted diseases. J Korean Med Assoc. 2008;51:868–874.
13. Marra CM. Syphilis and human immunodeficiency virus : prevention and politics. Arch Neurol. 2004;61:1505–1508. [PubMed]
14. Pfeiffer K. [Unusual findings in the cranial vault during therapy of tertiary bone syphilis] Fortschr Geb Rontgenstr Nuklearmed. 1958;88:574–578. [PubMed]
15. Podwińska J. Syphilis and AIDS. Arch Immunol Ther Exp (Warsz) 1996;44:329–333. [PubMed]
16. Strada M, Gariboldi M, Segalini G, Meregaglia D. [Secondary syphilis. Localizations in the bones of the skull] Radiol Med. 1987;73:453–454. [PubMed]

Articles from Journal of Korean Neurosurgical Society are provided here courtesy of The Korean Neurosurgical Society