View/Download PDF

Translate this page into:
Notice: Please configure GTranslate from WP-Admin -> Settings -> GTranslate to see it in action.

Case Report
2021
:12;
275
doi:
10.25259/SNI_435_2021

Spinal intramedullary abscess due to Candida albicans in an immunocompetent patient: A rare case report

, , , , , , , , ,
1Department of Medicine, Catanduva Medical School (FAMECA-UNIFIPA), Catanduva, São Paulo, Brazil.
2Department of Medicine, Pontifical Catholic University of Goiás, Goiânia, Brazil.
3Department of Medicine, Faculty of Medicine of ABC, Santo André, São Paulo, Brazil.
4Department of Medicine, School of Medicine of Pontifical Catholic University of São Paulo, Sorocaba, São Paulo, Brazil.
5Department of Neurosurgery, Santa Paula Hospital, São Paulo, Brazil.
6Department of Pathology, DASA, São Paulo, Brazil.
7Department of Neurology and Neurosurgery, University of Caxias do Sul, Caxias do Sul, Rio Grande do Sul, Brazil.
8Department of Medicine, Division of Neurology, Catholic Pontifical University of São Paulo, Sorocaba, São Paulo, Brazil.
*Corresponding author: Paulo Eduardo Albuquerque Zito Raffa, Catanduva Medical School (FAMECA-UNIFIPA), Catanduva, São Paulo, Brazil. pauloeduardoazr@gmail.com
Received: , Accepted: ,
Copyright: © 2021 Surgical Neurology International
Licence

This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: Raffa PE, Vencio RC, Ponce AC, Malamud BP, Vencio IC, Pacheco CC, et al. Spinal intramedullary abscess due to Candida albicans in an immunocompetent patient: A rare case report. Surg Neurol Int 2021;12:275.

Abstract

Background:

A spinal intramedullary abscess is a rare clinical entity in which patients classically present with a subacute myelopathy and progressive paraplegia, sensory deficits, and/or bowel and bladder dysfunction. We report the second case of spinal intramedullary abscess caused by Candida albicans to ever be published and the first case of its kind to be surgically managed.

Case Description:

A 44-year-old female presented with severe lumbar pain associated with paraparesis, incontinence, and paraplegia. She reported multiple hospital admissions and had a history of seizures, having already undergone treatment for neurotuberculosis and fungal infection of the central nervous system unsuccessfully. Nevertheless, no laboratory evidence of immunosuppression was identified on further investigation. Magnetic resonance imaging showed a D10-D11, well-circumscribed, intramedullary mass within the conus, which was hypointense on T1-weighted imaging and hyperintense on T2/STIR weighted. The patient underwent surgery for removal and biopsy of the lesion, which provided the diagnosis of an intramedullary abscess caused by C. albicans, a very rare condition with only one case reported in literature so far.

Conclusion:

C. albicans intramedullary abscess is a very rare clinical entity, especially in immunocompetent patients. We highlight C. albicans as an important etiology that must be considered in differential diagnosis. Critical evaluation of every case, early diagnosis, timely referral and surgical management of the abscess is essential to improve neurological outcome.

Keywords

Abscess
Candida albicans
Central nervous system
Conus medullaris
Intramedullary

INTRODUCTION

Involvement of the central nervous system (CNS) by Candida sp. usually leads to meningitis and cerebral microabscesses, especially in immunocompromised individuals or when anatomic barriers are breached by surgery or implanted devices.[17] However, CNS involvement in immunocompetent patients represents an even rarer entity.[7] To the best of our knowledge, Candida albicans intramedullary abscesses had only been reported once in an immunocompetent who did not need neurosurgery.[39] Here, we report a cauda equina C. albicans intramedullary abscess, representing the second report in literature and the first case approached surgically.

CASE REPORT

A 44-year-old female patient sought the neurosurgery service at Hospital Santa Paula with a complaint of lower back pain associated with paraparesis for 2 years with progressive worsening of the condition between crises. She reported development of paraplegia in the past year.

In an investigation of multiple hospital admissions, she previously underwent treatment for CNS fungal infection and neurotuberculosis in late 2020 with amphotericin B (for 6 weeks) followed by voriconazole (for 4 weeks) with an ineffective result. The cerebrospinal fluid (CSF) contained 43 cells (75% lymphocytes, monocytes 22%, macrophages 3%), protein 175 mg/dL; glucose 26 mg/dL; lactic acid 42 mg/dL; lactate dehydrogenase 30 U/L; adenosinedeaminase 6.2 U/L; gamma globulin 31.62%; microbiology negative; antibodies for syphilis, Borrelia burgdorferi, toxoplasmosis, cytomegalovirus (CMV), herpes simplex 1 and 2, herpes zoster, cysticercosis, human immunodeficiency virus (HIV), and human T-lymphotropic virus were not reactive; no cancerous cells were identified. She also presented a history of seizures and meningitis.

On neurological examination, the patient presented with dysarthria, horizontal nystagmus with preservation of extrinsic eye movement, Grade III strength in upper limbs with wrist spasticity, plegia in lower limbs, and anesthesia from the T12 level without proprioception. She also presented with bicipital, tricipital, and styloradial hyperreflexia in upper limbs, with bilateral exaltation points and positive Hoffmann’s sign, besides patellar and aquilean areflexia, and negative Babinski’s sign.

For better evaluation, magnetic resonance imaging (MRI) images of the brain and spine were requested. Brain MRI showed thickening and leptomeningeal impregnation by gadolinium compromising both lateral fissures, insula, and frontotemporal operculum, evidencing diffuse pachymeningitis [Figure 1]. The MRI of the spine showed marked diffuse leptomeningeal enhancement along the anterior and posterior pial surfaces of all medullary segments, as well as medullary cone, marked segmental narrowing of the medulla at T10-T11 levels with hypersignal in the weighted sequences at T1 and T2/STIR associated with the tumefactive effect of the cone spinal cord with intramedullary lesion at this level, hypointense in T1-weighted image and hyperintense in T2/STIR weighted [Figure 2].

Figure 1:: Preoperative magnetic resonance imaging. (a) Coronal gadolinium-enhanced image, (b) sagittal gadolinium-enhanced image, (c) axial T2/FLAIR-weighted image showing a slight reduction in the thickness of the leptomeningeal impregnation of the lateral fissures and frontotemporal operculum on both sides suggestive of diffuse pachymeningitis.
Figure 2:: Preoperative magnetic resonance imaging. (a) Axial T2/STIR-weighted image, sagittal T1 and T2/STIR-weighted image, (c) sagittal T2/STIR-weighted showing the intramedullary abscess hypointense on images (a and b) and hyperintense on image (c).

From the imaging findings and their relationship with the clinic, the patient underwent surgery and biopsy of the lesion. A T10-T11 laminectomy was performed and, after straight opening of the dura mater, a whitish lesion was found in the intramedullary region, suggestive of an abscess, being, therefore, drained and biopsied for analysis [Figure 3].

Figure 3:: Intramedullary abscess drainage surgery.

The culture of the surgical specimen tested positive for C. albicans and the histopathological evaluation of the lesion revealed extensive fibrinoid material and presence of hyaline septate pseudohyphae and fungal spores, with strong impregnation by Grocott methenamine silver stain [Figure 4]. These findings are consistent with intramedullary abscess due to C. albicans infection, being the second case report in the literature.

Figure 4:: Histopathology revealed numerous septate pseudohyphae and fungal spores embedded in fibrinoid material (a, H and E, ×400). There was intense silver impregnation by Grocott methenamine silver stain, suggestive of Candida albicans (b, GMS, ×400).

DISCUSSION

Candida spp. are considered opportunistic commensal pathogens that promote infection under host predisposing conditions.[55] When the CNS is involved in patients with systemic candidiasis, several clinical manifestations can be overlooked due to the severity of the patient’s situation. It is primarily found in immunocompromised hosts[56] or when anatomic barriers are breached by surgery, implanted devices, nonpenetrating blunt trauma to the back, or intermittent systemic corticosteroid,[17] typically leading to decrease in the level of consciousness, meningitis, and cerebral microabscesses.[7] However, other manifestations such as macroabscesses, sometimes suggestive of spinal tumors, are a more rare entity, especially when intramedullary.[56] Nevertheless, infection by Candida spp. has also been reported in immunocompetent individuals, as in this case report, but it is a rare event.[35,38,39,53]

There are a wide array of infectious causes of intramedullary myelitis, most of them caused by B. burgdorferi, Treponema pallidum, Mycobacterium tuberculosis, HIV, herpes simplex virus, varicella-zoster virus, CMV, Epstein-Barr virus), rabies, and schistosomiasis.[22] When the infection leads to an intramedullary spinal cord abscess, it is usually associated with high mortality and neurological morbidity and rarely encountered in modern neurosurgical practice.[10] We reviewed all cases of spinal intramedullary abscesses in the past 10 years [Table 1].[12-73] We found that approximately 68% of patients were male and 32% of female, the median age was 25, 86 years old (4 months – 82 years old), the main location was at thoracic level and the most frequent pathogen was from a tubercular source. Among all the reports, none of them identified an intramedullary abscess caused by C. albicans, which demonstrates the rarity of this entity once again.

Table 1:: Studies published in the past 10 years reporting intramedullary abscess.

C. albicans spinal infection still configures a challenging diagnostic. MRI is useful in diagnosing medullary abscesses, but those infections may mimic a neoplastic process as myxopapillary ependymoma or leptomeningeal carcinomatosis.[7] Besides, the albuminocytologic dissociation often present may come from Guillain–Barré syndrome, cerebral meningitis, malignant lymphoma, spinal hemangioma, multiple sclerosis, central hemorrhagic disease, myelitis, and cerebrospinal infarction, making diagnosis more difficult.[53] However, the CSF of patients with candidal CNS abscesses generally presents no biochemical or cytologic abnormalities. Proteins are usually discreetly high, and a discreet pleocytosis with polymorphonuclear or lymphocytic predominance can be equally frequent,[56] which increases the suspicion of a Candida infection, especially when the diagnosis is uncertain, as in the case presented. In addition, some studies still highlight genes related to candidiasis.[23] Colony-stimulating factor 2, Ras protein-specific guanine nucleotide-releasing factor 1, and phospholipase C gamma 2 are examples related to increased susceptibility to CNS Candida infection, but functional and bioinformatic studies are needed to identify possible variants that could have influenced Candida spp. infection reported in this article. As we have discussed, there are cases with no laboratory evidence of immunodeficiency or no identifiable cause of immunosuppression,[7] as we observed in this case report.

CONCLUSION

C. albicans intramedullary abscess represents a very rare clinical entity, especially in immunocompetent patients. It must, however, be considered as an important etiology for spinal intramedullary abscesses and as a differential diagnosis in intramedullary pathologies. Critical evaluation of every case, early diagnosis, timely referral and surgical management of the abscess is essential to improve neurological outcome.

Declaration of patient consent

Patient’s consent not required as patients identity is not disclosed or compromised.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

REFERENCES

  1. , . Congenital dermal sinus with extensive intramedullary expansion and an infected spinal epidermoid cyst in an infant. J Family Med Prim Care. 2018;7:1103-5.
    [Google Scholar]
  2. , , , . Intramedullary spinal cord abscess masquerading as spinal tumor. Indian Pediatr. 2011;48:973-4.
    [Google Scholar]
  3. , , . Fusarium species intramedullary spinal cord fungus ball: Case report. J Neurosurg Spine. 2019;31:440-6.
    [Google Scholar]
  4. , , . Chronic spinal cord abscess in an elderly patient. Surg Infect (Larchmt). 2011;12:333-4.
    [Google Scholar]
  5. , , , , , , . Ruptured long intramedullary spinal cord abscess successfully treated with antibiotic treatment. J Clin Neurosci. 2020;82:249-51.
    [Google Scholar]
  6. , , , , , . Finegoldia magna causing intramedullary thoracic spinal cord abscess in an infant. Anaerobe. 2019;56:57-60.
    [Google Scholar]
  7. , , , , , , , . Intradural cauda equina Candida abscess presenting with hydrocephalus: Case report. 20191-4.
    [Google Scholar]
  8. , , , , , . Intramedullary cervical abscess in the setting of aortic valve endocarditis. Asian Cardiovasc Thorac Ann. 2015;23:64-6.
    [Google Scholar]
  9. , , , . Disseminated coccidioidomycosis presenting with intramedullary spinal cord abscesses: Management challenges. Med Mycol Case Rep. 2016;15:1-4.
    [Google Scholar]
  10. , , . Intramedullary cervical spinal cord abscess. World Neurosurg. 2017;106:1049.e1-2.
    [Google Scholar]
  11. , . Infected congenital cervical dermal sinuses leading to spinal cord abscess: Two case reports and a review of the literature. Childs Nerv Syst. 2021;37:225-8.
    [Google Scholar]
  12. , , , . Intra-medullary tubercular abscess with spinal dysraphism: An unusual case. J Pediatr Neurosci. 2015;10:73-5.
    [Google Scholar]
  13. , , , , . Fulminant holocord intramedullary tubercular abscess with enigmatic presentation. Surg Neurol Int. 2013;4:32.
    [Google Scholar]
  14. , . Fatal Streptococcus melleri meningitis complicating missed infected intramedullary dermoid cyst secondary to dermal sinus in a Saudi child. J Trop Pediatr. 2013;59:246-9.
    [Google Scholar]
  15. , , . Precipitation sign: A new radiological sign for spinal intramedullary tubercular abscess. Spinal Cord. 2014;52:S1-2.
    [Google Scholar]
  16. , , , , , . Spinal congenital dermal sinus presenting as a diagnostic conundrum. Pediatr Neurosurg. 2012;48:187-90.
    [Google Scholar]
  17. . Intrathecal spinal abscesses due to Candida albicans in an immunocompetent man. 2018bcr-2017-223326.
    [Google Scholar]
  18. , . Intramedullary spinal cord abscess as complication of lumbar puncture: A case-based update. Childs Nerv Syst. 2013;29:1061-8.
    [Google Scholar]
  19. , , , . Intramedullary abscess by Staphylococcus aureus presenting as cauda equina syndrome to the emergency department. Case Rep Emerg Med. 2016;2016:9546827.
    [Google Scholar]
  20. , , , . Catastrophic intramedullary abscess caused by a missed congenital dermal sinus. J Korean Neurosurg Soc. 2015;57:225-8.
    [Google Scholar]
  21. , , , , . Paraplégie flasque due à des abcès intramédullaires et épiduraux à staphylocoques dorés. Méningite et spondylodiscite associées [Paraplegia associated with intramedullary spinal cord and epidural abcesses, meningitis and spondylodiscitis (Staphylococcus aureus)] Rev Neurol (Paris). 2012;168:868-72.
    [Google Scholar]
  22. , . Diagnosis and management of spinal cord emergencies. Handb Clin Neurol. 2017;140:319-35.
    [Google Scholar]
  23. , , , , , , . Impaired RASGRF1/ERK-mediated GMCSF response characterizes CARD9 deficiency in French-Canadians. J Allergy Clin Immunol. 2016;137:1178-88.e7.
    [Google Scholar]
  24. , , , . Spinal intramedullary tuberculosis with concurrent supra-and infratentorial intracranial disease in a 9-month-old boy: Case report and comprehensive review of the literature. World Neurosurg. 2017;106:37-45.
    [Google Scholar]
  25. , , , , . Intramedullary pyogenic abscess in the conus medullaris. Br J Neurosurg. 2012;26:118-9.
    [Google Scholar]
  26. , , , . A 51-year-old man with intramedullary spinal cord abscess having a patent foramen ovale. BMJ Case Rep. 2011;2011:bcr1120103512.
    [Google Scholar]
  27. , , , , . Intramedullary abscess of the cervical spinal cord in an otherwise healthy man. World Neurosurg. 2011;76:361.e15-9.
    [Google Scholar]
  28. , , , , . Recovery after tetraplegia caused by dermal sinus infection: Intramedullary abscess and tetraparesis. Pediatr Neurol. 2011;44:229-32.
    [Google Scholar]
  29. , , , , . Acute onset intramedullary spinal cord abscess with spinal artery occlusion: A case report and review. Eur Spine J. 2011;20:S294-301.
    [Google Scholar]
  30. , , . Proteus mirabilis abscess involving the entire neural axis. J Clin Neurosci. 2016;30:127-9.
    [Google Scholar]
  31. , , , , . Intramedullary spinal cord abscess: The result of a missed congenital dermal sinus. J Paediatr Child Health. 2015;51:223-5.
    [Google Scholar]
  32. , , , , . Intradural extramedullary cysticercal abscess of spine. Trop Parasitol. 2012;2:131-4.
    [Google Scholar]
  33. , , , , , . Intramedullary dermoid cyst infection mimicking holocord tumor: Should radical resection be mandatory? -A case report. Childs Nerv Syst. 2016;32:2249-53.
    [Google Scholar]
  34. , , , , . Distal cord-predominant longitudinally extensive myelitis with diffuse spinal meningitis and dural abscesses due to occult tuberculosis: A rare occurrence. J Pediatr Neurosci. 2016;11:77-9.
    [Google Scholar]
  35. , , , , . Intraoperative ultrasonographic evaluation of the spinal cord in cervical myelopathy. Spine (Phila Pa 1976). 1994;19:34-41.
    [Google Scholar]
  36. , , , . Intramedullary tubercular abscess with syrinx formation. J Pediatr Neurosci. 2012;7:61-3.
    [Google Scholar]
  37. , , , . Recurrent bacterial meningitis accompanied by a spinal intramedullary abscess. J Korean Neurosurg Soc. 2012;51:380-2.
    [Google Scholar]
  38. , , , . Subdural spinal fungal granuloma due to Candida tropicalis. Case report. J Neurosurg. 1979;50:395-6.
    [Google Scholar]
  39. , , , , , . Magnetic resonance image findings of spinal intramedullary abscess caused by Candida albicans Case report. Neurosurgery. 1995;36:411-2.
    [Google Scholar]
  40. , , , , , . Intramedullary tuberculoma combined with abscess: Case report and literature review. World Neurosurg. 2016;89:726.e1-4.
    [Google Scholar]
  41. , , , , , . An intramedullary tuberculous abscess of the conus in a 5-year-old child presenting with urinary dysfunction. World Neurosurg. 2011;76:592.e15-8.
    [Google Scholar]
  42. , , . Thoracic spinal cord glioblastoma mimicking epidural abscess: Case report and literature review. Cureus. 2017;9:e1631.
    [Google Scholar]
  43. , , , , , . Thoracic spinal cord intramedullary aspergillus invasion and abscess. J Clin Neurosci. 2015;22:404-6.
    [Google Scholar]
  44. , , , , . Multiple intracranial tuberculomas with an intra-medullary spinal cord tuberculoma in a pediatric patient. Cureus. 2020;12:e7248.
    [Google Scholar]
  45. , , . Management problems of intramedullary holocord abscess: An illustration in a pediatric case. Childs Nerv Syst. 2012;28:637-40.
    [Google Scholar]
  46. , , , , . Cervical spinal dermal sinus associated with intramedullary spinal cord abscess. Arq Neuropsiquiatr. 2017;75:133.
    [Google Scholar]
  47. , , . Cervical dermal sinus complicated with intramedullary abscess in a child: Case report and review of literature. Eur Spine J. 2014;23:192-6.
    [Google Scholar]
  48. , , , , . Dermal sinus tract of the thoracic spine presenting with intramedullary abscess and cranial nerve deficits. Pediatr Neurosurg. 2015;50:339-43.
    [Google Scholar]
  49. , , , . Holocord abscess in association with congenital dermal sinus. J Pediatr Neurosci. 2013;8:198-200.
    [Google Scholar]
  50. , , , , , . Concurrent intramedullary and intracranial tuberculomass. J Assoc Physicians India. 2018;66:72-4.
    [Google Scholar]
  51. , , , , , . Sequential magnetic resonance imaging finding of intramedullary spinal cord abscess including diffusion weighted image: A case report. Korean J Radiol. 2011;12:241-6.
    [Google Scholar]
  52. , , , , , . Concurrent intramedullary spinal cord and multiple intracranial tuberculomas with tuberculous optic neuritis: A rare case report. Indian J Tuberc. 2017;64:337-40.
    [Google Scholar]
  53. , , , , , , . Subdural spinal granuloma resulting from Candida albicans without immunosufficiency: Case report. Spine (Phila Pa 1976). 2002;27:E356-60.
    [Google Scholar]
  54. , , , , . Intramedullary dermoid cyst with relatively atypical symptoms: A case report and review of the literature. J Med Case Rep. 2013;7:104.
    [Google Scholar]
  55. , , , , , , . Differential behavior of nonalbicans Candida species in the central nervous system of immunocompetent and immunosuppressed mice. Front Microbiol. 2019;9:2968.
    [Google Scholar]
  56. , , , , . The central nervous system and infection by Candida species. Diagn Microbiol Infect Dis. 2000;37:169-79.
    [Google Scholar]
  57. , , , . The role of tertiary neurosurgical intervention in non-missile penetrating injuries of the spine. 2020
    [CrossRef] [Google Scholar]
  58. , . Pyomyelia presenting as acute flaccid paralysis. Oxf Med Case Reports. 2016;2016:omw052.
    [Google Scholar]
  59. , , , , , . Penetrating cervical spine injury and spinal cord intramedullary abscess. Arq Neuropsiquiatr. 2012;70:308-9.
    [Google Scholar]
  60. , , . Intramedullary abscess of the upper cervical spinal cord. Unusual presentation and dilemmas of management: Case report. Clin Neurol Neurosurg. 2013;115:1845-50.
    [Google Scholar]
  61. , , , , , . Abcès pottique intra médullaire: À propos d’un cas [Intramedullary Pott’s abscess: A case study] Pan Afr Med J. 2019;34:186.
    [Google Scholar]
  62. , , , . Intramedullary abscess of the cervical spinal cord caused by advanced periodontitis: Case report. No Shinkei Geka. 2016;44:685-9.
    [Google Scholar]
  63. , , , . Intramedullary spinal cord abscess and subsequent granuloma formation: A rare complication of vertebral osteomyelitis detected by diffusion-weighted magnetic resonance imaging. Neurosurg Focus. 2014;37:E12.
    [Google Scholar]
  64. , , , , . Rapidly progressive quadriparesis heralding disseminated coccidioidomycosis in an immunocompetent patient. J Clin Neurosci. 2014;21:1049-51.
    [Google Scholar]
  65. , , , . Intramedullary holocord abscess secondary to infected dermoid cyst. Acta Neurochir (Wien). 2018;160:209-12.
    [Google Scholar]
  66. , , , , . Intracranial and intramedullary tuberculoma with intravertebral abscess manifestation under anti-tuberculous treatment: A case report. Clin Neurol Neurosurg. 2013;115:1858-60.
    [Google Scholar]
  67. , , , , . Intramedullary abscess of the spinal cord in the setting of patent foramen ovale. World Neurosurg. 2011;76:361.e11-4.
    [Google Scholar]
  68. , , , . Metachronous occurrence of an intramedullary abscess following radical excision of a cervical intramedullary pilocytic astrocytoma. Neurol India. 2013;61:322-4.
    [Google Scholar]
  69. , , , , . Infected lumbar dermoid cyst mimicking intramedullary spinal cord tumor: Observations and outcomes. J Pediatr Neurosci. 2014;9:21-6.
    [Google Scholar]
  70. , , , . Spinal cord abscess secondary to infected dorsal dermal sinus in an infant: Uncommon presentation of a known entity. BMJ Case Rep. 2017;2017:bcr2017222366.
    [Google Scholar]
  71. , , . Intramedullary cervical abscess mimicking a spinal cord tumor in a 10-year-old girl: A case-based review. Childs Nerv Syst. 2018;34:2143-7.
    [Google Scholar]
  72. , , . Streptococcus pneumoniae meningitis complicated by an intramedullary abscess: A case report and review of the literature. J Med Case Rep. 2016;10:290.
    [Google Scholar]
  73. , , , , . Postoperative Mycoplasma hominis infections after neurosurgical intervention. J Neurosurg Pediatr. 2014;14:212-8.
    [Google Scholar]

Fulltext Views
10

PDF downloads
0
View/Download PDF
Download Citations
BibTeX
RIS
Show Sections

Related Posts:

© Copyright 2018 - Surgical Neurology International - All rights reserved.
Published by Scientific Scholar on behalf of Surgical Neurology International.