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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 11  |  Issue : 1  |  Page : 123-125

A rare case of calvarial tuberculosis in post-COVID-19 scenario


1 Department of Neurosurgery, IMS and SUM Hospital, Bhubaneswar, Odisha, India
2 Department of Microbiology, SUM Hospital, Bhubaneswar, Odisha, India

Date of Submission04-Sep-2021
Date of Decision10-Oct-2021
Date of Acceptance27-Nov-2021
Date of Web Publication12-Mar-2022

Correspondence Address:
Abhijit Acharya
Department of Neurosurgery, IMS and SUM Hospital, Bhubaneswar, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmy.ijmy_180_21

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  Abstract 


Isolated calvarial involvement with tuberculosis (TB) is a very rare entity, with the incidence of only 0.01% of all patients with mycobacterial infections. The factors attributable could be malnutrition, poor socioeconomic conditions, and immunodeficiency syndromes. We hereby present the case of a 35-year-old male who had recently recovered from coronavirus disease 2019 and a diagnosed case of Evan's syndrome with secondary hemophagocytic lymphohistiocytosis who presented with a scalp swelling on the right frontotemporal region. He presented to the emergency department with acute-onset generalized tonic − clonic seizures with high-grade fever. Clinically, the swelling appeared like a cystic swelling of the scalp. On evaluation, there was a collection present below the scalp communicating with the extradural space, involving the underlying skull bone. The patient was operated with drainage of the abscess plus excision of the pathological underlying skull bone. The pus revealed florid amount of acid-fast bacillus on Ziehl–Neelsen staining. The patient was started on four drugs Category 1 antitubercular regimen. The patient responded well to the combined surgical and medical treatment. It should be emphasized that TB can involve any part of the body. It should be kept as differential diagnosis of any chronic inflammatory lesion involving the bony skeleton, especially in endemic countries where combined surgical and medical treatment is usually sufficient to provide a cure.

Keywords: Calvarial tuberculosis, coronavirus disease 2019, Evans syndrome


How to cite this article:
Acharya A, Dash A, Otta S. A rare case of calvarial tuberculosis in post-COVID-19 scenario. Int J Mycobacteriol 2022;11:123-5

How to cite this URL:
Acharya A, Dash A, Otta S. A rare case of calvarial tuberculosis in post-COVID-19 scenario. Int J Mycobacteriol [serial online] 2022 [cited 2022 May 25];11:123-5. Available from: https://www.ijmyco.org/text.asp?2022/11/1/123/339502




  Introduction Top


Calvarial tuberculosis (CTB) is a rare entity reported in only 0.01% cases of mycobacterial infections; primary involvement of the calvarium without a focus of pulmonary tuberculosis (PTB) is still rarer.[1] However, its incidence seems to be on the rise, especially in developing countries. Some of the factors attributable for this rising incidence could be malnutrition, poor socioeconomic conditions, and immunodeficiency syndromes.[2] Evan's syndrome is an autoimmune disorder leading to autoimmune destruction of red blood cells, platelets, and often neutrophils while secondary hemophagocytic lymphohistiocytosis (HLH) is a severe systemic inflammatory syndrome causing overactive immune cells producing huge amounts of cytokine. We present hereby an interesting case of primary CTB in a known case of Evan's syndrome with HLH following recovery from coronavirus disease 2019 (COVID-19).


  Case Report Top


A 35-year-old male is a diagnosed case of Evan's syndrome with secondary HLH who is an advocate by profession presented to the emergency department with acute-onset generalized tonic − clonic seizures. He had recently recovered from COVID-19 1 month ago. Post-COVID recovery, he developed a scalp swelling associated with high-grade fever without chills or rigor. The scalp swelling was present on the right frontotemporal region, which was slowing and progressively increasing in size. The swelling on the clinical examination was globular in shape, 4 cm × 4 cm in size with stretched out skin surface, and ill-defined margins, with no sinus or scar. The swelling was nonpulsatile, nonreducible, fluctuant, and nontender, with a normal overlying skin temperature. The swelling was present above the calvarial bone. On evaluation, magnetic resonance imaging (MRI) of the brain suggested osteomyelitis of the right frontal and right parietal bone with a subgaleal and epidural abscess [Figure 1]. The patient was operated with drainage of the abscess plus excision of the pathological underlying skull bone. The pus was whitish, thick, and around 40–50 ml. The bone was pathological with destroyed calvarial bone segment. The pus was sent for cultures and tubercular, bacterial, and fungal stains. The bone fragments were sent for biopsy. The pus revealed florid amount of acid-fast bacillus (AFB) on Ziehl–Neelsen staining [Figure 2]. The biopsy revealed epithelioid granulomas with Langhan's giant cells. The patient was started on four drugs Category 1 antitubercular intensive phase for 3 months followed by three drugs for the next 6 months. The patient responded well to the combined surgical and medical treatment. The section from the mesenteric lymph node revealed multiple coalescent caseating granulomas and epithelioid granulomas with Langhans giant cells. Gradually, he became afebrile, symptoms have subsided.
Figure 1: T2-weighted image of the axial section of the brain showing a hyperintense subgaleal collection present in the scalp which is in communication with the epidural collection

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Figure 2: Florid amount of acid-fast bacilli present in the pus aspirated from the abscess cavity

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  Discussion Top


PTB is quite common in developing nations with poor hygiene, low-socioeconomic status, and poor access to health-care facilities. Since the late 20th century, the incidence and prevalence of both pulmonary and extraPTBs have increased owing to the increased prevalence of human immunodeficiency virus infection and travel to endemic countries. Other common risk factors for acquiring the infection include ethanol abuse, intravenous drug abuse, older age, and diabetes mellitus.[3]

Primary cytotrophoblast (CTB) is a rare disorder even among the communities with the high incidence of PTB, accounting for about 0.1%–3.7% of the skeletal TB.[4] There are about 370 cases of CTB in adults and children in the English literature until December 2019.[5] Most cases of CTB occur in the first two decades of life because of the greater affinity of mycobacteria for spongy bone. However, our patient was a 35-year-old male contradicting the above finding probably due affected because of interplay of his preexisting Evan's syndrome and recent COVID-19 affection. Furthermore, there are many instances of TB and COVID-19 coinfection worldwide with similar age and sex profiles as our case, although most of the reported cases were pulmonary in nature needing a deeper look into their correlation.[6]

Hematogenous spread or direct inoculation to the diploe has been proposed to be a cause of CTB.[7] However, in our patient even with careful history taking, examination, and investigations, no evidence of other primary tubercular focus could be elucidated. There was no previous trauma or surgery that could the cause of direct inoculation. There was no history of contact with any active TB patient. Similar to our patient in the series published by Raut et al.,[8] none of the patients had a primary focus. Thus, we propose that rampant steroid and immunosuppressant use during COVID-19 superadded with both his preexisting illness may have reactivated a long-forgotten bacillus following an inconsequential minor trauma.

After the granulation starts, the infection spreads through the inner and outer table of the skull, although a rare case of spread through diploe has also been reported.[9] Sutures are no barriers for the spread of granulation tissue[8] although dura can offer resistance. In our patient as well, there was tubercular abscess with extensive destruction of the calvarial bone. Parietal and frontal are the most common bone affected as per the literature because of the large proportion of cancellous bones.[2],[10],[11] Our patient also presented with a frontotemporal swelling without any sinus. There are three types of bony lesions described in CTB-(1) Small, circumscribed punched out defect in the bone with granulation tissue covering both internal and external aspects with little tendency for spread around the defect,[12] (2) Widespread destruction of the inner table with abundant extradural granulation tissue called diffuse TB of the skull,[13] and (3) Circumscribed and sclerotic type with marked thickening of the bone;[14] the first type being the most common. Our patient had widespread destruction of the inner table with interspersed punched-out lesions.

A high index of suspicion of TB is required for the early diagnosis of calvarial TB. Computed tomography imaging usually shows a punched-out, osteolytic lesion with ring enhancement and cannot differentiate between pyogenic osteomyelitis, metastasis, myeloma, hemangioma, and Langerhans cell histiocytosis.[4] MRI can show high-signal intensity soft-tissue mass and peripheral capsular contrast enhancement[8] and has an edge over CT to delineate the parenchymal involvement. AFB was found in the bone lesion in about 20% of these patients, and granuloma (compatible or suggestive with TB) was present in most cases.[2],[10],[11] In an endemic country like India, pus of nonpyogenic etiology should always be subjected for AFB examination and tubercular culture or GeneXpert examination.

Combined treatment with surgery and long-term antitubercular regimen helps to cure the disease.[2] Although there is no consensus on the duration of the antitubercular regimen, four-drug regimen for 3 months followed by three-drug regimen for 6 months was helpful in our case for clinical as well as microbiological cure. The early diagnosis often helps in preventing further spread of infection and associated complications.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initial will not be published and due efforts will be made to conceal his identity. Further, there is no ethical concern raised in the present case report.

Acknowledgment

We acknowledge SOA University, Bhubaneswar, Odisha, India, for their help in doing this work.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Davidson PT, Horowitz I. Skeletal tuberculosis. A review with patient presentations and discussion. Am J Med 1970;48:77-84.  Back to cited text no. 1
    
2.
Datta SG, Bhatnagar V, Pan S, Mehta R, Sharma C. Primary calvarial tuberculosis: A report of three cases. Br J Neurosurg 2019;33:196-201.  Back to cited text no. 2
    
3.
Villanueva Sáenz E, Martínez Hernández Magro P, Fernando Alvarez-Tostado Fernández J, Valdés Ovalle M. Colonic tuberculosis. Dig Dis Sci 2002;47:2045-8.  Back to cited text no. 3
    
4.
Gupta PK, Kolluri VR, Chandramouli BA, Venkataramana NK, Das BS. Calvarial tuberculosis: A report of two cases. Neurosurgery 1989;25:830-3.  Back to cited text no. 4
    
5.
Sant'Ann CC, Perinei PL, Machado de Pessôa AC, Goudouris E, Cisne Frota AC. Calvarial tuberculosis: Two cases in children. Rev Inst Med Trop Sao Paulo 2020;62:e68.  Back to cited text no. 5
    
6.
Ortiz-Martinez Y, Mejia-Alzate C, Vega-Vera A, Fajardo-Rivero JE, Rodriguez-Morales AJ. Drug-resistant tuberculosis and COVID-19 co-infection: A systematic review of case reports. Int J Mycobacteriol 2021;10:214-5.  Back to cited text no. 6
    
7.
Biniwale SN, Rajshekhar V. Tuberculous osteomyelitis of the bone flap following craniotomy for a glioma. Neurol India 2000;48:91-2.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Raut AA, Nagar AM, Muzumdar D, Chawla AJ, Narlawar RS, Fattepurkar S, et al. Imaging features of calvarial tuberculosis: A study of 42 cases. AJNR Am J Neuroradiol 2004;25:409-14.  Back to cited text no. 8
    
9.
Barton CJ. Tubercolosis of the vault of the skull. Br J Radiol 1961;34:286-90.  Back to cited text no. 9
    
10.
Das S, Nayar A, Paul DK, Basu S, Sen S, Bhattacharya M, et al. Calvarial tuberculosis with multiple intracranial tuberculomas: A rare association in a severely malnourished child with disseminated tuberculosis. J Pediatr Infect Dis 2016;11:042-7.  Back to cited text no. 10
    
11.
Pati S, De S, Ghosh TN, Ghosh MK. Multifocal pure tubercular osteomyelitis: An unusual presentation in childhood. Indian J Tuberc 2017;64:136-40.  Back to cited text no. 11
    
12.
Volkman R. Perforating tuberculosis of the bones of the calvaria. Central Bl Chir 1880;7:305-7.  Back to cited text no. 12
    
13.
Koening F. Traite de Pathologie et Chirurgie Speciale. Paris: Delahaye et Lecrosnier; 1888. p. 184.  Back to cited text no. 13
    
14.
Mohanty S, Rao CJ, Mukherjee KC. Tuberculosis of the skull. Int Surg 1981;66:81-3.  Back to cited text no. 14
    


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