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Primary tuberculosis of zygomatic bone: A rare case report
G Model JIPH-1211; No. of Pages 3
ARTICLE IN PRESS Journal of Infection and Public Health xxx (2019) xxx–xxx
Contents lists available at ScienceDirect
Journal of Infection and Public Health journal homepage: http://www.elsevier.com/locate/jiph
Primary tuberculosis of zygomatic bone: A rare case report Hari Ram a , Satish Kumar b,∗ , Virendra Atam b , Madhu Kumar c a
Department of Oral and Maxillofacial Surgery, King George’s Medical University, Lucknow, India Department Of Medicine, King George’s Medical University, Lucknow, India c Department of Pathology, King George’s Medical University, Lucknow, India b
a r t i c l e
i n f o
Article history: Received 15 October 2018 Received in revised form 26 July 2019 Accepted 23 October 2019 Keywords: Zygomatic bone Tuberculosis Discharging sinus Anti-tubercular chemotherapy
a b s t r a c t Tubercular osteomyelitis of zygomatic bone is extremely rare. Here we have reported a rarest case of primary tuberculosis (TB) of zygomatic bone in a 20 year male who presented with discharging sinus over right zygomatic region. Anti-tubercular chemotherapy given and patient improved well. © 2019 The Author(s). Published by Elsevier Ltd on behalf of King Saud Bin Abdulaziz University for Health Sciences. This is an open access article under the CC BY-NC-ND license (http://creativecommons. org/licenses/by-nc-nd/4.0/).
Introduction Tuberculosis (TB) is one of the oldest disease of the world whose existence was acknowledged about 4000 years back by civilizations at that time such as Atharva Veda, Rig Veda, 3000-1800 BC and Samhita of Charaka and Shushruta, 1000 and 600 BC, Egyptian and the Greco-Roman [1,2]. The global prevalence of TB is 30 million out of which approximately 10 millions (30%) patients found in India only and among them 1–3% are cases of bone and joint TB. About 8 million TB cases develop every year and a minimum of one expiry at each 15 s [2]. It is a chronic granulomatous infectious disease next to Human Immune deficiency Virus (HIV) in causing fatality and loss of millions of lives, especially in underdeveloped nations [3]. Impoverishment, overpopulated living places, unawareness, health education, attempts to combat TB, progression of multi-drug resistant mycobacteria and rapid spread of HIV infection all leads to increase fatality and high infectiousness of TB [4]. Mycobacterium tuberculosis is the causative bacterial agent for TB which may affects a number of body systems like lungs (commonest site), lymph nodes and lymphatics, renal system, central nervous system, hepatic system, skeletal system, gastrointestinal system and oral cavity. Oral cavity TB found in 10–15% of all [5]. Of two tubercle bacilli, Human and bovine, bovine is causative agent for approximately 80% osteo-articular joint TB in less than 10 year of age, while in India human bacilli is major culprit [2].TB in oro-facial
∗ Corresponding author. E-mail addresses: [email protected] (H. Ram), [email protected] (S. Kumar).
region have indifferent clinical appearance. Hence it is difficult to diagnose. Oral lesions in oro-facial TB makes confusion and misdiagnosis because systemic symptoms appear subsequently in flow of disease. In different regions of head and neck, TB has been characterized but association with zygomatic bone is a rare entity [6]. Next to lungs, large bones and vertebral column are the frequent sites. Small flat bones of face, sinuses, naso-pharynx, nose, and facial bones are the unusual sites of TB while maxillary/zygoma TB is even rarer [7]. Zygomatic bone involvement as primary site of tubercular osteomyelitis is very rare. Skull TB is reported in 0.1%–3.7% of cases of all skeletal TB. Skull TB is found in 50% of children less than 10 years [8]. Here we reported a case of primary tuberculosis of zygoma in a young boy along with discussion on its characteristic features and management. To give knowledge of this unique and rare entity and to familiarize the clinicians, we have presented this case. Till now fewer subjects of zygomatic bone having primary TB have been narrated in the literature [9].
Case report A 20-year-old male presented to us with mild to moderate-grade fever, swelling and discharging sinus over right side zygomatic region since last 5 months. History of trauma, smoking, alcohol intake, drug intake, previous lung TB and TB contact was negative. Five months back patient developed a painful and tender swelling on his right zygomatic region. After one month of which swelling bursts and yellow pus discharge was noted. On local examination there was a sinus measuring 3 × 4 cm having undermined edges
https://doi.org/10.1016/j.jiph.2019.10.008 1876-0341/© 2019 The Author(s). Published by Elsevier Ltd on behalf of King Saud Bin Abdulaziz University for Health Sciences. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Please cite this article in press as: Ram H, et al. Primary tuberculosis of zygomatic bone: A rare case report. J Infect Public Health (2019), https://doi.org/10.1016/j.jiph.2019.10.008
G Model JIPH-1211; No. of Pages 3 2
ARTICLE IN PRESS H. Ram et al. / Journal of Infection and Public Health xxx (2019) xxx–xxx
Treatment Standard 4 drugs/first line anti-tubercular regimen (rifampicin 450 mg, pyrazinamide 1500 mg, isoniazid 300 mg, and ethambutol 800 mg) for 2 months (intensive phase), followed by isoniazid 300 mg and rifampicin 450 mg (continuation phase) was given for 7 months. Aspiration of creamy pus was done on two separate occasions. Patient was showing improvement with healing of the sinus in intensive phase. According to the World Health Organisation (WHO) recommendation of treatment for the tuberculosis of the bones is 9–12 months [9].
Outcome and follow up Fig. 1. CT of face coronal view showing radiolucency around zygomatic bone.
After full course treatment by ATT patient was totally asymptomatic. On follow up of two years post treatment, patient was without any recurrence.
Discussion
Fig. 2. CT of face axial view.
and yellowish pus discharge over the right zygomatic region. On palpation infra orbital along with lateral orbital margins were normal. Regional lymphadenopathy was absent. On the basis of history and clinical examination patient was send for routine blood investigations, chest X-ray and fine needle aspiration cytology (FNAC). Lab investigations showed haemoglobin (Hb) 8.9 gm%, erythrocyte sedimentation rate (ESR) 38 mm/h, total leukocyte count (TLC) 6800 cells/mm3 with polymorphs 58% and lymphocytes 42%. Tuberculin test was strongly positive with 16 × 18 mm induration. Chest X-ray was normal. Ziehl Neelsen staining of pus smear for AFB was negative. Culture of pus was sterile for pyogenic organisms, while it was positive for Mycobacterium tuberculosis. Cartridge based nucleic acid amplification test (CBNAAT) was done and it was positive for Mycobacterium tuberculosis. It was sensitive to all first-line antitubercular drugs. Computed tomography (CT) scan of face showed well defined circumscribed radiolucency around zygomatic bone (Figs. 1 and 2). On FNAC, cheesy material obtained showing multiple epithelioid cells granulomas comprising of epithelioid cells, lymphocytes and histiocytes in the background of caseous necrosis on microscopy. This made the diagnosis of tuberculosis of zygomatic bone with discharging sinus. Final diagnosis was made on the basis of histopathology. Differential diagnosis includes Septic abscess, Tubercular abscess, Orbital cellulitis.
Zygomatic bone TB is an extra-pulmonary TB which is rarely seen [8]. Only one case of TB of the zygomatic bone was explained by Penfold and Revington in analysis of a series of tuberculosis of the head and neck in 23 patients [10]. Penfold et al. also reported a case of orbital TB involving zygoma because of direct extension from paranasal sinus, lacrimal gland or maybe due to hematogenous spread [11]. Generally TB of facial bones is found to be associated with TB of other organs elsewhere in the body. It may be skeletal or pulmonary TB and both may occur with together. TB of skull is almost secondary to TB of lungs [8].But in our case there were no clinical or radiological evidence of pulmonary TB. Lysis of these tubercular foci leads to spreading of tubercular bacilli via blood or lymphatic route and ultimately TB of facial bones. Seeding of bacilli may also occur by direct spread from tuberculosis of side structures like orbit, paranasal sinuses, nasal mucosa and face. Secondary to pulmonary TB, TB of maxillary sinus is very common. Progression of the disease depends on immunity of host and virulence of bacilli. This is usually a disease of children and adolescents with male predominance [12]. Usually fluctuant swelling with or without discharging sinus are the first symptoms. In this case first symptom was zygomatic swelling followed by discharging sinus. Common presentations of facial bones TB are non-healing wounds and pus discharge from the sinus. Neuralgia and non-specific headache are other symptoms. In our case infection was not responding to conventional antibiotics which made suspicion of tuberculosis. We made the diagnosis on behalf of cytology and it was supported by osteolytic lesions in radiology. If radiology and histopathology are non-evident, enzyme linked immune sorbent assay (ELISA) for detection of IgG and IgM antibodies of selected mycobacterial antigens, and GeneXpert/CBNAAT, recent polymerase chain reaction (PCR) for their de-oxy-ribose nucleic acid (DNA) is required [13]. Many times the disease may go undiagnosed because of its chronic nature and vague symptoms and signs. Once diagnosis made, the management of TB done by adequate ATT drugs. Conservative mode of treatment by giving ATT is main therapy while surgery is done in cases of extensive destruction, presence of secondary infection, and intracranial involvement. We managed and treated our case by 9 months of ATT and no any surgical intervention required, because there was no evidence of extensive destruction or intracranial invasion.
Please cite this article in press as: Ram H, et al. Primary tuberculosis of zygomatic bone: A rare case report. J Infect Public Health (2019), https://doi.org/10.1016/j.jiph.2019.10.008
G Model JIPH-1211; No. of Pages 3
ARTICLE IN PRESS H. Ram et al. / Journal of Infection and Public Health xxx (2019) xxx–xxx
3
Conclusion
References
Zygomatic bone TB is very rare and it should not be ignored in cases of facial swellings and discharging sinuses. ATT plus surgical intervention needed in cases of extensive destruction, presence of secondary infection, and intracranial involvement. Use of appropriate diagnostic methods, along with timely intervention, leads to a favourable outcome.
[1] Duraiswamy PK, Tulsii SM. Five thousand years of orthopaedics in India. Clin Orthop 1971;75:269. [2] Sankaran B. Tuberculosis of bones and joints. Ind J Tub 1993;40:109–18. [3] World Health Organization. Global tuberculosis report 2018. World Health Organization; 2018. [4] Mahajan S, Srikant N, George T. Atypical presentation of oral tuberculosis ulcer. N Y State Dent J 2007;73:48–50. [5] Andrade NN, Mhatre TS. Orofacial tuberculosis — a 16 year experience with 46 cases. J Oral Maxillofacial Surg 2012;70:12–22. [6] Moon WK, Han MH, Chang KH, Im JG, Kim HJ, Sung KJ, et al. CT and MR imaging of head and neck tuberculosis. Radio Graphics 1997;17(2):91–402. [7] Mahindra S, Bais AS, Sohail MA, Maheshwari HB. Granulomatous osteomyelitis of the maxillary sinus. Otolaryngol 1979;8:255–8. [8] Sachdeva OP, Gulati SP, Kakkar V, Arora B. Tuberculous osteomyelitis of zygoma. Trop Doct 1993;23:190–1. [9] Al-Hazmi WA. Tuberculosis of the malar and zygomatic bone: a case report. Int J Health Sci 2011;5(July (2)):197. [10] Lau SK, Wei WI, Hsu C, Engzell UCG. Efficacy of the needle aspiration cytology in the diagnosis of tuberculous cervicallymphadenopathy. J Laryngol Otol 1990;104:24–7. [11] Penfold CN, Revington PJ. A review of 23 patients with tuberculosis of the head and neck. Br J Oral Maxillofac Surg 1996;34(6):508–10. [12] Mohanty S, Rao CJ, Mukherjee KC. TB of the skull. Intern Surg 1981;66:81–3. [13] Daniel MT. Tuberculosis. Harrison’s principles of internal medicine, vol 1, 13th edn New York: McGraw Hill; 1994. p. 714.
Funding No funding sources. Competing interests None declared. Ethical approval Not required. Acknowledgement None.
Please cite this article in press as: Ram H, et al. Primary tuberculosis of zygomatic bone: A rare case report. J Infect Public Health (2019), https://doi.org/10.1016/j.jiph.2019.10.008
ARTICLE IN PRESS Journal of Infection and Public Health xxx (2019) xxx–xxx
Contents lists available at ScienceDirect
Journal of Infection and Public Health journal homepage: http://www.elsevier.com/locate/jiph
Primary tuberculosis of zygomatic bone: A rare case report Hari Ram a , Satish Kumar b,∗ , Virendra Atam b , Madhu Kumar c a
Department of Oral and Maxillofacial Surgery, King George’s Medical University, Lucknow, India Department Of Medicine, King George’s Medical University, Lucknow, India c Department of Pathology, King George’s Medical University, Lucknow, India b
a r t i c l e
i n f o
Article history: Received 15 October 2018 Received in revised form 26 July 2019 Accepted 23 October 2019 Keywords: Zygomatic bone Tuberculosis Discharging sinus Anti-tubercular chemotherapy
a b s t r a c t Tubercular osteomyelitis of zygomatic bone is extremely rare. Here we have reported a rarest case of primary tuberculosis (TB) of zygomatic bone in a 20 year male who presented with discharging sinus over right zygomatic region. Anti-tubercular chemotherapy given and patient improved well. © 2019 The Author(s). Published by Elsevier Ltd on behalf of King Saud Bin Abdulaziz University for Health Sciences. This is an open access article under the CC BY-NC-ND license (http://creativecommons. org/licenses/by-nc-nd/4.0/).
Introduction Tuberculosis (TB) is one of the oldest disease of the world whose existence was acknowledged about 4000 years back by civilizations at that time such as Atharva Veda, Rig Veda, 3000-1800 BC and Samhita of Charaka and Shushruta, 1000 and 600 BC, Egyptian and the Greco-Roman [1,2]. The global prevalence of TB is 30 million out of which approximately 10 millions (30%) patients found in India only and among them 1–3% are cases of bone and joint TB. About 8 million TB cases develop every year and a minimum of one expiry at each 15 s [2]. It is a chronic granulomatous infectious disease next to Human Immune deficiency Virus (HIV) in causing fatality and loss of millions of lives, especially in underdeveloped nations [3]. Impoverishment, overpopulated living places, unawareness, health education, attempts to combat TB, progression of multi-drug resistant mycobacteria and rapid spread of HIV infection all leads to increase fatality and high infectiousness of TB [4]. Mycobacterium tuberculosis is the causative bacterial agent for TB which may affects a number of body systems like lungs (commonest site), lymph nodes and lymphatics, renal system, central nervous system, hepatic system, skeletal system, gastrointestinal system and oral cavity. Oral cavity TB found in 10–15% of all [5]. Of two tubercle bacilli, Human and bovine, bovine is causative agent for approximately 80% osteo-articular joint TB in less than 10 year of age, while in India human bacilli is major culprit [2].TB in oro-facial
∗ Corresponding author. E-mail addresses: [email protected] (H. Ram), [email protected] (S. Kumar).
region have indifferent clinical appearance. Hence it is difficult to diagnose. Oral lesions in oro-facial TB makes confusion and misdiagnosis because systemic symptoms appear subsequently in flow of disease. In different regions of head and neck, TB has been characterized but association with zygomatic bone is a rare entity [6]. Next to lungs, large bones and vertebral column are the frequent sites. Small flat bones of face, sinuses, naso-pharynx, nose, and facial bones are the unusual sites of TB while maxillary/zygoma TB is even rarer [7]. Zygomatic bone involvement as primary site of tubercular osteomyelitis is very rare. Skull TB is reported in 0.1%–3.7% of cases of all skeletal TB. Skull TB is found in 50% of children less than 10 years [8]. Here we reported a case of primary tuberculosis of zygoma in a young boy along with discussion on its characteristic features and management. To give knowledge of this unique and rare entity and to familiarize the clinicians, we have presented this case. Till now fewer subjects of zygomatic bone having primary TB have been narrated in the literature [9].
Case report A 20-year-old male presented to us with mild to moderate-grade fever, swelling and discharging sinus over right side zygomatic region since last 5 months. History of trauma, smoking, alcohol intake, drug intake, previous lung TB and TB contact was negative. Five months back patient developed a painful and tender swelling on his right zygomatic region. After one month of which swelling bursts and yellow pus discharge was noted. On local examination there was a sinus measuring 3 × 4 cm having undermined edges
https://doi.org/10.1016/j.jiph.2019.10.008 1876-0341/© 2019 The Author(s). Published by Elsevier Ltd on behalf of King Saud Bin Abdulaziz University for Health Sciences. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Please cite this article in press as: Ram H, et al. Primary tuberculosis of zygomatic bone: A rare case report. J Infect Public Health (2019), https://doi.org/10.1016/j.jiph.2019.10.008
G Model JIPH-1211; No. of Pages 3 2
ARTICLE IN PRESS H. Ram et al. / Journal of Infection and Public Health xxx (2019) xxx–xxx
Treatment Standard 4 drugs/first line anti-tubercular regimen (rifampicin 450 mg, pyrazinamide 1500 mg, isoniazid 300 mg, and ethambutol 800 mg) for 2 months (intensive phase), followed by isoniazid 300 mg and rifampicin 450 mg (continuation phase) was given for 7 months. Aspiration of creamy pus was done on two separate occasions. Patient was showing improvement with healing of the sinus in intensive phase. According to the World Health Organisation (WHO) recommendation of treatment for the tuberculosis of the bones is 9–12 months [9].
Outcome and follow up Fig. 1. CT of face coronal view showing radiolucency around zygomatic bone.
After full course treatment by ATT patient was totally asymptomatic. On follow up of two years post treatment, patient was without any recurrence.
Discussion
Fig. 2. CT of face axial view.
and yellowish pus discharge over the right zygomatic region. On palpation infra orbital along with lateral orbital margins were normal. Regional lymphadenopathy was absent. On the basis of history and clinical examination patient was send for routine blood investigations, chest X-ray and fine needle aspiration cytology (FNAC). Lab investigations showed haemoglobin (Hb) 8.9 gm%, erythrocyte sedimentation rate (ESR) 38 mm/h, total leukocyte count (TLC) 6800 cells/mm3 with polymorphs 58% and lymphocytes 42%. Tuberculin test was strongly positive with 16 × 18 mm induration. Chest X-ray was normal. Ziehl Neelsen staining of pus smear for AFB was negative. Culture of pus was sterile for pyogenic organisms, while it was positive for Mycobacterium tuberculosis. Cartridge based nucleic acid amplification test (CBNAAT) was done and it was positive for Mycobacterium tuberculosis. It was sensitive to all first-line antitubercular drugs. Computed tomography (CT) scan of face showed well defined circumscribed radiolucency around zygomatic bone (Figs. 1 and 2). On FNAC, cheesy material obtained showing multiple epithelioid cells granulomas comprising of epithelioid cells, lymphocytes and histiocytes in the background of caseous necrosis on microscopy. This made the diagnosis of tuberculosis of zygomatic bone with discharging sinus. Final diagnosis was made on the basis of histopathology. Differential diagnosis includes Septic abscess, Tubercular abscess, Orbital cellulitis.
Zygomatic bone TB is an extra-pulmonary TB which is rarely seen [8]. Only one case of TB of the zygomatic bone was explained by Penfold and Revington in analysis of a series of tuberculosis of the head and neck in 23 patients [10]. Penfold et al. also reported a case of orbital TB involving zygoma because of direct extension from paranasal sinus, lacrimal gland or maybe due to hematogenous spread [11]. Generally TB of facial bones is found to be associated with TB of other organs elsewhere in the body. It may be skeletal or pulmonary TB and both may occur with together. TB of skull is almost secondary to TB of lungs [8].But in our case there were no clinical or radiological evidence of pulmonary TB. Lysis of these tubercular foci leads to spreading of tubercular bacilli via blood or lymphatic route and ultimately TB of facial bones. Seeding of bacilli may also occur by direct spread from tuberculosis of side structures like orbit, paranasal sinuses, nasal mucosa and face. Secondary to pulmonary TB, TB of maxillary sinus is very common. Progression of the disease depends on immunity of host and virulence of bacilli. This is usually a disease of children and adolescents with male predominance [12]. Usually fluctuant swelling with or without discharging sinus are the first symptoms. In this case first symptom was zygomatic swelling followed by discharging sinus. Common presentations of facial bones TB are non-healing wounds and pus discharge from the sinus. Neuralgia and non-specific headache are other symptoms. In our case infection was not responding to conventional antibiotics which made suspicion of tuberculosis. We made the diagnosis on behalf of cytology and it was supported by osteolytic lesions in radiology. If radiology and histopathology are non-evident, enzyme linked immune sorbent assay (ELISA) for detection of IgG and IgM antibodies of selected mycobacterial antigens, and GeneXpert/CBNAAT, recent polymerase chain reaction (PCR) for their de-oxy-ribose nucleic acid (DNA) is required [13]. Many times the disease may go undiagnosed because of its chronic nature and vague symptoms and signs. Once diagnosis made, the management of TB done by adequate ATT drugs. Conservative mode of treatment by giving ATT is main therapy while surgery is done in cases of extensive destruction, presence of secondary infection, and intracranial involvement. We managed and treated our case by 9 months of ATT and no any surgical intervention required, because there was no evidence of extensive destruction or intracranial invasion.
Please cite this article in press as: Ram H, et al. Primary tuberculosis of zygomatic bone: A rare case report. J Infect Public Health (2019), https://doi.org/10.1016/j.jiph.2019.10.008
G Model JIPH-1211; No. of Pages 3
ARTICLE IN PRESS H. Ram et al. / Journal of Infection and Public Health xxx (2019) xxx–xxx
3
Conclusion
References
Zygomatic bone TB is very rare and it should not be ignored in cases of facial swellings and discharging sinuses. ATT plus surgical intervention needed in cases of extensive destruction, presence of secondary infection, and intracranial involvement. Use of appropriate diagnostic methods, along with timely intervention, leads to a favourable outcome.
[1] Duraiswamy PK, Tulsii SM. Five thousand years of orthopaedics in India. Clin Orthop 1971;75:269. [2] Sankaran B. Tuberculosis of bones and joints. Ind J Tub 1993;40:109–18. [3] World Health Organization. Global tuberculosis report 2018. World Health Organization; 2018. [4] Mahajan S, Srikant N, George T. Atypical presentation of oral tuberculosis ulcer. N Y State Dent J 2007;73:48–50. [5] Andrade NN, Mhatre TS. Orofacial tuberculosis — a 16 year experience with 46 cases. J Oral Maxillofacial Surg 2012;70:12–22. [6] Moon WK, Han MH, Chang KH, Im JG, Kim HJ, Sung KJ, et al. CT and MR imaging of head and neck tuberculosis. Radio Graphics 1997;17(2):91–402. [7] Mahindra S, Bais AS, Sohail MA, Maheshwari HB. Granulomatous osteomyelitis of the maxillary sinus. Otolaryngol 1979;8:255–8. [8] Sachdeva OP, Gulati SP, Kakkar V, Arora B. Tuberculous osteomyelitis of zygoma. Trop Doct 1993;23:190–1. [9] Al-Hazmi WA. Tuberculosis of the malar and zygomatic bone: a case report. Int J Health Sci 2011;5(July (2)):197. [10] Lau SK, Wei WI, Hsu C, Engzell UCG. Efficacy of the needle aspiration cytology in the diagnosis of tuberculous cervicallymphadenopathy. J Laryngol Otol 1990;104:24–7. [11] Penfold CN, Revington PJ. A review of 23 patients with tuberculosis of the head and neck. Br J Oral Maxillofac Surg 1996;34(6):508–10. [12] Mohanty S, Rao CJ, Mukherjee KC. TB of the skull. Intern Surg 1981;66:81–3. [13] Daniel MT. Tuberculosis. Harrison’s principles of internal medicine, vol 1, 13th edn New York: McGraw Hill; 1994. p. 714.
Funding No funding sources. Competing interests None declared. Ethical approval Not required. Acknowledgement None.
Please cite this article in press as: Ram H, et al. Primary tuberculosis of zygomatic bone: A rare case report. J Infect Public Health (2019), https://doi.org/10.1016/j.jiph.2019.10.008