E-ISSN 1858-8360 | ISSN 0256-4408
 

Original Article 


SUDANESE JOURNAL OF PAEDIATRICS

2021; Vol 21, Issue No. 1

ORIGINAL ARTICLE

Diverse clinical presentations of tubercular parotitis in children: case series with review of literature

Shruti Dhingra (1), Ruchika Juneja (1)

(1) Department of Otolaryngology and Head and Neck Surgery, Maulana Azad Medical College, New Delhi, India

Correspondence to:

Shruti Dhingra

Department of Otolaryngology and Head and Neck Surgery, Maulana Azad Medical College, New Delhi, India

Email: shrudoc [at] hotmail.com

Received: 09 May 2020 | Accepted: 23 July 2020

How to cite this article:

Dhingra S, Juneja R. Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudan J Paediatr. 2021;21(1):67–75.

https://doi.org/10.24911/SJP.106-1588918898


ABSTRACT

Tuberculosis of the parotid gland is a rare entity in children. Manifestations can vary from acute parotitis to indolent benign parotid lesions, abscesses and fistulas in the parotid region. Diagnosis is challenging in the absence of constitutional symptoms and essentially non-contributory test reports. We discuss a series of six cases of parotid tuberculosis in children with different clinical presentations and management and review the literature on the subject.


KEYWORDS

Parotid; Tuberculosis; Children; Non-tubercular mycobacteria; Abscess.


INTRODUCTION

The most common extrapulmonary manifestation of Mycobacterium tuberculosis is cervical lymphadenopathy. Primary salivary tuberculosis remains an uncommon entity even in endemic countries like India. This may be attributed to the inhibitory effect of saliva over the infecting mycobacterium [1]. Poor dental hygiene and trauma lead to the inoculation of the oral mucosa. Most cases of parotid tuberculosis have been reported in adult patients. There are only a few isolated case reports of primary parotid tuberculosis in children in the world literature. We present a series of six cases of tuberculosis involving the parotid gland in children with their varied clinical presentations, seen over only 1 year, and discuss the literature review on the subject.


CASE REPORT

Case 1

A 4-year-old boy presented with right preauricular swelling that was gradually progressive for the past 20 days (Figure 1a). It was a diffuse swelling about 1 cm × 1.5 cm, non-tender with ill-defined borders. The oral cavity was normal. The facial nerve was intact. The patient had a history of associated low-grade fever for the past 2 weeks. There was no history of cough. A family history was positive for tuberculosis in the grandmother who lived with the family.

On investigation, the chest X-ray was normal. The erythrocyte sedimentation rate (ESR) was 26 mm/hour (Westergren). Sputum could not be obtained for analysis. The gastric aspirate was negative for acid-fast bacilli (AFB). Ultrasound revealed a well-defined cystic lesion in the right parotid parenchyma (Figure 1b). Computed tomography (CT) scan showed a hypoattenuating lesion, 15 mm × 9 mm, within the parotid suggestive of an abscess (Figure 1c). Ultrasound-guided fine-needle aspiration cytology (FNAC) was suggestive of epitheloid granulomas. Stain for AFB was negative. The patient had been vaccinated at birth, and bacillus Calmette-Guérin (BCG) scar was present. Tuberculin skin test had an induration of 10 mm. He was started on the standard four drug regimens (isoniazid: 5 mg/kg/day, rifampicin: 10 mg/kg/day, pyrazinamide: 25 mg/kg/day and ethambutol: 15 mg/kg/day) for 2 months, followed by 4 months of isoniazid and rifampicin. The cultures were positive for M. tuberculosis. The patient was clinically better after 2 months of therapy, and the swelling completely disappeared after 6 months.

Figure 1. (a) A 4-year-old child with right-sided parotid swelling. (b) Ultrasound revealed a well-defined cystic lesion in the right parotid parenchyma (arrow). (c) CT scan showed a hypoattenuating lesion, 15 mm × 9 mm, within the parotid suggestive of an abscess (arrow).

Case 2

A 7-year-old boy presented with swelling in the left parotid region for the past 1 year. He noticed another swelling around the same region that had developed in the past 1 month (Figure 2). There was no other cervical lymphadenopathy. The swelling was painless to begin with but had now become tender and painful. The parents also gave a history of chronic low-grade fever over the past 6 months. They had taken over the counter medications for fever and antibiotics prescribed by a private practitioner, but the swelling had persisted. There was no history of contact with tuberculosis, no history of chronic cough or significant weight loss.

On examination, both the swellings were fluctuant, and the overlying skin was erythematous. The chest X-ray was clear with no evidence of tuberculosis. Mantoux test was 25 mm × 25 mm induration. ESR was 34 mm/hour. An FNAC revealed pus filled with AFB. The culture of the pus revealed the growth of M. tuberculosis on Lowenstein-Jensen (LJ) media after 6 weeks. The patient was started immediately on antitubercular treatment for 6 months and called for repeated aspirations of pus. The pus was aspirated once a week for the first 4 weeks. Gradually, the pus aspirate decreased, and there was no further fluctuance. The patient was followed up for 10 months. There was no recurrence of the lesion at the site.

Figure 2. A 7-year-old child with left-sided tubercular abscess in the parotid region.

Case 3

A 9-month-old female child was brought by her parents with the complaints of swelling in the right parotid and the right submandibular region for the past 2 months. The swelling in the right parotid region was about 1 cm × 1 cm in size. There was a history of tuberculosis in the father who was diagnosed and started on antitubercular treatment before 1 month. The child had not been vaccinated at birth and had no BCG scar. FNAC done from both the parotid and submandibular swellings was suggestive of granulomatous lymphadenitis. Stain for AFB was positive. The chest X-ray was clear. After 3 months of starting antitubercular treatment, the lesions became fluctuant and spontaneously burst. On examination, there were draining sinuses at places, where the lesions existed (Figure 3). The antitubercular treatment was, however, continued till completion for 6 months. At 6 months of follow-up, the child showed clinical improvement in terms of weight gain and general development. However, the sinuses persisted at the submandibular and parotid region without any discharge.

Figure 3. A 9-month-old child presented with multiple discharging fistulas in the parotid and cervical region.

Case 4

A 5-year-old boy presented with right parotid and multiple cervical swellings for the past 8 months (Figure 4). He had been previously diagnosed based on FNAC samples from the parotid swelling and had been on antitubercular treatment for the past 4 months. This visit was due to the concern of the parents that the swelling in the parotid region had increased in size and was painful. On examination, the swelling in the parotid region was seen to be approximately 2 cm × 2.5 cm in size along with multiple ipsilateral cervical swelling. The swelling was tender, and the overlying skin was erythematous. The patient had no history of the recent onset of fever or upper respiratory tract infection. The chest X-ray was clear. Ultrasound of the neck and parotid regions revealed the involvement of both the superficial and deep lobes of the parotid with multiple microabscess and few enlarged lymph nodes in the neck. FNAC was suggestive of pus and was full of polymorphonuclear leukocytes. The patient was started on antibiotics for secondary infection along with the continuation of antitubercular treatment. The fluctuation resolved in about 2 weeks of antibiotic treatment. The patient was symptom free after this episode with no further fluctuation in the swellings which resolved at the completion of antitubercular treatment at 6-month follow-up visit.

Figure 4. A 5-year-old boy with right parotid and multiple cervical neck swellings.

Case 5

A 12-year-old boy presented with a right-sided parotid swelling and chronically draining sinus of the cheek for the past 11 months (Figure 5a). He had another palpable lymph node over the right supraclavicular region with excoriation of the overlying skin (Figure 5b). This patient had a history of multidrug-resistant pulmonary tuberculosis 7 years back, for which he had received antitubercular treatment for 9 months. He had been well over the past 6 years. The present episode was associated with fever, evening rise in temperature and weight loss (more than 5 kg in the past 6 months), and decreased appetite. The swelling in the parotid region was about 1.8 × 2 cm in size, non-tender with induration. The buccal mucosa was intact. On palpation of the swelling, pus could be seen emanating from the sinus opening in the skin. A second lymph node swelling in the right supraclavicular region was 2 cm × 2 cm with skin excoriation. A pus sample from the discharging sinus was suggestive of coagulase-negative Staphylococcus. FNAC from the parotid swelling revealed acute inflammatory exudate. Culture for AFB was awaited. Purified protein derivative (PPD) skin test showed an induration of 20 mm × 20 mm. ESR was 24 mm/hour. Gastric aspirate for AFB was negative. Human immunodeficiency virus (HIV) test was negative. Chest X-ray showed evidence of old healed pulmonary tuberculosis. Parotid ultrasound showed hetero-echoic lesions predominantly involving the superficial lobe of the parotid with a few central necrotic areas and sinus tract opening into the skin. Since no positive evidence of tuberculosis was present, the patient was started on antibiotics. Nevertheless, a strong suspicion of tuberculosis prevailed. After 2 weeks of antibiotics, an ultrasound-guided repeat FNAC from the parotid and supraclavicular lymph node was planned. The supraclavicular lymph node showed necrosis, degenerated cells and inflammatory infiltrate; stain for AFB was negative. However, the parotid swelling showed a similar picture, but stain for AFB was positive. The culture report obtained after 6 weeks was positive. The patient was started on antitubercular treatment once again. At 8 months of follow-up, the cheek sinus and excoriation of skin at the supraclavicular region persisted, however, there was no discharge from the draining sinus, and the supraclavicular lymph node had resolved.

Figure 5. (a) A 12-year-old boy with right parotid swelling and sinus tract in the cheek. (b) The same boy with supraclavicular lymph node and excoriation of the overlying skin.

Figure 6. A 4-year-old child with left parotid swelling that did not resolve with standard antitubercular therapy and had to the surgically excised.

Table 1. Comparison of six patients with regard to clinical presentations.

S no. Gender Age at presentation Laterality Clinical symptoms Cervical lymph nodes Chest X-ray ESR Sputum for AFB Gastric aspirate
1 Male 4 years Right Low-grade fever × 2 weeks, swelling parotid for 20 days.
Positive history of contact.
Small, non-palpable, diagnosed on CT scan NAD 26 Negative Negative
2 Male 7 years Left Swelling parotid for 1 year, another fluctuant parotid swelling for 1 month
Low-grade fever for 6 months.
None NAD 34 - -
3 Female 9 months Right Swelling parotid and submandibular gland. Sinus on skin after starting ATT for 3 months. Present NAD - - -
4 Male 5 years Right Swelling parotid for 8 months. Already on ATT. Developed abscess in parotid due to secondary infection present NAD - - -
5 Male 12 years Right Sinus on parotid 11 months. Supraclavicular node. Received ATT for 9 months-7 years back for MDR tuberculosis. Present supraclavicular Old healed lesions of tuberculosis 24 negative negative
6 Male 4 years Left Parotid swelling 2 months. No response to ATT. No constitutional symptoms, no history of contact. none NAD 30 negative Negative

Table 2. Comparison of the 6 patients with relevant investigations and findings.

Sno. USG FNAC PPD AFB stain Culture For AFB
1 USG and CT scan suggestive Epitheloid granulomas 10 mm Negative Positive
2 USG not done. Pus 25 mm Positive Positive
3 USG not done Granulomatous lymphadenitis - Positive -
4 Microabcesses in both superficial and deep lobes Polymorphonuclear Leukocytes in pus. Already diagnosed and receiving ATT.
5 Heteroechoic lesions in the superficial parotid lobe. Acute inflammatory exudate 20 mm Positive Positive
6 - Granulomatous lymphadenitis 13 mm Initially, AFB stain and culture negative. Patient did not improve on ATT. Superficial parotidectomy specimen. M. bovis was isolated. Unpasteurized milk consumption.

Case 6

A 4-year-old boy presented with left-sided parotid swelling for the past 2 months (Figure 6). He had no history of low-grade fever, cough, weight loss, loss of appetite or history of contact. ESR was 30 mm/hour. PPD skin test showed an induration of 13 mm × 13 mm. BCG vaccine scar was present. A chest X-ray showed no evidence of pulmonary tuberculosis. The gastric aspirate was negative. FNAC showed the features of granulomatous lymphadenitis, but stain for AFB was negative. The patient was started on standard antitubercular treatment for extrapulmonary tuberculosis. AFB culture report was available after 6 weeks which showed no growth of AFB on LJ media. After 3 months of treatment, the patient was not responding well to the antitubercular drugs. The pre-auricular swelling persisted and was the same size as before. A suspicion of drug-resistant tuberculosis/non-tubercular mycobacteria or other granulomatous condition was kept. A superficial parotidectomy was planned with facial nerve monitoring, and the lesion was excised. The sample was sent for nucleic acid amplification, polymerase chain reaction (PCR) and specific antigen testing. Mycobacterium bovis was isolated. Later history elicited the consumption of unpasteurised milk from the cattle that the family owned. The patient did well postoperatively and was on follow-up for 1 year. He was adequately counselled for drinking boiled and pasteurised milk. There was no recurrence of lymph nodes at any other site and his ESR gradually normalised.

In summary, (see Table 1 and Table 2) all of the cases were young children between 9 months and 12 years of age and came from low socioeconomic backgrounds. Male:female ratio was 5:1, and the right side of parotid predominated over the left was 4:2. None of the cases had bilateral parotid involvement. Besides the involvement of the parotid gland, four of the six children also had cervical lymphadenopathy. Skin over the parotid swelling was normal in two patients, and erythematous in two with abscess formation and presented with draining sinuses in 2 patients. The facial nerve was intact in all the cases. Constitutional symptoms such as fever, weight loss and loss of appetite were present in only two of the six cases. The chest X-ray was normal in five cases; only one child showed evidence of previously treated healed tuberculosis. Sputum and gastric aspirate were negative in all cases. One patient was already on antitubercular treatment at the time of presentation. Of the five cases, AFB stain was positive in three cases. AFB was isolated on culture in one case, in which the stain was negative. Epitheloid granulomas could be identified only in three of five cases. Culture and sensitivity were done in all cases, before initiating antitubercular treatment. All cases responded to antitubercular treatment except one, who later required a superficial parotidectomy and proved to be a case of atypical mycobacterium (M. bovis).


DISCUSSION

The first case of parotid tuberculosis was described by Von Stubenrauch in 1894 [2]. Primary tuberculosis of the parotid can evolve from a focus in the tonsil or gingivobuccal sulcus and ascend along the Stenson’s duct to involve the gland. This ascending infection can spread to the intraparotid, periparotid and cervical lymph nodes through the lymphatic drainage from the oral cavity or by hematogenous spread from a pulmonary focus [3]. Carmody [4] formulated a canalicular mode of spread from infected molar teeth. The submandibular gland is more often affected by secondary hematogenous spread than the parotid gland. Continuous salivary flow and inhibitory effect of proteolytic enzymes inhibit the growth of bacteria in the glands. Many years following a pulmonary infection, mycobacteria that are encapsulated within the intraglandular lymph nodes can get reactivated and present as salivary gland tuberculosis at a later date.

Clinically, tubercular parotitis can present in two forms. The first is an acute painful inflammatory lesion with diffuse glandular oedema simulating an acute parotitis or abscess. This occurs due to the involvement of intra- or periglandular lymph nodes. The second is a gradually progressive painless localised swelling involving the parenchyma of the parotid region that mimics a neoplasm. The lymphadenitis can progress to caseation and necrosis and can cause erythema of the overlying skin. It may occasionally rupture and present as a chronically draining sinus or fistula that does not heal with antibiotic treatment. Constitutional signs such as fever, weight loss and night sweats may be absent or minimal. The involvement of the facial nerve is rare and can occur in painful and invasive forms, mimicking a malignant neoplasm [5]. Differential diagnosis includes parotitis, mumps, sarcoidosis, actinomycosis, Sjogren’s syndrome, sialosis, cat-scratch disease, toxoplasmosis, tularaemia, actinomycosis and benign/malignant neoplasms of the parotid. Histology plays an important role in finding the pathogenic organism in these cases, in addition to ruling out benign tumours and malignant changes.

The diagnosis of tuberculosis of the parotid can be challenging. Since the lesions are slowly progressive and do not cause alarming systemic symptoms, it is usually long before parents notice them in children. A battery of tests is needed in order to clinch the diagnosis as no single test is completely reliable. Chest X-ray is usually clear in cases of primary parotid tuberculosis. Even in pulmonary tuberculosis, children rarely develop cavitary lesions. ESR is non-diagnostic. Diagnosis may only be suspected on FNAC that suggests granulomatous infection with epitheloid histiocytes. Cytology combined with AFB staining of the aspirated material achieves a better result. The cultures of mycobacteria are complimentary; however, the yield rate is not high [6].

The polymerase chain reaction is more sensitive and specific. However, obtaining an adequate specimen for analysis from a deep-seated parotid abscess in a child can be daunting and requires an accurate needle placement in an ultrasound-guided FNAC. Biopsy increases the risk of fistulisation and should be avoided. One feared complication of repeat FNAC in atypical mycobacteria is the creation of a persistently draining fistula. This can be avoided by doing accurate USG-guided aspiration the first time whenever there is a suspicion of tuberculosis. Avoiding the use of a very thick needle during the procedure may also be of some help. A strong index of suspicion for tuberculosis must be kept in endemic countries even if most of the test results are negative. There is an increased need for performing antigen and molecular testing such as deoxyribonucleic acid extraction and PCR amplification for the early identification and treatment of mycobacterium.

Non-tubercular mycobacterial (NTM) infections and multidrug-resistant tuberculosis of the parotid glands pose challenges and difficulties in diagnosis. In fact, NTM is more common in children <5 years of age (16-32 months). M. bovis, Mycobacterium avium intracellulare, Mycobacterium scrofulaceum and Mycobacterium kansasii are common in soil, water, domestic and wild animals and milk. In an endemic country like India where slum-dwelling and direct consumption of milk from cattle, without pasteurisation, are common practices, it is not uncommon to see multidrug-resistant and NTM infections. An increased close person-to-person contact, unhygienic living conditions, spread of HIV and immunocompromised status, especially in children, can explain a relatively high occurrence of a rare clinical presentation such as parotid tuberculosis in children.

PPD in postvaccinated children is unreliable and may be negative in non-tubercular mycobacterial disease. BCG only affords protection against disseminated and meningeal tuberculosis and death from tuberculosis. BCG vaccination at birth in institutional deliveries is a norm in most developing countries like India. Therefore, in previously vaccinated children and in countries where tuberculosis is endemic and exposure is common, the result of PPD testing cannot be a reliable indicator to rule in or rule out tuberculosis. Clinical suspicion is of prime importance along with supporting laboratory evidence.

In the experience of treating parotid tuberculosis, the authors observed clinical improvement at 6 months of antitubercular treatment for typical mycobacteria and 9 months-1 year in atypical mycobacteria. It may occasionally be essential to surgically excise the superficial parotid lymph nodes in case of no response to typical antitubercular therapy.


CONCLUSION

Through this case series, we would like to highlight that tuberculosis of the parotid gland in children can have varied clinical presentations and can be easily confused with acute parotitis and benign/malignant lesions of the parotid, leading to inadvertent parotidectomy. Sometimes, the lesions may be indolent without constitutional symptoms and may go unnoticed for a considerable length of time. The diagnosis of parotid tuberculosis can be challenging as a battery of tests performed is negative or non-contributory.

In the presence of strong suspicion of tuberculosis, the clinician needs to resort to better diagnostic modalities such as an ultrasound-guided FNAC, nucleic acid amplification test, tuberculosis (M. tuberculosis) PCR or BACTEC culture to clinch the diagnosis. Superficial parotidectomy may be required, in inconclusive cases or those not responding to anti-tubercular treatment, for histopathological diagnosis.


CONFLICTS OF INTEREST

The authors declare that there is no conflict of interest regarding the publication of this article.


FUNDING

None.


CONSENT FOR PUBLICATION

Written informed consent was obtained from all the participants


ETHICAL APPROVAL

Ethical clearance has been taken from the departmental ethics committee. All patient guardians have given consent for the participation and publication of medical details. Confidentiality was ensured at all stages.


REFERENCES

  1. Shah I. Multidrug resistant tuberculosis in children. Pediatr Infect Dis J. 2012;31(9):970–2. https://doi.org/10.1097/INF.0b013e318260b8a7
  2. Von Stubenrauch L. Einen Uberfall von tuberculoser Parotitis. Arch klin Chir. 1894;47:26–32.
  3. Kontopoulou T, Fanourgiakis P, Samarkos M, Mintzias D, Peppas C, Rontogianni D, et al. Tuberculosis of the parotid gland: case report and literature review. Med Mal Infect. 2004;34(10):488–90. https://doi.org/10.1016/S0399-077X(04)00178-7
  4. Carmody TE. Tuberculosis of parotid gland. Laryngoscope. 1914;24:873. https://doi.org/10.1288/00005537-191410000-00005
  5. Touiheme N, Kettani M, Messary A. Primary tuberculosis of the parotid gland: a rare differential diagnosis of parotid tumor. Laryngo Rhino Otologic. 1997;76(5):30811.
  6. Chou YH, Tiu CM, Liu CY, Hong TM, Lin CZ, Chiou HJ, et al. Tuberculosis of the parotid gland: sonographic manifestations and sonographically guided aspiration. J Ultrasound Med. 2004;23:1275–81. https://doi.org/10.7863/jum.2004.23.10.1275


How to Cite this Article
Pubmed Style

Dhingr S, Juneja R. Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudan J Paed. 2021; 21(1): 67-75. doi:10.24911/SJP.106-1588918898


Web Style

Dhingr S, Juneja R. Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. http://www.sudanjp.com/?mno=105530 [Access: October 16, 2021]. doi:10.24911/SJP.106-1588918898


AMA (American Medical Association) Style

Dhingr S, Juneja R. Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudan J Paed. 2021; 21(1): 67-75. doi:10.24911/SJP.106-1588918898



Vancouver/ICMJE Style

Dhingr S, Juneja R. Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudan J Paed. (2021), [cited October 16, 2021]; 21(1): 67-75. doi:10.24911/SJP.106-1588918898



Harvard Style

Dhingr, S. & Juneja, . R. (2021) Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudan J Paed, 21 (1), 67-75. doi:10.24911/SJP.106-1588918898



Turabian Style

Dhingr, Shruti, and Ruchika Juneja. 2021. Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudanese Journal of Paediatrics, 21 (1), 67-75. doi:10.24911/SJP.106-1588918898



Chicago Style

Dhingr, Shruti, and Ruchika Juneja. "Diverse clinical presentations of tubercular parotitis in children: case series with review of literature." Sudanese Journal of Paediatrics 21 (2021), 67-75. doi:10.24911/SJP.106-1588918898



MLA (The Modern Language Association) Style

Dhingr, Shruti, and Ruchika Juneja. "Diverse clinical presentations of tubercular parotitis in children: case series with review of literature." Sudanese Journal of Paediatrics 21.1 (2021), 67-75. Print. doi:10.24911/SJP.106-1588918898



APA (American Psychological Association) Style

Dhingr, S. & Juneja, . R. (2021) Diverse clinical presentations of tubercular parotitis in children: case series with review of literature. Sudanese Journal of Paediatrics, 21 (1), 67-75. doi:10.24911/SJP.106-1588918898





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