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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 4  |  Page : 254-256

Tubercular sinonasal mass: A rare cause of recurrent epistaxis


1 Department of Paediatrics, Indian Naval Hospital Ship Asvini; Mumbai, Maharashtra, India
2 Department of ENT, Indian Naval Hospital Ship Asvini; Mumbai, Maharashtra, India

Date of Submission20-Jun-2021
Date of Decision13-Oct-2021
Date of Acceptance06-Nov-2021
Date of Web Publication29-Nov-2021

Correspondence Address:
Dr. Hitesh Daryani
Department of Paediatrics, Indian Naval Hospital Ship Asvini; Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ipcares.ipcares_197_21

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  Abstract 

Background: Epistaxis is common in children. Almost 60% experience at least one episode by 10 years of age. Most are benign and self-resolving. Episodes that are severe, frequent, take longer to resolve, or unilateral should be investigated. Although extrapulmonary tuberculosis (TB) is common in children, presentation as a sinonasal mass is rare. Clinical Description: A 4-year-old girl presented with a history of recurrent, left-sided epistaxis for 2 months, with diffuse ipsilateral facial swelling for 2 weeks. There was a history of preceding oral swelling and weight loss. Nasal endoscopy revealed a friable left nasal mass. Imaging delineated an infiltrative, highly cellular lesion in the left maxillary sinus, infiltrating surrounding bone and extending into the left nasal cavity, jaw, and orbital floor. Management: Endoscopic biopsy was suggestive of noncaseating granulomatous lesion. Langerhans cell histiocytosis, granulomatosis with polyangiitis and microscopic polyangiitis, and malignancy were ruled out. Nucleic acid amplification tests and culture established microbiological diagnosis of primary sinonasal TB. Initiation of antitubercular therapy led to complete recovery. Conclusion: The diagnosis of sinonasal TB is challenging and requires multidisciplinary collaboration. Its rarity and nonspecific presentation requires a high index of suspicion by the treating team. Clinical, histopathological, and microbiological criteria should be used to establish a diagnosis. Primary sinonasal TB is a rare entity in young immunocompetent children.

Keywords: Epistaxis, extrapulmonary tuberculosis, sinonasal mass


How to cite this article:
Shankar S, Gautam S, Daryani H, Naga R. Tubercular sinonasal mass: A rare cause of recurrent epistaxis. Indian Pediatr Case Rep 2021;1:254-6

How to cite this URL:
Shankar S, Gautam S, Daryani H, Naga R. Tubercular sinonasal mass: A rare cause of recurrent epistaxis. Indian Pediatr Case Rep [serial online] 2021 [cited 2022 Jan 20];1:254-6. Available from: http://www.ipcares.org/text.asp?2021/1/4/254/331370

Around 18 lakh cases of tuberculosis (TB) were notified in India in 2020, and out of these, around 5.7% were in children under 15 years of age.[1] Pulmonary TB is the most common presentation in children, while extrapulmonary TB occurs in 25%–35% of cases.[2] Among the latter, lymph nodes are the most common site. Epistaxis is common in children, with almost 60% of children experiencing at least one nasal bleed by 10 years of age.[3] The vast majority are benign, self-resolving, and do not require medical workup or intervention. Local causes such as trauma or mucosal irritation should be looked for. However, episodes that are more severe, frequent, take a longer time to resolve, or are unilateral should be investigated to exclude bleeding disorders, or pathological conditions such as tumors, granulomatous disorders, and vascular causes.

Nasal or sinonasal TB presenting as recurrent epistaxis has been described in children in scientific literature, but to the best of our knowledge, only 15 cases have been reported in the past two decades. Most are secondary, and primary sinonasal TB is rare.[4] We present a 4-year-old child with recurrent epistaxis due to primary sinonasal TB to increase sensitize about this rare entity among clinicians.


  Clinical Description Top


A 4-year-old immunized girl presented with recurrent, self-limiting episodes of nosebleeds from the left nostril for 2 months. The bleeding occurred spontaneously, was intermittent (every 7–10 days), of scant amount (around 10–15 drops per episode), increased on crying and bending forward, and got relieved by applying digital pressure on the nostrils. The child did not have a habit of picking her nose. There was no history of bleeding from other sites, easy bruising, bluish skin lesions, black stools, or progressive pallor. The episodes were not associated with fever or recurrent upper respiratory tract infections, symptoms suggestive of allergic rhinitis (excessive sneezing, nasal obstruction, or congestion), preceding nasal trauma, or use of nasal medications. Her parents had also observed a progressively increasing, diffuse, painless swelling over the left cheek for 2 weeks. It was insidious in onset, and associated with brown discoloration of the skin below her left eye. There was a history of significant weight loss; she had lost one kilogram (7%) over the past 3 months. There was a significant past history of having an oral swelling in the left upper jaw 6 months back, which was diagnosed as gingivitis by a dentist, and treated with oral antibiotics. There was no history of any contact with a case of TB. There was no history of any previous hospital admission. The family history was not contributory.

Management and outcome

On examination, the girl was hemodynamically stable and normotensive, with appropriate anthropometric parameters for age. Pallor was present. She had a diffuse, nontender, left maxillary swelling that resulted in a narrowed appearance of the left palpebral fissure [[Figure 1], refer to cover page]. There was no significant lymphadenopathy, petechiae, or mucosal bleeds. Anterior rhinoscopy revealed reduced air blast and mucoid discharge on the left, with no active bleeding. The right-sided nasal cavity appeared normal. On direct nasal endoscopy, a diffuse, friable mass that bled on touch was seen emerging from the left maxillary ostium, completely blocking the left osteomeatal complex, and filling the middle meatus and left nasal cavity [Figure 2]. The medial wall of the maxilla bulged inside the nasal cavity causing medialization of the middle turbinate. No mass was seen in the nasopharynx and the nasal septum was in midline. Bulging of the gums was noted behind the left upper molars on examination of the oral cavity. The ophthalmological and systemic examination was unremarkable.
Figure 1: Reduced left vertical palpebral fissure due to diffuse nontender left maxillary swelling

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Figure 2: A diffuse, firm, friable mass coming from the left maxillary ostium, completely blocking left osteomeatal complex, filling middle meatus and left nasal cavity, on direct nasal endoscopy

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On preliminary investigation, she was found to have hemoglobin of 6.6 g/dl, total leukocyte count of 11,400/mm3, platelet count of 4.8 lakhs/mm3, and microcytic hypochromic anemia. The coagulation profile was also normal. Computed tomography scan of the head and neck revealed a large soft tissue mass, completely opacifying the left maxillary sinus and destroying the surrounding bones. The mass was found to extend into the nasal cavity, with associated mucosal thickening and opacification of the ipsilateral ethmoid air cells. The radiological differentials given were a neoplasm or Langerhans cell histiocytosis (LCH). MRI of the paranasal sinuses was planned for a more detailed anatomical delineation. This showed an expansile, infiltrative, highly cellular lesion epicentered in the left maxillary sinus that extended into the left nasal cavity and infiltrated the maxillary bone (anteriorly), left ethmoid sinus (superomedially), and soft tissue in the left maxillary alveolar process (infero-anteriorly). The lesion encased the left upper premolars and canine causing bulging on the left orbital floor, and also encased the left infraorbital vessels, sparing the lamina papyracea, nasal septum, and orbital contents [Figure 3].
Figure 3: T1- and T2-weighted coronal magnetic resonance images showing ill-defined expansile lesion involving (L) maxillary sinus and adjacent structure, with no involvement of infraorbital region or intracranial extension

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Endoscopic tissue biopsy from the left maxillary mass was performed under general anesthesia. This revealed noncaseating granulomas composed of histiocytes and multinucleated giant cells with entrapped bony fragments. Ziehl–Neelsen (ZN), Periodic acid–Schiff (PAS), and Grocott methenamine silver stains did not reveal any organisms. No lytic lesions were found in radiographs of the skull, ribs, vertebrae, or long bones. Immunohistochemistry performed to rule out LCH was positive for CD68 (histiocytes) and negative for S100 and CD1a. Serum angiotensin-converting enzyme levels were normal (45 U/L). Antineutrophil cytoplasmic antibodies (ANCA) – diffuse cytoplasmic (c-ANCA) and perinuclear (p-ANCA) – were negative by immunofluorescence assay, thus ruling out granulomatosis with polyangiitis and microscopic polyangiitis. The tuberculin skin test was strongly positive (27 mm induration at 48 h), but the chest radiograph and abdominal ultrasound were normal. An endoscopic excision biopsy of the lesion was undertaken but did not reveal anything different. However, the Xpert MTB/RIF Ultra for Mycobacterium tuberculosis was positive on the endoscopic tissue biopsy specimen (and incidentally also on the gastric aspirate), sensitive to rifampicin. Rapid mycobacterium culture (automated fluorescent MALDI-TOF-MS) grew M. tuberculosis complex, sensitive to first-line antitubercular treatment (ATT). HIV serology was nonreactive, and blood sugar profile was normal.

The final diagnosis was extrapulmonary primary sinonasal TB. ATT was initiated,[3] on which there was a gradual resolution of the facial and nasal swelling, with cessation of epistaxis within a month. After completing 6 months of ATT, the child was thriving with resolution of symptoms, and adequate weight gain.


  Discussion Top


Sinonasal tumors are rare in children. They may be congenital (i.e., glioma and teratoma), benign (i.e., polyp, papilloma, hemangioma, or leiomyomas), or malignant (non-Hodgkin's lymphoma, rhabdomyosarcoma, or squamous cell carcinoma). Most malignancies present as rapidly growing nasal masses that manifest with recurrent epistaxis, deformities of the nose and cheek, weight loss, and constitutional symptoms.

Sinonasal TB presents with features of rhinosinusitis and has three subtypes: i) mucosal (the most common type) – presenting as a mucosal polyp with scanty purulent discharge; ii) bony – leading to fistula formation and development of a mid-facial defect; and iii) hyperplastic – causing granuloma formation and mimicking malignancy.[5],[6] Our patient had overlapping features of both bony and hyperplastic subtypes. An exhaustive literature search identified 13 cases of nasal TB in children from 2000 onward, the details of which are given in [Table 1]. Only two cases of sinonasal TB were identified in addition to this one: a 15-year-old girl with a proliferative maxillary mass[7] and a 5-year-old boy presenting with sinusitis.[8]
Table 1: Cases of nasal tuberculosis cases in children (circa 2000 onward)

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The diagnosis of sinonasal TB is challenging. Its rarity and nonspecific presentation requires a high index of suspicion. Beltran et al.[9] proposed the following diagnostic criterion: (i) absence of clinical response to antibiotics; (ii) caseous granulomatosis on histopathology; and iii) identification of M. tuberculosis by polymerase chain reaction (PCR) assay, confirmation by culture, and response to ATT. Histologically, both caseating and noncaseating granulomas have been described. In our case, the granuloma was noncaseating and ZN staining was negative, but PCR assay was positive, the culture was confirmatory, and there was good response to medical management. Surgical excision and reconstruction are rarely required in cases with severe disfigurement.



Declaration of patient consent

The authors certify that they have obtained the appropriate consent from the parent. The legal guardian has given his consent for the images and other clinical information to be reported in the journal. The guardian understands that the name and initials will not be published, and due efforts have been made to conceal the same, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Central TB Division, Ministry of Health and Family Welfare. India TB Report 2021. New Delhi: JK Offset Pvt. Ltd.; 2021. p. 352.  Back to cited text no. 1
    
2.
Cameron LH, Starke JR. Tuberculosis (Mycobacterium tuberculosis). In: Kliegman RM, St Geme JW, editors. Nelson Textbook of Pediatrics. 21st ed. Canada: Elsevier; 2020. P 1565-81.  Back to cited text no. 2
    
3.
Misra A, Basu A, Mandal PK, et al. Management of pediatric epistaxis in different age group in a tertiary care centre. Int J Contemp Pediatr 2016;3:1206-9.  Back to cited text no. 3
    
4.
Kim KY, Bae JH, Park JS, et al. Primary sinonasal tuberculosis confined to the unilateral maxillary sinus. Int J Clin Exp Pathol 2014;7:815-8.  Back to cited text no. 4
    
5.
Malik JN, Jan S, Monga S, et al. Sinonasal tuberculosis: Report of three atypical cases. Indian J Tuberc 2016;63:268-72.  Back to cited text no. 5
    
6.
Bansal R, Jain A, Mittal S. Orofacial tuberculosis: Clinical manifestations, diagnosis and management. J Family Med Prim Care 2015;4:335-41.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Jha D, Deka RC, Sharma MC. Tuberculosis of the maxillary sinus manifesting as a facial abscess. Ear Nose Throat J 2002;81:102-4.  Back to cited text no. 7
    
8.
Khan S, Pujani M, Jetley S. Primary nasal tuberculosis: Resurgence or coincidence – A report of four cases with review of literature. J Lab Physicians 2017;9:26-30.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Beltran S, Douadi Y, Lescure FX, et al. A case of tuberculous sinusitis without concomitant pulmonary disease. Eur J Clin Microbiol Infect Dis 2003;22:49-50.  Back to cited text no. 9
    


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