Oropharyngeal Mucocele as an Unusual Cause of Recurrent Pneumonia in a Young Infant

Amrita Banerjee; Dipyaman Ghosh

doi:10.4103/ipcares.ipcares_298_22

CASE REPORT

Year : 2023 | Volume : 3 | Issue : 2 | Page : 110-112

Oropharyngeal Mucocele as an Unusual Cause of Recurrent Pneumonia in a Young Infant

Amrita Banerjee, Dipyaman Ghosh
Department of Pediatrics, AIIMS, Patna, Bihar, India

Date of Submission	30-Dec-2022
Date of Decision	06-Apr-2023
Date of Acceptance	07-Apr-2023
Date of Web Publication	24-May-2023

Correspondence Address:
Dr. Dipyaman Ghosh
13, Kankulia Road, Kolkata - 700 029, West Bengal
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ipcares.ipcares_298_22

Abstract

Background: Oral mucoceles, though frequently seen as asymptomatic lesions in young adults, very little is known about their existence in infants and the complications arising out of them in this age group. Clinical Description: A 4-month-old infant presented with a history of recurrent episodes of breathing difficulty since 15 days of life, every episode being managed as aspiration pneumonia. The child presented in the fifth such episode with tachypnea tachycardia and chest indrawing. Auscultation revealed bilateral crepitations and chest X-ray showed diffuse haziness in the bilateral chest predominantly in the right middle lobe, rest of the systemic examination being normal. Oral cavity examination showed a bluish globular bulge behind the tongue with surrounding congested mucosa and thick secretions. Management and Outcome: The baby was managed with respiratory support in the form of continuous positive airway pressure, intravenous antibiotics Piperacillin-Tazobactam, amikacin and metronidazole, and anti-reflux measures of oral Domperidone. Flexible laryngoscopy showed a cystic lesion behind the tongue and contrast-enhanced computerized tomography confirmed a hypodense lesion arising from the uvula suggestive of a mucocele. Excision of the oro-pharyngeal cystic lesion was done and the histopathological examination confirmed it to be a mucocele. The baby was gradually weaned off oxygen support and proper breastfeeding was re-established. Conclusion: This case creates awareness regarding the fact that an unsuspected oral mucocele may be the cause of recurrent respiratory symptoms in an infant, thereby reiterating the importance of oral examination even in young infants.

Keywords: Aspiration, recurrent pneumonia, respiratory distress, retention cyst, salivary gland

How to cite this article:
Banerjee A, Ghosh D. Oropharyngeal Mucocele as an Unusual Cause of Recurrent Pneumonia in a Young Infant. Indian Pediatr Case Rep 2023;3:110-2

How to cite this URL:
Banerjee A, Ghosh D. Oropharyngeal Mucocele as an Unusual Cause of Recurrent Pneumonia in a Young Infant. Indian Pediatr Case Rep [serial online] 2023 [cited 2023 Jun 3];3:110-2. Available from: http://www.ipcares.org/text.asp?2023/3/2/110/377519

Oral mucoceles, one of the commonest benign lesions of the minor salivary glands, develop from accumulation of mucoid secretions inside or around the gland or its duct. They are seen mostly in young adults, as a result of mechanical trauma to the excretory duct of the gland.^[1],[2]

However, the presentation of oral mucocele in a young infant is very uncommon. We report an interesting case of a young infant presenting with recurrent pneumonia due to an unsuspected oral mucocele.

Clinical Description

A 4-month-old boy, firstborn child to a nonconsanguineously married couple, presented with fever and fast breathing since the past 7–8 days, associated with vomiting, lethargy, poor feeding, without any decreased urine output. Baby was not accepting feeds well.

The baby had a history of recurrent such episodes of fever and fast breathing noticed since 15 days of life. After an apparently asymptomatic initial 15 days of life, the mother noticed multiple episodes of regurgitation of milk from mouth and nose after breastfeeding. Subsequently, the baby developed fever, fast breathing, and difficulty in breathing for which he was taken to a local practitioner and was treated as an episode of pneumonia. The baby recovered over 5 days. The baby continued to have some feeding difficulties despite the subsidence of the acute episode. In the next 3 months, the child developed another three similar episodes, all treated as pneumonia and attributed to faulty feeding techniques leading to aspiration and pneumonia. When the baby developed fifth such episode at 4 months of life, he presented to us with fever for 7 days, fast and labored breathing for 4–5 days, associated with vomiting, lethargy, and poor feeding. Mother also noticed worsening of respiratory distress in the baby on supine position but improvement in lateral position. There was no history of stridor, cyanosis, feeding diaphoresis, or swelling over body. There was no history of any recurrent loose stools, ear discharge, skin infections like boils, pustules, etc., There was no significant family history, nor any contact with tuberculosis.

The baby was born at full term through a normal vaginal delivery with a birth weight of 3 kg and was on exclusive breastfeeding. He had received vaccines as per schedule at birth and at 6 weeks. The antenatal and natal periods were uneventful.

On examination, the baby was conscious, alert, with a weight of 4.2 kg (<3^rd centile), height 64 cm (50^th–75^th centile), and head circumference 40 cm (15^th–50^th centile). Baby was febrile with a temperature of 100.7°F, heart rate of 154/min, respiratort rate of 74/min. There was no pallor, cyanosis or icterus. Oral examination revealed a congested oral cavity with thick secretions in posterior pharyngeal wall. A 3 cm × 3 cm globular bluish bulge was seen in the posterior pharyngeal region. Possible differential diagnosis of such globular lesions was considered at this time including hemangioma, lipoma, teratoma, thyroglossal cyst or an oral mucocele. There was no history of oral trauma, or premature erupted teeth or any midline cranial defector cleft palate. Chest examination revealed bilateral crepitations. Rest of the systemic examination was normal.

Management and Outcome

The infant was hospitalized and provided respiratory support in the form of continuous positive airway pressure with oxygen at 5l/min along with intravenous antibiotics (Piperacillin-Tazobactam, amikacin, metronidazole) with anti-reflux measures (oral domperidone). Investigations showed Hemoglobin 10.6 g/dL, TLC 24730/uL, N 65%, L 31%, C-reactive protein 360.3 mg/L, urea 14.2 mg/dL, creatinine 0.15 mg/dL, serum bilirubin 0.44 mg/dL (T), 0.30 mg/dL (U), aspartate transaminase 49.3U/L, and alanine transaminase 31.3 U/L. Blood culture was sterile. Chest X-ray showed diffuse haziness in bilateral chest, predominantly involving right middle lobe.

Flexible laryngoscopy confirmed a globular bluish cystic lesion with vessels over the lesion arising from the left pharyngeal wall behind the base of the tongue; hence, differentials were narrowed down to thyroglossal cyst or an oral mucocele [Figure 1]. A Contrast-enhanced computed tomography (CT) skull base-mediastinum was done, which revealed a well-defined nonenhancing hypodense cystic lesion in posterior part of tongue bulging into oropharynx, possibly arising from uvula of soft palate suggesting mucus retention cyst or an oral mucocele [Figure 2].

Figure 1: Flexible laryngoscopy showing a cystic mass originating from pharyngeal wall behind base of tongue

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Figure 2: CECT skull base showed a well-defined non-enhancing hypodense cystic mass arising from uvula of the soft palate bulging into oropharynx. CECT: Contrast-enhanced computed tomography

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The baby underwent surgery under general anesthesia with excision of the oropharyngeal cyst. Tissue histopathology showed fibro collagenous cyst wall lined by stratified epithelium suggestive of mucus retention cyst or an oral mucocele. The baby was successfully extubated postprocedure and oxygen support weaned off and breast feeding was re-established. The baby was discharged in stable condition. During follow-up after 6 weeks, there had been no such episode of respiratory difficulty since discharge, requiring emergency management.

Discussion

The baby described above, presented with recurrent episodes of fast breathing with mild fever, subsiding every time with symptomatic treatment. Such a clinical presentation is commonly encountered by pediatricians in routine practice, but an oral examination is seldom done especially in an infant as young as 2–4 months. The identification of an oral mucocele and its successful removal helped in subsidence of symptoms without further recurrences.

Though common in the second decade of life, oral mucoceles are an uncommon occurrence in young infants. An incidence of 2.5 per 1000 individuals is reported in adults in the second decade of life. Clinically, they are characterized by single or multiple, painless, soft, smooth, spherical, translucent, fluctuant nodules, which usually occur on lower lip (70%) and are mostly asymptomatic.^[3] Spontaneous resolution occurs in 44% of cases after an average of 3 months.^[4]

Pathologically, oral mucoceles are classified as extravasation or retention type.^[5] The extravasation type is a pseudocyst without defined walls and is not lined by epithelium. They are caused due to mechanical trauma to the excretory duct of the gland leading to transection or rupture, with consequent extravasation of mucin into the connective tissue stroma. They are seen frequently on lower labial mucosa, buccal mucosa, and retromolar area.^[6] The retention type is less common than extravasation, usually affects older individuals and is seen frequently on the upper lip, hard palate, the floor of the mouth, and maxillary sinus.^[7] In mucous retention phenomena, mucus may be retained in the duct and/or acini as a result of duct obstruction by sialolith or strictures.

While most of the time, oral mucoceles are asymptomatic; an unusual site and an early age of presentation as in our case may lead to atypical manifestations. Depending on their size and location, oral mucoceles can rarely cause significant presentations in the form of, airway blockade, recurrent aspirations and pneumonia, interference with speech, and interference in mastication and swallowing.^[8],[9]

A thorough history taking and examination of the lesion is crucial for diagnosing oral mucoceles correctly. Some identifying points for a mucocele include its characteristic location, history of trauma, rapid appearance, variations in size, bluish color, and the consistency. For specific cases, the diagnosis may require routine radiographs, ultrasonography, or advanced diagnostic methods-CT and magnetic resonance imaging for better visualizing the form, diameter, position and determination of the lesion origin.^[2]

The treatment for oral mucoceles shall be either complete excision, marsupialization, dissection, cryosurgery, carbon dioxide lasers, electrocautery, intra-lesional injection of sclerosing agent OK-432 or steroid injection.^[8] In our case, the lesion was a typical a retention type of mucocele and was considered to be due to the congenital dysplasia of excretory duct which is known to re-occur. Thus, surgical excision was preferred to prevent any further recurrences.

Conclusion

There is a paucity of data on mucoceles in young infants and peculiarities of presentation in this age group. We report this case to highlight the same and emphasize the importance of a careful oral examination for early diagnosis and intervention in such cases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	More CB, Bhavsar K, Varma S, et al. Oral mucocele: A clinical and histopathological study. J Oral Maxillofac Pathol 2014;18:S72-7.
2.	Jani DR, Chawda J, Sundaragiri SK, et al. Mucocele – A study of 36 cases. Indian J Dent Res 2010;21:337-40. [PUBMED] [Full text]
3.	Khan A, Anwar, Azizah A, et al. Cases of mucocele treated in the dental department of Penang Hospital. Arch Orofacial Sci 2008;3:7-10.
4.	Syebele K. Regression of both oral mucocele and parotid swellings, following antiretroviral therapy. Int J Pediatr Otorhinolaryngol 2010;74:89-92.
5.	Garcia Leon N, Marrugo Pardo GE. Mucocele of the glands of Blandin-Nuhn: A case report. Colomb Med (Cali) 2013;44:46-7.
6.	Ata Ali J, Carrillo C, Bonet C, et al. Oral mucocele: Review of the literature. J Clin Exp Dent 2010;2:e18-21.
7.	Re Cecconi D, Achilli A, Tarozzi M, et al. Mucoceles of the oral cavity: A large case series (1994-2008) and a literature review. Med Oral Patol Oral Cir Bucal 2010;15:e551-6.
8.	Nico MM, Park JH, Lourenço SV. Mucocele in pediatric patients: Analysis of 36 children. Pediatr Dermatol 2008;25:308-11.
9.	Mouravas V, Sfoungaris D, Papageorgiou I, et al. Mucoceles of the lesser salivary glands in neonates demonstrate a particular clinicopathological pattern and mandate urgent management. J Stomatol Oral Maxillofac Surg 2018;119:238-41.