E-ISSN 1858-8360 | ISSN 0256-4408
 

Case Report 


SUDANESE JOURNAL OF PAEDIATRICS

2021; Vol 21, Issue No. 1

CASE REPORT

Infant with a parotid swelling: beware of the rare

Anam Siddiqui (1), Chandrika Azad (1), Dharmender Kumar Singh (2)

(1) Department of Pediatrics, Government Medical College and Hospital, Chandigarh, India

(2) Department of Radiodiagnosis, Government Medical College and Hospital, Chandigarh, India

Correspondence to:

Anam Siddiqui

Government Medical College and Hospital, Chandigarh,160030, India

Email: doll.anamsiddiqui786 [at] gmail.com

Received: 26 December 2019 | Accepted: 28 December 2020

How to cite this article:

Siddiqui A, Azad C, Singh DK. Infant with a parotid swelling: beware of the rare. Sudan J Paediatr. 2021;21(1):95–97.

https://doi.org/10.24911/SJP.106-1572965402


ABSTRACT

Salivary gland tumours are rare in children. We present the case of an infant who presented with a progressively increasing swelling of short duration in the parotid region accompanied by a small preauricular cutaneous haemangioma. Failing to comply with the commoner diagnoses like parotitis, help of imaging was taken that pointed towards the rarer diagnosis of parotid haemangioma. The child was started on oral propranolol and dramatic reduction in parotid and cutaneous swelling was seen after three months of therapy without any side effects.


KEYWORDS

Parotid Gland; Hemangioma; Propranolol; Child.


INTRODUCTION

Salivary gland tumours are rare in occurrence with only less than 5% of them occurring in paediatric population. Parotid haemangiomas constitute more than half of the salivary gland tumours in children [1]. Usual presentation is a rapidly growing swelling during early infancy followed by regression during latter half of first year [2]. The diagnosis can be made on clinical grounds and is confirmed with imaging. Magnetic resonance imaging (MRI) is the best imaging technique to diagnose and define the extent of parotid haemangioma [3]. Usually, treatment is required in cases with complications, functional impairment or for cosmetic reasons [4].


CASE REPORT

We present the case of a 2-month-old male infant who presented with a progressively increasing swelling in left pre auricular and infra-auricular area for 5 days (Figure 1A). There was no history of fever, lethargy, refusal to feed, discharge or bleed from the swelling, any rash, oral lesion or maternal breast infection. The child was born via preterm vaginal delivery, cried immediately after birth and his birth weight was appropriate for gestational age. The antenatal period was uneventful.

On examination, the swelling was 4 × 4 cm, non-tender, boggy and non-adherent to overlying or underlying structures. A small haemangioma 2 × 2 cm was present in left preauricular area since birth. At presentation, patient had normal vitals with no significant systemic findings. The initial blood investigations showed haemoglobin of 94 g/l, total leukocyte count of 11.6 × 109/l with 36% neutrophils, platelet count of 488 × 109/l, and C-reactive protein level of 68 mg/l. A possibility of parotitis was kept and intravenous antibiotics were given for 7 days. But, the swelling kept on increasing in size. The ultrasonography of the region revealed increased vascularity, thus MRI was done which showed hyperintense lesion on T2 images with intense post contrast enhancement on T1, pointing towards diagnosis of parotid haemangioma. Axial Short Tau Inversion Recovery (STIR) image revealed multiple flow voids representing intratumoral blood (Figure 2).

Figure 1. (A) Parotid swelling at admission (white arrow) and cutaneous haemangioma (yellow arrow). (B) After 3 months of propranolol therapy: regressed parotid swelling (white arrow) and cutaneous haemangioma (yellow arrow).

The child was started on oral propranolol at 2 mg/kg/day in two divided doses. The patient was followed up in person as well as telephonically during the treatment period and no abnormality was reported by the parents or treating physician in vitals, blood sugar levels, bowel habits or sleep cycle. After 3 months of therapy, superficial haemangioma faded in colour and lost its depth, and size of parotid decreased becoming almost invisible on gross examination (Figure 1B).

Figure 2. Axial STIR image demonstrates a hyperintense mass (open white arrow) in left parotid gland extending to deep lobe (solid white arrow) with multiple flow voids (small yellow arrows) representing intratumoral blood.


DISCUSSION

Infantile haemangioma (IH) is the commonest tumour of the parotid during infancy [1]. It is characterised by abnormal proliferation of endothelial cells and aberrant blood vessel architecture. Males are less commonly affected. The initial appearance is usually before 4 weeks of age and achieves full growth before first half of infancy. The involution starts around one year of age and is complete in next 3 years. The superficial infantile haemangiomas (IHs) tend to appear as well as involute earlier than their deep counterparts. They are further classified as focal, multifocal or segmental [2]. The index case had a focal haemangioma involving the parotid gland which presented as a rapidly growing parotid swelling at 2 months of age. It was also associated with a cutaneous haemangioma, which served as the supportive evidence in this case. Female gender, preterm birth, low birth weight, multiple gestations, maternal vaginal bleeding during the first trimester, progesterone use, preeclampsia, older maternal age, placenta previa, use of in vitro fertilisation and positive family history have all been linked to the development of IH [5]. In the index case, preterm birth can be linked as a risk factor. MRI proved to be superior to ultrasound in terms of defining size, extent in depth and relationship with adjacent structures and guided to the conclusive diagnosis.

Rapid growth of the mass may act as a harbinger of local and systemic complications which depend upon the location, size and type of IH. Some of the complications are ulceration, bleeding, visual impairment, auditory impairment, congestive heart failure and airway obstruction which are more likely to occur in case of segmental and facial haemangiomas. Treatment is indicated for these complications, cosmetic defects and imminent functional impairment [2]. In the index case, there were no associated complications or functional impairment and treatment was sought solely for cosmetic reasons. Earlier corticosteroids were used as first line therapy, but owing to the serious side effects and rebound phenomenon they are not used much anymore [6]. Propranolol is now the preferred first line agent, it acts via vasoconstriction, inhibition of angiogenesis and stimulation of apoptosis [7]. It is administered orally at a dose of 1 to 3.4 mg/kg/day.The potential side effects of propranolol therapy are sinus bradycardia, hypotension, hypoglycaemia, seizures, reactive airways, sleep disturbance, diarrhoea and cold extremities [4]. Other treatment options are topical timolol and intralesional/topical corticosteroids but their dosing and safety profile is not much studied. Laser treatment of IHs can be tried in early, nonproliferating and superficial lesions [2]. The cases with failure of/contraindication to pharmacotherapy or favourable anatomical location might require surgical resection [8]. In the index case, propranolol was started at a dose of 2 mg/kg/day and no side effects were experienced [4]. On follow up, patient thrived well with no long-term side effects of therapy, rebound increase in size of old lesions or appearance of new lesions, highlighting the fact that propranolol therapy proved to be safe and effective in this case.


CONCLUSION

Amidst a plethora of infectious conditions which are more common, especially in tropical areas, clinicians need to be aware of parotid haemangioma as an important differential diagnosis of swelling in this area to avoid diagnostic and treatment errors and complications.


ACKNOWLEDGEMENTS

The authors would like to thank the treating team of the Pediatrics Department.


CONFLICT OF INTEREST

The authors declare that they have no conflicts of interest.


FUNDING

Nil.

Ethical approval

Signed informed consent for participation and publication of medical details was obtained from the parents of this child. Confidentiality of patient’s data was ensured at all stages. The authors declare that ethics committee approval was not required for this case report.


REFERENCES

  1. Ellies M, Schaffranietz F, Arglebe C, Laskawi R. Tumors of the salivary glands in childhood and adolescence. J Oral Maxillofac Surg. 2006;64:1049–58. https://doi.org/10.1016/j.joms.2006.03.006
  2. Darrow DH, Greene AK, Mancini AJ, Nopper AJ, the section on dermatology SOO-H &amp, Neck Surgery AS on PS. Diagnosis and management of infantile hemangioma: executive summary. Pediatrics. 2015;136:786–91. https://doi.org/10.1542/peds.2015-2482
  3. Roebuck DJ, Ahuja AT. Hemangioendothelioma of the parotid gland in infants: sonography and correlative MR imaging. Am J Neuroradiol. 2000;21:219–23.
  4. Schiestl C, Neuhaus K, Zoller S, et al.Efficacy and safety of propranolol asfirst-line treatment for infantilehemangiomas. Eur J Pediatr. 2011;170:493–501. https://doi.org/10.1007/s00431-010-1324-2
  5. Smith CJF, Friedlander SF, Guma M, Kavanaugh A, Chambers CD. Infantile hemangiomas: an updated review on risk factors, pathogenesis, and treatment. Birth Defects Res. 2017;109:809–15. https://doi.org/10.1002/bdr2.1023
  6. George ME, Sharma V, Jacobson J,Simon S, Nopper AJ. Adverse effects of systemic glucocorticosteroid therapy in infants with hemangiomas. Arch Dermatol. 2004;140:963–9. https://doi.org/10.1001/archderm.140.8.963
  7. Ji Y, Chen S, Xu C, Li L, Xiang B. The use of propranolol in the treatment of infantile haemangiomas: an update on potential mechanisms of action. Br J Dermatol. 2015;172:24–326. https://doi.org/10.1111/bjd.13388
  8. Soltani AM, Reinisch JF. Algorithmicapproach to the management ofhemangiomas. J Craniofac Surg. 2011;22:585–8. https://doi.org/10.1097/SCS.0b013e31820873ac


How to Cite this Article
Pubmed Style

Siddiqui A, Azad C, Singh DK. Infant with a parotid swelling: beware of the rare. Sudan J Paed. 2021; 21(1): 95-97. doi:10.24911/SJP.106-1572965402


Web Style

Siddiqui A, Azad C, Singh DK. Infant with a parotid swelling: beware of the rare. http://www.sudanjp.com/?mno=72514 [Access: October 16, 2021]. doi:10.24911/SJP.106-1572965402


AMA (American Medical Association) Style

Siddiqui A, Azad C, Singh DK. Infant with a parotid swelling: beware of the rare. Sudan J Paed. 2021; 21(1): 95-97. doi:10.24911/SJP.106-1572965402



Vancouver/ICMJE Style

Siddiqui A, Azad C, Singh DK. Infant with a parotid swelling: beware of the rare. Sudan J Paed. (2021), [cited October 16, 2021]; 21(1): 95-97. doi:10.24911/SJP.106-1572965402



Harvard Style

Siddiqui, A., Azad, . C. & Singh, . D. K. (2021) Infant with a parotid swelling: beware of the rare. Sudan J Paed, 21 (1), 95-97. doi:10.24911/SJP.106-1572965402



Turabian Style

Siddiqui, Anam, Chandrika Azad, and Dharmender Kumar Singh. 2021. Infant with a parotid swelling: beware of the rare. Sudanese Journal of Paediatrics, 21 (1), 95-97. doi:10.24911/SJP.106-1572965402



Chicago Style

Siddiqui, Anam, Chandrika Azad, and Dharmender Kumar Singh. "Infant with a parotid swelling: beware of the rare." Sudanese Journal of Paediatrics 21 (2021), 95-97. doi:10.24911/SJP.106-1572965402



MLA (The Modern Language Association) Style

Siddiqui, Anam, Chandrika Azad, and Dharmender Kumar Singh. "Infant with a parotid swelling: beware of the rare." Sudanese Journal of Paediatrics 21.1 (2021), 95-97. Print. doi:10.24911/SJP.106-1572965402



APA (American Psychological Association) Style

Siddiqui, A., Azad, . C. & Singh, . D. K. (2021) Infant with a parotid swelling: beware of the rare. Sudanese Journal of Paediatrics, 21 (1), 95-97. doi:10.24911/SJP.106-1572965402





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