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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 5  |  Issue : 1  |  Page : 29-31

A case of cystic hygroma in adults and its management


Department of General Surgery, Dr. Mehta Hospital Global Campus, Chennai, Tamil Nadu, India

Date of Submission09-Jan-2021
Date of Acceptance29-Jan-2021
Date of Web Publication03-Jul-2021

Correspondence Address:
Dr. Pandiaraja Jayabal
26/1, Kaveri Street, Rajaji Nagar, Villivakkam, Chennai - 600 049, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/aiao.aiao_14_20

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  Abstract 


Cystic hygroma is a congenital abnormality due to lymphoid sequestration in the sac. Incidence of cystic hygroma in adult is rarely report. It is most commonly present in posterior triangle as cystic mass. Even in adult patient, any cystic lesion present in posterior triangle cystic hygroma should be considered one of the differential diagnoses. Here, we present a rare occurrence of cystic hygroma in adult male patient present as cystic lesion. This case is reported for its adult presentation and discusses the management for cystic hygroma.

Keywords: Adult presentation, cystic hygroma, lymphoid sequestration, management of cystic hygroma


How to cite this article:
Jayabal P. A case of cystic hygroma in adults and its management. Ann Indian Acad Otorhinolaryngol Head Neck Surg 2021;5:29-31

How to cite this URL:
Jayabal P. A case of cystic hygroma in adults and its management. Ann Indian Acad Otorhinolaryngol Head Neck Surg [serial online] 2021 [cited 2021 Dec 4];5:29-31. Available from: https://www.aiaohns.in/text.asp?2021/5/1/29/320570




  Introduction Top


Cystic hygroma is otherwise known as cystic lymphangioma or water tumor. It is a benign congenital tumor due to the congenital malformation of lymphatic system. It is due to failure of fusion of lymphatic drainage system to the venous communication system leading to lymphoid sequestration in the sac. It usually affects infant and children <2 years. It can rarely present in adult. However, cystic hygroma should be considered one of the differential diagnoses of adult cystic neck mass. It is most commonly present in the posterior triangle as cystic mass.


  Case Report Top


A 24-year-old male presented with a swelling and pain on the left side of the neck for the past 6 months. The pain is mild to moderate in severity, dull aching in nature and nonradiating. There is no history of breathing difficulty, difficulty in swallowing, fever, and purulent discharge. He denies any history of local trauma. On examination, a solitary swelling of 4 cm × 3 cm present on the left submandibular region. The swelling is soft and nontender. It has well-defined margin with smooth surface and the skin overlying the swelling is normal [Figure 1].
Figure 1: A solitary swelling of 4 cm × 3 cm present on the left submandibular region

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The routine blood investigations are normal and the computed tomography (CT) shows a cystic swelling in the left submandibular region with a suggestive of a retention cyst [Figure 2]. Fine-needle aspiration cytology of the lesion is inconclusive.
Figure 2: The computed tomography shows a cystic swelling in the left submandibular region with a suggestive of a retention cyst

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He is posted for excision under general anaesthesia. The intraoperative findings shows superficial cystic swelling with few loculi over the muscle layer of the mylohyoid and the anterior belly of the digastric with few finger like extensions between the muscle planes [Figure 3]. The swelling is excised in toto and the histopathology shows multiple lymph spaces lined by lymphatic epithelium with a diagnosis of cystic hygroma of the neck [Figure 4].
Figure 3: The intraoperative findings shows superficial cystic swelling with few loculi over the muscle layer of the mylohyoid and the anterior belly of the digastric with few finger like extensions between the muscle planes

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Figure 4: Histopathology shows multiple lymph spaces lined by lymphatic epithelium with a diagnosis of cystic hygroma of the neck (H and E, ×200)

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Post excision biopsy, the patient recovered well and patient is on follow-up for more than 2 years without any recurrence.


  Discussion Top


Cystic hygroma is a benign congenital tumor due to abnormal proliferation of lymphoid tissue sequestrated by sacs.[1] It usually affect infant and children <2 years. It can rarely present in adult.[2] The occurrence of cystic hygroma mostly in cervicofacial (75%) followed by axilla (20%), but there are reports of other location also. It can also occur in mediastinum, abdomen, retroperitoneum, mesentery, tongue, oral cavity, groin, orbit, and solid internal organs.[3]

Cystic hygromas are result of the sequestration of lymph sacs.[4] It has a clinical and histological classification. Based on clinical findings, it is classified as (a) macro cystic-hygroma under the mylohyoid muscle with in the cervical triangle, (b) micro cystic-hygroma above the mylohyoid muscle, with in the oral cavity, and (c) mixed. Based on histology, it is classified as (i) capillary lymphangioma, (ii) cavernous lymphangioma, and (iii) cystic lymphangioma [Table 1].
Table 1: Classification of cystic hygroma

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Cystic hygroma usually start manifesting after 2 years, but there are reports of adult presentation also. Patient present to health-care team for cosmetic reasons, but it can also present with bleeding, purulent discharge, infections, airway difficulty (cervical), numbness in upper limb (axillary), abdominal pain and distension (abdomen), chest discomfort (mediastinum), and loss of vision (orbit). The adult presentation may be triggered by direct trauma or following oropharyngeal infection.

The imaging investigations such as ultrasound, CT, magnetic resonance imaging helps us for tissue delineation with surrounding structures. It helps for proper preoperative planning of surgery. Cystic hygroma in ultrasound appear as hypoechoic lesion, CT Scan reveals a homogenous cystic lesion.[5] The role of fine-needle aspiration cytology in cystic hygroma is controversial. It is only helpful for working out differential diagnosis of neck mass. On histology cystic hygroma appear as benign proliferation of lymphatic tissue along with fibrous tissue.

Surgical excision remains the standard of care in cystic hygroma. Other modalities such as cryotherapy, sclerotherapy, and radiotherapy can have mixed results. The treatment includes depending on the site and extent a complete surgical excision is the treatment of choice but can be technically difficult with a recurrence rate as high as 20%.[6] The other nonsurgical interventions are radiofrequency ablation, CO2 laser, embolization, radiotherapy, steroid administration and sclerotherapy with OK-432, bleomycin, doxycycline, cyclophosphamide, alcohol, and sirolimus.[7]


  Conclusion Top


Cystic hygromas are benign lesions arising due an abnormality of lymphoid development and rarely present in adults. Proper preoperative imaging and correct plan of management will reduce the recurrence rate. Several adjunctive therapies have been shown to be beneficial in recurrent or inaccessible 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 Top

1.
Dogruyol T, Tozum H, Eren TS. Rapidly growing cystic hygroma in an adult patient. Asian Cardiovasc Thorac Ann 2017;25:395-7.  Back to cited text no. 1
    
2.
Dokania V, Rajguru A, Kaur H, Agarwal K, Kanetkar S, Thakur P, et al. Sudden onset, rapidly expansile, cervical cystic hygroma in an adult: A rare case with unusual presentation and extensive review of the literature. Case Rep Otolaryngol 2017;2017:1061958.  Back to cited text no. 2
    
3.
Damaskos C, Garmpis N, Manousi M, Garmpi A, Margonis GA, Spartalis E, et al. Cystic hygroma of the neck: Single center experience and literature review. Eur Rev Med Pharmacol Sci 2017;21:4918-23.  Back to cited text no. 3
    
4.
Karkos PD, Spencer MG, Lee M, Hamid BN. Cervical cystic hygroma/lymphangioma: An acquired idiopathic late presentation. J Laryngol Otol 2005;119:561-3.  Back to cited text no. 4
    
5.
Bhatia V, Taksande R, Sondankar D. Cystic hygroma in an adult male: A diagnostic challenge. Iran J Pathol 2015;10:310-3.  Back to cited text no. 5
    
6.
Veeraraghavan G, Denny C, Lingappa A. Cystic hygroma in an adult; A case report. Libyan J Med 2009;4:160-1.  Back to cited text no. 6
    
7.
Upadhyaya VD, Bhatnagar A, Kumar B, Neyaz Z, Kishore JS, Sthapak E. Is multiple session of intralesional bleomycin mandatory for complete resolution of macrocystic lymphatic malformation? Indian J Plast Surg 2018;51:60-5.  Back to cited text no. 7
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