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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 6  |  Issue : 1  |  Page : 52-54

Cystic hygroma: Clinical, ultrasonographic, and cytological co-relation


1 Department of Oral Medicine and Radiology, Swargiya Dadasaheb Kalmegh Smruti Dental College and Hospital, Nagpur, Maharashtra, India
2 Department of Conservative Dentistry, Sharad Pawar Dental College, DMIMS, Sawangi (M), Wardha, Maharashtra, India
3 Department of Oral Medicine and Radiology, Sharad Pawar Dental College, DMIMS, Sawangi (M), Wardha, Maharashtra, India

Date of Web Publication11-Aug-2016

Correspondence Address:
Rakhi Chandak
44, Jeevan-Chhaya Building, Behind Hotel Center Point, New Ramdaspeth, Nagpur - 440 010, Maharashtra
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2229-6360.188237

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  Abstract 

Cystic hygromas are rare, congenital, benign lesions occurring early in life, mainly in the head, neck, and oral cavity. It is an aberrant proliferation of lymphatic vessels resulting from abnormal development of the lymphatic system. Lymphangiomas are classified as microcystic, macrocystic, and cystic hygromas according to the size of the lymphatic cavities incorporated. They are soft, vary in size and shape, and tend to grow extensively if not surgically excised. They are multilocular cysts filled with clear or yellow lymph fluid. Histopathologically, lymphangiomas are of three types: Lymphangioma simplex, cavernous lymphangioma, and cystic hygroma depending on the size of vascular spaces and thickness of the adventitia. The present report describes a case of cystic hygroma of the left posterior region of jaw in an 11-year-old male child and its clinical, ultrasonographic, and cytological correlation.

Keywords: Benign lesions; congenital; lymph fluid; lymphangiomas; multilocular cysts


How to cite this article:
Chandak R, Chandak M, Rawlani S. Cystic hygroma: Clinical, ultrasonographic, and cytological co-relation. Indian J Multidiscip Dent 2016;6:52-4

How to cite this URL:
Chandak R, Chandak M, Rawlani S. Cystic hygroma: Clinical, ultrasonographic, and cytological co-relation. Indian J Multidiscip Dent [serial online] 2016 [cited 2019 Sep 22];6:52-4. Available from: http://www.ijmdent.com/text.asp?2016/6/1/52/188237


  Introduction Top


Cystic hygromas are rare, congenital, benign lesions occurring mainly in the head, neck, and oral cavity. It is an aberrant proliferation of lymphatic vessels resulting from abnormal development of the lymphatic system. [1],[2] Three theories have been proposed to explain the origin of this abnormality. The first suggests that a blockage or arrest of normal growth of the primitive lymph channels occurs during embryogenesis; the second suggests that the primitive lymphatic sac does not reach the venous system while the third advances the hypothesis that during embryogenesis, lymphatic tissue lays in the wrong area. [1],[2] Lymphangiomas are classified as microcystic, macrocystic, and cystic hygromas according to the size of the lymphatic cavities incorporated. [1],[2]

Histopathologically, lymphangiomas are of three types: Lymphangioma simplex, cavernous lymphangioma, and cystic hygroma depending on the size of vascular spaces and thickness of the adventitia. These account for 6% of benign tumors in childhood and 5% of vascular tumors, invading various parts of the body including the retroperitoneum, mesentery, groin, extremities, chest wall, mediastinum, and within viscera, and the head and neck region accounting for 40-70% of all lesions. [3],[4] The present report describes a case of cystic hygroma of the left posterior region of jaw in an 11-year-old male child and its clinical, ultrasonographic, and cytological correlation.


  Case Report Top


An 11-year-old male patient's father reported with complaint of pain and swelling in the lower left posterior region of jaw for 10 days. He stated that swelling was present on the left lateral side of neck since birth and no pain was associated with swelling. At the age of 1.5 years, the patient was operated and the case diagnosed as cystic hygroma; after that, swelling was decreased in size, but small swelling was present in lower left posterior region of the jaw. The patient gave a history that swelling was increased during cough, cold, and fever. At present, swelling increased in size and associated with pain for 10 days; drainage of swelling was done in a medical college at Yavatmal and referred patient here for further treatment.

Clinical examination revealed that extraorally, swelling was extending from left corner of mouth to left angle of mandible and superoinferiorly from line joining outer canthus of eye to 2 cm below the angle of mandible. It was approximately 8 cm × 6 cm in size. Overlying skin was normal and color was normal; temperature slightly raised [Figure 1]. All the margins were diffused; swelling was tender on pressure, mobile, nonpulsatile, soft in consistency at the superior border and hard at inferior border, noncompressible, fluctuant, and the transillumination test was positive. No bruit was there on auscultation.
Figure 1: Clinical extraoral photograph of swelling in the left posterior region of jaw

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Intraorally, swelling was present from mesial surface of the left second deciduous molar to distal surface of left first permanent molar obliterating buccal mucosa. Consistency was soft to firm, nontender on palpation, color was normal; size is 2 cm × 3 cm approximately. Mouth opening was reduced after swelling. When swelling was seen at the posterior region of jaw, it was important to formulate the differential diagnosis since this would help further evaluation of the condition and management of the patient. After considering all clinical findings, following entities were considered in differential diagnosis: Hemangioma, lymphangioma, cystic hygroma, lymphadenoma. After that, the patient has advised investigatory workup included complete hemogram, intraoral radiographs, orthopantomograph, ultrasonography, and fine-needle aspiration cytology (FNAC) or biopsy. Routine hematological investigations were within normal limit. Orthopantomograph showed no abnormality [Figure 2].
Figure 2: Orthopantomograph showing no abnormality

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Ultrasonographic findings of cystic hygroma characteristically appeared as a multiloculated anechoic mass with hyperechoic septae of variable thickness [Figure 3]. In our case, sonographic appearances of cystic hygroma were encountered anechoic with thick hyperechoic septae. FNAC revealed clear lymphoid fluid. FNAC smears of fluid aspirated from a lesion contained cholesterol crystals, lymphoid cells mainly small lymphocytes in variable number, and endothelial cells, [Figure 4] so the diagnosis of cystic hygroma was confirmed on the basis of clinical, ultrasonographic, and cytological findings. Ultrasound was very helpful in determining the extent of cystic hygromas before surgery and in assessing postoperative complications and recurrences.
Figure 3: Ultrasonographic image shows multiloculated cystic mass with thick septae

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Figure 4: Cytology smears shows cystic spaces lined by endothelial cells with aggregates of mature lymphocytes

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Under all aseptic precautions and conditions, cystic hygroma treated surgically with wide block excision while preserving cosmetic function, and histopathologically, diagnosis of cystic hygroma was confirmed. Postoperatively, the patient recovered well with no signs of any neurological dysfunction.


  Discussion Top


Cystic hygroma is believed to arise from a congenital malformation of the lymphatic system, in which a failure of communication between the lymphatic and venous pathways leads to lymph accumulation. [5] They are usually encountered at birth or in early infancy and rarely extend into the mediastinum. [6] Lymphangiomas may be divided histologically into two major groups based on the depth and the size of abnormal lymph vessels. The superficial ones are called lymphangioma circumscriptum. The more deep-seated ones are cavernous lymphangioma or cystic hygroma. [7] Cystic hygromas are deeply seated in areas of areola or loose connective tissues. They appear early in life as large soft-tissue mass on the axilla, cheek, neck, or groin. They are soft, vary in size and shape, and tend to grow extensively if not surgically excised. They are multilocular cysts filled with clear or yellow lymph fluid. [8] Usually, cystic hygromas are diagnosed clinically with large size, location, and translucence. Cystic hygromas may be associated with Turner syndrome, Noonan syndrome, trisomies, fetal alcohol syndrome, chromosomal aneuploidy, cardiac anomalies, and fetal hydrops. [9]

In the present case, we had performed surgery but other treatment modalities include aspiration, radiation, and injection of sclerosing agents, in particular the agent OK-432, derived from a strain of Streptococcus pyogenes, which has been used successfully, especially in macrocystic lymphangiomas and in patients who are at increased anesthetic risk. It is not uncommon for infants to develop neural paresis or paralysis after excision of massive cervical lymphangiomas. These congenital abnormalities tend to distort normal anatomy. [10]


  Conclusion Top


Cystic hygromas are benign tumors of lymphatic system, manifesting mainly in early childhood and are best treated surgically with conservative approach. For the confirmation of final diagnosis, correlation of clinical, ultrasonographic, and cytological finding is needed.

Acknowledgment

We would like to acknowledge General support by departmental chair, electricity.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest

 
  References Top

1.
Kennedy TL. Cystic hygroma-lymphangioma: A rare and still unclear entity. Laryngoscope 1989;99:1-10.  Back to cited text no. 1
    
2.
Sichel JY, Udassin R, Gozal D, Koplewitz BZ, Dano I, Eliashar R. OK-432 therapy for cervical lymphangioma. Laryngoscope 2004;114:1805-9.  Back to cited text no. 2
    
3.
Stromberg BV, Weeks PM, Wray RC Jr. Treatment of cystic hygroma. South Med J 1976;69:1333-5.  Back to cited text no. 3
[PUBMED]    
4.
Van Cauwelaert P, Gruwez JA. Experience with lymphangioma. Lymphology 1978;11:43-8.  Back to cited text no. 4
[PUBMED]    
5.
Schefter RP, Olsen KD, Gaffey TA. Cervical lymphangioma in the adult. Otolaryngol Head Neck Surg 1985;93:65-9.  Back to cited text no. 5
[PUBMED]    
6.
Grosfeld JL, Skinner MA, Rescorla FJ, West KW, Scherer LR 3 rd . Mediastinal tumors in children: Experience with 196 cases. Ann Surg Oncol 1994;1:121-7.  Back to cited text no. 6
    
7.
Mordehai J, Kurzbart E, Shinhar D, Sagi A, Finaly R, Mares AJ. Lymphangioma circumscriptum. Pediatr Surg Int 1998;13:208-10.  Back to cited text no. 7
    
8.
Rasidaki M, Sifakis S, Vardaki E, Koumantakis E. Prenatal diagnosis of a fetal chest wall cystic lymphangioma using ultrasonography and MRI: A case report with literature review. Fetal Diagn Ther 2005;20:504-7.  Back to cited text no. 8
    
9.
Carr RF, Ochs RH, Ritter DA, Kenny JD, Fridey JL, Ming PM. Fetal cystic hygroma and Turner's syndrome. Am J Dis Child 1986;140:580-3.  Back to cited text no. 9
[PUBMED]    
10.
Ogita S, Tsuto T, Nakamura K, Deguchi E, Tokiwa K, Iwai N. OK-432 therapy for lymphangioma in children: Why and how does it work? J Pediatr Surg 1996;31:477-80.  Back to cited text no. 10
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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