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Cystic Hygroma in an Adult - Case Report and Review of Literature
Ketan R Vagholkar*, Haritosh Kvelankar+, Shalini A Nair**,
Meghal J Sanghavi***, Amit S Anguwar***
Abstract
Cystic hygromas are benign lesions arising due to a developmental anomaly in the lymphatic system. It is commonly seen in children but very rare in adults. Pre operative confirmation of diagnosis with CT imaging is essential in adults. Surgical excision is the mainstay of treatment. A case of cystic hygroma in an adult is presented with a brief review of literature.
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| Fig. 1 : Clinical photograph of the swelling situated on the left side of the neck. |
Fig. 2 : Encircled region showing acid fast bacilli suggestive of tuberculosis. |
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Introduction
Cystic hygromas are multiloculated cystic
structures that are benign in nature. They are also called haemangiolymphomas as they are a benign proliferation of lymphoid tissue. It is commonly seen in the paediatric population but hardly seen in adults. A case of a cystic hygroma presenting in adulthood is presented along with a review of literature.
Case Report
A 20 year old male presented with a 3 week history of a swelling on the left side of the neck. The swelling had increased in size rapidly. Patient gave a history of fever which was associated with an increase in the size of the swelling. There were no symptoms suggestive of obstruction such as dysphagia or dyspnoea.
Physical examination revealed a multiloculated cystic swelling measuring 12 x 10 x 8 cms extending from the left mastoid process to the supraclavicular region vertically, while horizontally it extended from just lateral to the midline of the neck to the posterior border of the sternomastoid muscle (Fig. 1). There was no movement of the swelling with deglutition. The swelling was brilliantly transilluminant. Oropharyngeal endoscopy did not reveal any oropharyngeal extension. CT scan of the neck revealed a multicystic swelling. There was no intrathoracic extension. FNAC of the swelling revealed abundant lymphocytes. No malignant cells were seen.
Wide surgical excision of the swelling was done through a Z shaped incision made over the lateral aspect of the neck (Fig. 2). Utmost care was taken to safeguard the major veins and sternomastoid muscle. Post operative recovery was uneventful. The patient has been followed up for the last 10 months with no evidence of recurrence as yet.
Discussion
Radenbacher described cystic hygroma in 1828. The term means a moist tumour. Cystic hygroma belongs to a group of diseases now recognized as lymphatic malformations.1 These are benign in nature. They form as the result of budding lymphatics and therefore are frequently encountered in the neck (75%) and axilla (25%).Lymphatic channels are formed from a series of clefts that develop in the mesenchyme around the sixth week of gestation. From these channels sacs are formed that establish drainage with the venous system. Failure to establish venous drainage results in dilated disorganized lymph channels. These retain the potential for growth in various areas especially the neck, which in the largest form present as cystic hygromas.2 They frequently engulf neurovascular structures. Three types have been described.1 capillary characterized by small thin walled lymphatic channels.2 cavernous with large channels with a fibrous coat3 cystic characterized by large cystic endothelial lined spaces. Airway obstruction is the most critical complication of cystic hygroma.They may involve the oropharynx, tongue, base of tongue and the supraglottic larynx are involved. The risk of infection is very high. These lymphatic malformations are commonly seen at birth or during the prenatal period. They are usually seen in patients suffering from chromosomal anomalies usually Turner’s syndrome. They are also seen in Down’s and Noonan’s syndrome.2 They are rarely encountered in adulthood. Hence in adults the diagnosis could pose a diagnostic dilemma.
The treatment of choice is surgical excision but this in a few cases could be difficult due to engulfment of neurovascular structures. However the recurrence rate is low if removal is complete. The recurrence rate is 15%.3,4
Other therapies have been suggested but with limited success. Results with intralesional injection of OK-432 are encouraging.6
Bleomycin and fibrin tissue sealant, have shown to be beneficial but the pulmonary toxicity limits the use of bleomycin.7,8 Since this lesion is rare in adults preoperative imaging is of utmost importance either a CT or a MRI is of great help.4,5 Wide surgical excision is the treatment of choice.4
In conclusion cystic hygromas are very rare in adults. Pre operative imaging with CT or MRI is of utmost importance in the diagnosis. Surgical excision is the treatment of choice.
Acknowledgements
We are grateful and thank Dr. V.B. Shukla, Medical Superintendent of Rajawadi Municipal General hospital Ghatkopar Mumbai - 77. for allowing us to publish this case report.
References
1. Albanese CT, Wiener ES: Cystic Hygroma. In : Spitz L, Coran AG, eds. Pediatric Surgery. London: Chapman and Hall; 1995 : 94-9.
2. Feins NR, Raffensperger JG. Cystic Hygroma, Lymphangioma, and lymphedema. In: Raffensperger JG. Ed. Swenson’s Pediatric Surgery. 5th ed. Norwalk; 1990:167-72.
3. Chappius IIP. Current aspects of cystic lymphangoima in the neck. Aishives de pediatre ; Jan1995; 1 (2) : 186-92.
4. Wiggs WJ, Sismanis A. Cystic hygroma in the adults: two case reports. Otolaryngology and Head and Neck surgery. Feb. 1994; 239-41.
5. Obon PJ et al. Reported case of cystic hygroma in an adult. Acta otorrinolaringologia Espanola. 1993: 45(3); 223-5.
6. Smith RJ et al. OK-432 therapy for lymphangiomas. Archives. Otolaryngology and head and Neck surgery. Nov 1996; 122 (11) :
1195-9.
7. Orford J et al. Bleomycin therapy for cystic hygroma. Journal of Paediatric Surgery 1995; 30 (9) : 325-8.
8. Castanon GAM et al. Cystic lymphangioma: treatment with adhesive fibrin tissue.Follow up study. Chirurgia Pediatrica 1996; 9 (1) : 36-9.
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LACK OF EVIDENCE FOR BENEFIT OF MULTIVITAMINS
A panel convened by the US National Institutes of Health concluded last week that there is insufficient evidence to either support or advise against the use of multivitamin and mineral supplements in the prevention of chronic diseases such as cancer; age-related sensory loss; and cardiovascular, endocrine, neurological, musculoskeletal, gastroenterological, renal, and pulmonary diseases.
More than half of American adults take a dietary supplement, mostly multivitamins and minerals, at a cost of US $23 billion a year. In addition, 65% of Americans take foods or drinks fortified by vitamins and minerals, in a market worth $36 billion.
The Lancet, 2006; 367 : 1704.
STROKE-UNIT CARE
‘Admission to a stroke-unit ward with dedicated beds and staff within 48 h of onset should be recommended for all patients with acute stroke, irrespective of age’
In an observational follow-up study, Livia Candelisa and colleagues compare the long-term outcomes of patients with acute stroke cared for in stroke units with those of individuals who received care in conventional wards. They showed that stroke-unit care was associated with a reduction in the likelihood of death or disability. The authors conclude that, provided admission occurs within 48 h of onset, patients with acute stroke should be cared for in stroke-unit wards with dedicated beds and staff. In a Comment, Peter Rothwell discusses the weaknesses and advantages of observational comparisons of outcomes with different clinical services.
Lancet, 2007; 254, 299. |
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