An Unusual Case of Acquired (Delayed Onset) Cystic Hygroma in a Child.
PBJ Sanneerappa, L Cunningham, P Gallagher, N Ramesh
Midland Regional Hospital, Portlaoise, Co. Laois, Republic of Ireland.
We present a case report of a 5-year and 8-month old girl presenting to our Accident and Emergency Unit with right sided facial swelling and redness for 3 weeks. She was seen by GP initially and was thought to have a slapped cheek disease, but as swelling enlarged and spread to the zygomatic area, she admitted and treated with intravenous Penicillin V and Flucloxicillin for 5 days as buccal cellulitis. There was no history of fever, ear ache, temporomandibular joint pain or trauma. She had received her MMR vaccination 2 weeks prior to the development of the swelling.
On examination her temperature was 37.3 degree Celsius, heart rate was 110, respiratory rate was 26 and oxygen saturations was 97%. It was a right sided, soft, boggy, non-tender swelling which was present both on the buccal and temporal area. There were non-tender cervical jugulo-digastric lymph nodes palpable bilaterally, measuring about 2x2cm. There was no hepatosplenomegaly or axillary or inguinal lymphadenopathy. No temporomandibular joint tenderness. Teeth and gums were normal. ENT exam and other systemic exam was within normal limits.
Her investigations were ESR 42, WCC-7.43, N-3.25, PLT-294 and CRP-2.53 (0-0.5). Urea, Creatinine, electrolytes, liver function test and serum amylase was all normal. Throat swab and blood culture were normal as well. Ultrasound parotid was normal, ultrasound neck suggested it to a type 2 branchial cyst and CT Neck demonstrated a large cystic hygroma on the right side.
She was treated with Oral Augmentin by General Practitioner. Then with intravenous benzyl penicillin and flucloxacillin for 5 days, then continued with oral for a total of 10 days from the hospital. While she was awaiting ENT input, and gradual swelling increase in size, she had diagnostic fine needle aspiration cytology which confirmed the diagnosis. Due to the complexity in the facial structures, she received therapeutic intralesional slerotherapy with Picibanil (OK-432). Her swelling gradually decreased in size to almost normal without further complications.
Cystic hygroma (CH) is one of the congenital malformations of the lymphatic system, usually seen in a new born period. In a new born, it can commonly present in the neck1 and sometimes in unusual sites such as cheek, supra-sternal area, lumbar area and scrotum2. This case report demonstrates that cystic hygroma can also present as a delayed onset swelling in the face even after infancy. Cystic hygromas are diagnosed clinically as a transilluminating cystic swelling and radiologically by ultrasound, Computed Tomography and Magnetic Resonance Imaging. It is confirmed by fine needle aspiration of the fluid and histology. Treatment depends on the anatomical complexity surrounding the cystic hygroma. Surgical excision is considered for a peripheral CH with no deep tissue infiltration and sclerotherapy with Picibanil (OK-432) has become the first-line therapy for CH of head and neck3.
Phani Bhushan Jeerigehalli Sanneerappa
- Barry S, Allotey J, Brundler AM, Duggal MS. Case report: cystic hygroma ,Eur Arch Paediatr Dent.2012 Dec;13(6):323-5.
- Bilal Mirza, Lubna Ijaz, Muhammad Saleem, Muhammad Sharif, and Afzal Sheikh, Cystic Hygroma: An Overview, J Cutan Aesthet Surg. 2010 Sep-Dec; 3(3): 139–144, doi: 4103/0974-2077.74488
- Elena Rebuffini, Luca Zuccarino, Emma Grecchi, Francesco Carinci, and Vittorio Emanuele Merulla Picibanil (OK-432) in the treatment of head and neck lymphangiomas in children Dent Res J (Isfahan). 2012 Dec; 9(Suppl 2): S192–S196. doi: 4103/1735-3327.109752