Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub. 2022, 166(1):117-125 | DOI: 10.5507/bp.2020.051

Cervical lymphocele: A rare complication following salvage surgery for ocular adnexal apocrine adenocarcinoma. A case report and review of the literature

Lukas Hauer^a, Jiri Gencur^a, Petr Posta^a, Zdenek Kasl^b, Stepan Rusnak^b, Alena Skalova^c, Miroslava Chalupova^a, Jan Liska^a, Vlasta Merglova^a

^a Department of Dentistry, University Hospital and Faculty of Medicine in Pilsen, Charles University, Alej Svobody 80, 304 60 Pilsen, Czech Republic

^b Clinic of Ophthalmology, University Hospital and Faculty of Medicine in Pilsen, Charles University, Alej Svobody 80, 304 60 Pilsen, Czech Republic

^c Sikl's Department of Pathology, Faculty of Medicine in Pilsen, Charles University, Ed. Benese 13, 30599, Pilsen, Czech Republic

Background: An ocular adnexal apocrine adenocarcinoma (OAAA) is an extremely rare, but potentially aggressive and life-threatening tumor with ill-defined management based only on recommendations from a limited number of reported cases. The development of cervical lymphocele following neck dissection is a very rare complication, but one with well established methods for prevention and treatment. Here we describe a previously unreported case of salvage surgery including neck dissection for OAAA in addition to an emergence of cervical lymphocele. A literature review of current knowledge on both pathological conditions is included.

Methods and Results: A 58-year-old man suffering from OAAA, previously treated with multiple eye-sparing excisions and adjuvant proton therapy, underwent salvage surgery for locoregional recurrence of the tumor. A partial orbitectomy with orbital exenteration, primary reconstruction and left-sided neck dissection was performed. The procedure was complicated by a cervical lymphocele resolved after the surgical therapy. The patient remained disease-free during the one-year follow-up.

Conclusion: OAAA is a locally aggressive tumor with potential to local or distant metastatic spread. Whole-body staging, regular clinico-radiological follow-up and stage-dependent therapy with surgery as the first-choice treatment is required. A cervical lymphocele as a complication of especially left-sided neck dissection is managed with a conservative or surgical therapy according to the level of lymph leakage, extent and localization of lesions, presence of local or systemic disorders and the period from primary surgery.

Keywords: cervical lymphocele, thoracic duct injury, neck dissection, adnexal tumor, apocrine adenocarcinoma, orbit, salvage surgery

Received: July 23, 2020; Revised: October 20, 2020; Accepted: October 22, 2020; Prepublished online: November 26, 2020; Published: March 1, 2022  Show citation