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

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

Lukas Hauera, Jiri Gencura, Petr Postaa, Zdenek Kaslb, Stepan Rusnakb, Alena Skalovac, Miroslava Chalupovaa, Jan Liskaa, Vlasta Merglovaa 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

Hauer, L., Gencur, J., Posta, P., Kasl, Z., Rusnak, S., Skalova, A., ... Merglova, V. (2022). Cervical lymphocele: A rare complication following salvage surgery for ocular adnexal apocrine adenocarcinoma. A case report and review of the literature. Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub.,166(1),117-125. doi:10.5507/bp.2020.051

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