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.

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

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

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

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Shashni AK, Sen S, Meel R, Sharma MC, Nag TC. Primary apocrine adenocarcinoma of caruncle in an elderly male. Can J Ophthalmol 2019;54(3):e123-5. PubMed DOI

Zhang L, Ge S, Fan X. A brief review of different types of sweat-gland carcinomas in the eyelid and orbit. Onco Targets Ther 2013;6:331-40. PubMed DOI

Duke TG, Fahy GT, Brown LJ. Malignant apocrine sweat gland carcinoma of the caruncle. Orbit 2000;19(1):7-11. PubMed DOI

Hoang A, Khine KT, Zaldivar R. Apocrine adenocarcinoma of lacrimal sac presenting as chronic dacryocystitis. J Ophthalmic Clin Res 2017;4(1):027. DOI

Stout AP, Cooley SG. Carcinoma of sweat glands. Cancer 1951;4(3):521-36. DOI

Rosales Santillan M, Tschen JA, Soparkar CN. Apocrine adenocarcinoma of the eyelid: case report and literature review on management. Dermatol Online J 2019;25(5). pii: 13030/qt2p62h61q. DOI

Hamilton BE, Nesbit GM, Gross N, Andersen P, Sauer D, Harnsberger HR. Characteristic imaging findings in lymphoceles of the head and neck. AJR Am J Roentgenol 2011;197(6):1431-5. PubMed DOI

Kimura M, Ohto H, Shibata A, Yamada H, Nishiwaki S, Umemura M. Cervical chyloma after neck dissection: a case report. Nagoya J Med Sci 2017;79(1):91-5.

Laccourreye O, Espinoza S, Mukundian S, Bonfils P. Prevention and cure of lymphorrhea and lymphocele after cervical lymph-node surgery. Eur Ann Otorhinolaryngol Head Neck Dis 2013;130(4):229-32. PubMed DOI

Stárek I, Salzman R, Pokorná Z, Skanderová D. Cyst of the right lymphatic duct. Eur Ann Otorhinolaryngol Head Neck Dis 2020;137(2):123-5. DOI

Whorton CM, Patterson JB. Carcinoma of Moll's glands with extramammary Paget's disease of the eyelid. Cancer 1955;8(5):1009-15. DOI

Aurora AL, Luxenberg MN. Case report of adenocarcinoma of glands of Moll. Am J Ophthalmol 1970;70(6):984-90. DOI

Futrell JW, Krueger GR, Chretien PB, Ketcham AS. Multiple primary sweat gland carcinomas. Cancer 1971;28(3):686-91. DOI

Ni C, Wagoner M, Kieval S, Albert DM. Tumours of the Moll's glands. Br J Ophthalmol 1984;68(7):502-6. DOI

Thomson SJ, Tanner NS. Carcinoma of the apocrine glands at the base of eyelashes; a case report and discussion of histological diagnostic criteria. Br J Plast Surg 1989;42(5):598-602. PubMed DOI

Seregard S. Apocrine adenocarcinoma arising in Moll gland cystadenoma. Ophthalmology 1993;100(11):1716-9. DOI

Paties C, Taccagni GL, Papotti M, Valente G, Zangrandi A, Aloi F. Apocrine carcinoma of the skin. A clinicopathologic, immunocytochemical, and ultrastructural study. Cancer 1993;71(2):375-81. DOI

Simionescu C, Damian C. Adenocarcinoma of Moll's glands. Oftalmologia 1997;41(4):333-4. PubMed

Paridaens D, Mooy CM. Apocrine sweat gland carcinoma. Eye (Lond) 2001;15(Pt 2):253-4. PubMed DOI

Shintaku M, Tsuta K, Yoshida H, Tsubura A, Nakashima Y, Noda K. Apocrine adenocarcinoma of the eyelid with aggressive biological behavior: report of a case. Pathol Int 2002;52(2):169-73. PubMed DOI

Pornpanich K, Chindasub P. Eyelid tumors in Siriraj Hospital from 2000-2004. J Med Assoc Thai 2005;88 Suppl 9:S11-4.

Barker-Griffith AE, Streeten BW, Charles NC. Moll gland neoplasms of the eyelid: a clinical and pathological spectrum in 5 cases. Arch Ophthalmol 2006;124(11):1645-9. DOI

Kanazawa S, Kubo T, Tañag M, Yamaguchi Y, Hoshida Y, Yano K, Hosokawa K. Apocrine adenocarcinoma originating from the Moll's gland of the eyelid: Report of a case. Eur J Plast Surg 2008;31(6):333-6. DOI

Akcay EK, Simsek S, Cagil N, Belenli O, Gumus M. Apocrine adenocarcinoma of the right eyelid and apocrine adenoma of the left maxillary sinus. Can J Ophthalmol 2008;43(5):609-10. PubMed DOI

Kumar S, Zafar SF, Raufi AM, Heath EI. Apocrine carcinoma of the face in a 62-year-old Asian man. Clin Pract 2011;1(3):e50. PubMed DOI

Valenzuela AA, Cupp DG, Heathcote JG. Primary apocrine adenocarcinoma of the eyelid. Orbit 2012;31(5):316-8. PubMed DOI

Hunold AC, Herwig MC, Holz FG, Fischer HP, Loeffler KU. Pigmented tumour of the eyelid with unexpected findings. Case Rep Pathol 2012;2012:471368. PubMed DOI

Figueira EC, Danks J, Watanabe A, Khong JJ, Ong L, Selva D. Apocrine adenocarcinoma of the eyelid: case series and review. Ophthalmic Plast Reconstr Surg 2013;29(6):417-23. PubMed DOI

Aldrees SS, Zoroquiain P, Alghamdi SA, Logan P, Kavalec C, Burnier M. Apocrine adenocarcinoma of the eyelid. Int J Ophthalmol 2016;9(7):1086-8.

Pagano Boza C, Vigo R, Premoli JE, Croxatto J, Gonzalez Barlatay J. A case report of a primary apocrine adenocarcinoma of the eyelid with literature review. Orbit 2018;37(5):389-92. PubMed DOI

Seelig MH, Klingler PJ, Oldenburg WA. Treatment of a postoperative cervical chylous lymphocele by percutaneous sclerosing with povidone-iodine. J Vasc Surg 1998;27(6):1148-51. PubMed DOI

Touska P, Constantinides VA, Palazzo FF. A rare complication: lymphocele following a re-operative right thyroid lobectomy for multinodular goitre. BMJ Case Rep 2012;2012. pii: bcr0220125747.

Chantarasak DN, Green MF. Delayed lymphocoele following neck dissection. Br J Plast Surg 1989;42(3):339-40. PubMed DOI

Nouwen J, Hans S, Halimi P, Laccourreye O. Lymphocele after neck dissection. Ann Otol Rhinol Laryngol 2004;113(1):39-42. DOI

Roh JL, Park CI. OK-432 sclerotherapy of cervical chylous lymphocele after neck dissection. Laryngoscope 2008;118(6):999-1002. PubMed DOI

Al-Suqri B, Dutton J. Iatrogenic chyloma in the neck: lymphoscintigraphic findings. Clin Nucl Med 2012;37(11):1126-8. PubMed DOI

Thiagarajan S, Shenoy AM, Veerabadriah P, Chavan P, Halkud R. Massive Chylorrhea following Total Thyroidectomy and Neck Dissection. Int J Head Neck Surg 2012;3(1):45-8. DOI

Campisi CC, Boccardo F, Piazza C, Campisi C. Evolution of chylous fistula management after neck dissection. Curr Opin Otolaryngol Head Neck Surg 2013;21(2):150-6. DOI