Erişkinlerde Baş ve Boyun Lenfanjiomları: Tek Merkez Deneyimi

Öz

Amaç: Hastanemizde cerrahi rezeksiyon uygulanan erişkin baş boyun lenfanjiomlarının klinikopatolojik özelliklerini ortaya koymayı amaçladık. Materyal ve Metod: 2008-2020 yılları arasında laboratuvarımızda incelenen 18 yaş üstü hastaların baş boyun bölgesinden yapılan tüm cerrahi rezeksiyon materyalleri retrospektif olarak incelendi. Lenfanjiyom tanısı konulan olgular çalışmaya dahil edildi. Bulgular: Çalışmaya dahil edilen 34 lenfanjiomdan 17'si kadın, 17'si erkekti. Hastaların yaşları 19 ile 76 arasında değişmekteydi. Medyan lenfanjiyom boyutu 0.8 cm idi. Lezyonlar 15 (% 44,1) hastada yüzde, dokuz (% 26,5) hastada ağız boşluğunda, altı (% 17,6) hastada boyunda, üç hastada (% 8,8) orbital ve perioküler bölgede, bir hastada (%2,9) kulakta lokalize idi. Lenfanjiyomların 16'sı (%47,0) lenfanjiyoma sirkumscriptum, 14'ü (%41,2) kavernöz lenfanjiyom ve dördü (%11,8) kistik lenfanjiyom olarak tanımlandı. 1.5 cm'den büyük tüm lezyonlar boyun veya ağız boşluğunda yerleşmişti. Ağız boşluğundaki tüm lezyonlara kavernöz lenfanjiom tanısı konuldu. Dört kistik lenfanjiomun tamamı boyunda tespit edildi. Lenfanjiom iki olguda nüksetti; malign transformasyon saptanmadı. Sonuç: Baş boyun lenfanjiyomları erişkinlerde nadir değildir, her yaşta görülebilirler. Kistik lenfanjiomlar sıklıkla boyunda ve kavernöz lenfanjiomlar ağız boğluğunda görülür. Total cerrahi rezeksiyon hem tedavi hem de kesin tanı sağlar. Nüks görülebilir ancak malign transformasyon beklenmez.

Anahtar Kelimeler

• lenfanjiyom
• baş ve boyun
• kavernöz lenfanjiyom
• kistik lenfanjiyom
• lenfanjiyoma sirkumskriptum
• vasküler malformasyon

Head and Neck Lymphangiomas in Adults: A Single Center’s Experience

Öz

Aim: We aimed to reveal the clinicopathologic features of adult head and neck lymphangiomas that underwent surgical resection in our hospital. Material and Method: All surgical resection materials of patients aged older than 18 years from the head and neck region that were examined at our labaratory between 2008 and 2020 were retrospectively reviewed. Cases diagnosed as lymphangioma were included in the study. Results: Of the 34 lymphangiomas included in the study, 17 were from females and 17 from males. The ages of the patients ranged between 19 and 76 years. The median lymphangioma size was 0.8 cm. Lesions were located on the face in 15 (44.1%) patients, in the oral cavity in nine (26.5%), on the neck in six (17.6%), on the orbital and periocular region in three (8.8%), and on the ear in one (2.9%) patient. Of the lymphangiomas, 16 (47.0%) were defined as lymphangioma circumscriptum, 14 (41.2%) as cavernous lymphangioma, and four (11.8%) as cystic lymphangioma. All lesions larger than 1.5 cm were located in the neck or oral cavity. All lesions in the oral cavity were diagnosed as cavernous lymphangioma. All four cystic lymphangiomas were identified in the neck. Lymphangioma recurred in two cases; no malignant transformation was detected. Conclusion: Head and neck lymphangiomas are not uncommon in adults, they can be seen at any age. Cystic lymphangioma is often seen in the neck and cavernous lymphangioma in the oral cavity. Total surgical resection provides both treatment and definitive diagnosis. Recurrence can be seen, but malignant transformation is not expected.

Anahtar Kelimeler

• lymphangioma
• head and neck
• cavernous lymphangioma
• cystic lymphangioma
• lymphangioma circumscriptum
• vascular malformation

Kaynakça

1. Referans1: Thway K, Doyle LA. Lymphangioma and lymphangiomatosis. In: WHO Classification of Tumours Editorial Board. eds. Soft Tissue and Bone Tumours. 5th ed. Lyon: IARC Press, 2019:154-155
2. Referans2: Student PG, Rao CA. Lymphangioma of Neck in a 35-year Old Female: A Case Report of Rare Entity. Journal of Chalmeda Anand Rao Institute of Medical Sciences. Vol 2018;16(2): 207.
3. Referans3: Kaira V, Kaira P, Agarawal T. Cervical Cystic Lymphangiomas in Adults: A Case Series of a Rare Entity with Literature Review. Head and Neck Pathology. 2020;1-6.
4. Referans4: Patel GA, Schwartz RA. Cutaneous lymphangioma circumscriptum: frog spawn on the skin. International journal of dermatology. 2009;48(12):1290-1295.
5. Referans5: Jha SK, Jha S, Ranjan R, et al Management of Head And Neck Lymphangioma. Orissa Journal of Otolaryngolgy and HNS. 2019;13(1):39-43
6. Referans6: Porwal PK, Dubey KP, Morey A, et al. Bleomycin sclerotherapy in lymphangiomas of head and neck: prospective study of 8 cases. Indian Journal of Otolaryngology and Head & Neck Surgery. 2018;70(1):145-148.
7. Referans7: Valletti PA, Brucoli M, Boffano P, et al. A single-center experience in the management of head and neck lymphangiomas. Oral and Maxillofacial Surgery. 2020;24(1):109-115.
8. Referans8: Lerat J, Mounayer C, Scomparin A, et al. Head and neck lymphatic malformation and treatment: clinical study of 23 cases. European annals of otorhinolaryngology, head and neck diseases. 2016;133(6):393-396.

9. Referans9: Fatima S, Uddin N, Idrees R, et al. Lymphangioma circumscriptum: clinicopathological spectrum of 29 cases. Journal of the College of Physicians and Surgeons Pakistan. 2015;25(9):658.
10. Referans10: Eren S, Cebi AT, Isler SC, et al. Cavernous lymphangioma of the tongue in an adult: a case report. Journal of Istanbul University Faculty of Dentistry. 2017:51(2):49.
11. Referans11: Lee DH, Yoon TM, Lee JK, et al. Cavernous Lymphangioma in the Maxillary Sinus. Journal of Craniofacial Surgery. 2019;30(8):2520-2521.
12. Referans12: Tan B, Zhang SY, Wang YN, et al. Jejunal cavernous lymphangioma manifested as gastrointestinal bleeding with hypogammaglobulinemia in adult: A case report and literature review. World Journal of Clinical Cases. 2020;8(1):140.
13. Referans13: Wang X, Meng S, Duan K, et al. Treatment of Retroperitoneal Cavernous Lymphangioma: A Case Report. Chinese Medical Sciences Journal. 2020;35(3):283-285.
14. Referans14: Leong JF, Levin KB, Rajkumar V. Cavernous Lymphangioma of the Digits: A Rare Cause of Macrodactyly. Medicine & Health. 2019;14(2).
15. Referans15: Loetscher KCQ, Jandali AR, Garzoli E. Axillary cavernous lymphangioma in pregnancy and puerperium. Gynecologic and obstetric investigation. 2005;60(2):108-111.
16. Referans16: Cadena-Piñeros E, Rojas Gutiérrez A. Cervical lymphangioma in adults: case report and current treatment. Case reports. 2018;4(1):61-68.
17. Referans17: Azim MT, Hussain SM, Mughal MA. Primary Supraclavicular Cystic Lymphangioma in an Adult: A Rare Presentation. Journal of the College of Physicians and Surgeons--Pakistan: JCPSP. 2019;29(6):S11-S12.
18. Referans18- Aprea G, Guida F, Canfora A. Mesenteric cystic lymphangioma in adult: a case series and review of the literature. In BMC Surgery. 2013;13(S1):A4
19. Referans19- Lerat J, Mounayer C, Scomparin A. Head and neck lymphatic malformation and treatment: clinical study of 23 cases. European annals of otorhinolaryngology, head and neck diseases. 2016;133(6):393-396.
20. Referans20- Kotsis T, Exarchos G, Metaxa L. Recurrent Neck Lymphangioma in a Young Adult: Twenty-Three Years After Successful Treatment. Vascular and endovascular surgery. 2019;53(2):170-176.
21. Referans21- Usha V, Sivasankari T, Jeelani S. Lymphangioma of the tongue-a case report and review of literature. Journal of clinical and diagnostic research: JCDR. 2014;8(9):ZD12.
22. Referans22- Kolay SK, Parwani R, Wanjari S. Oral lymphangiomas–clinical and histopathological relations: An immunohistochemically analyzed case series of varied clinical presentations. Journal of oral and maxillofacial pathology: JOMFP. 2018;22(Suppl 1):S108.