Archives of Craniofacial Surgery (대한두개안면성형외과학회지)

Korean Cleft Palate-Craniofacial Association (대한두개안면성형외과학회)
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Lymphaticovenular anastomosis for Morbihan disease: a case report

  • Jung Hyun Hong (Department of Plastic and Reconstructive Surgery, Pusan National University School of Medicine) ;
  • Changryul Claud Yi (Department of Plastic and Reconstructive Surgery, Pusan National University School of Medicine) ;
  • Jae Woo Lee (Department of Plastic and Reconstructive Surgery, Pusan National University School of Medicine) ;
  • Yong Chan Bae (Department of Plastic and Reconstructive Surgery, Pusan National University School of Medicine) ;
  • Ryuck Seong Kim (Department of Plastic and Reconstructive Surgery, Pusan National University School of Medicine) ;
  • Joo Hyoung Kim (Department of Plastic and Reconstructive Surgery, Pusan National University School of Medicine)
  • Received : 2022.11.22
  • Accepted : 2023.03.02
  • Published : 2023.06.20
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Abstract

Morbihan disease (MD) is a very rare condition characterized by rosaceous or erythematous lymphedema on the upper two-thirds of the face. A definitive management strategy for MD is lacking, and treatment is challenging. Herein, we present a case of persistent bilateral eyelid edema treated by lymphaticovenular anastomosis (LVA) and lymph node-vein bypass surgery. The patient experienced persistent bilateral eyelid edema. Indocyanine green lymphography was performed, and the diagnosis of bilateral facial lymphedema was confirmed. On the right side, a preauricular lymphatic vessel was anastomosed to a vein. On the left side, lymphostomy on the preauricular lymph node was done, with anastomosis to the transected proximal end of the concomitant vein of the transverse facial artery. Furthermore, a preauricular lymphatic vessel was anastomosed to a vein. Eyelid edema decreased and progressively improved on both sides. The outcome of this case suggests that LVA and lymph node-vein bypass surgery are appropriate for treating persistent eyelid edema related to MD.

Keywords

Acknowledgement

This work was supported by Pusan National University Research Grant, 2022.

References

  1. Vasconcelos RC, Eid NT, Eid RT, Moriya FS, Braga BB, Michalany AO. Morbihan syndrome: a case report and literature review. An Bras Dermatol 2016;91(5 suppl 1):157-9. https://doi.org/10.1590/abd1806-4841.20164291
  2. Veraldi S, Persico MC, Francia C. Morbihan syndrome. Indian Dermatol Online J 2013;4:122-4. https://doi.org/10.4103/2229-5178.110639
  3. Kim JH. Treatment of Morbihan disease. Arch Craniofac Surg 2021;22:131-4. https://doi.org/10.7181/acfs.2021.00185
  4. Donthi D, Nenow J, Samia A, Phillips C, Papalas J, Prenshaw K. Morbihan disease: a diagnostic dilemma: two cases with successful resolution. SAGE Open Med Case Rep 2021;9: 2050313X211023655.
  5. Kang JK, Lee JS, Choi JH, Shin MS, Yun BM. Surgical treatment of rhinophyma using an ordinary bur. Arch Craniofac Surg 2020;21:264-7. https://doi.org/10.7181/acfs.2020.00199
  6. Jang JW, Kang SY. Evaluation and management of facial granuloma caused by various injection materials. Arch Craniofac Surg 2021;22:26-32. https://doi.org/10.7181/acfs.2020.00689
  7. Lee CL, Lee IS, Jung SG. A case of nodal marginal zone B-cell lymphoma of the lower eyelid. Arch Craniofac Surg 2020;21:249-52. https://doi.org/10.7181/acfs.2020.00241
  8. Kim JE, Sim CY, Park AY, Hong SA, Park YL, Jang SY, et al. Case series of Morbihan disease (extreme eyelid oedema associated with rosacea): diagnostic and therapeutic approaches. Ann Dermatol 2019;31:196-200. https://doi.org/10.5021/ad.2019.31.2.196
  9. Chalasani R, McNab A. Chronic lymphedema of the eyelid: case series. Orbit 2010;29:222-6. https://doi.org/10.3109/01676831003695735
  10. Bechara FG, Jansen T, Losch R, Altmeyer P, Hoffmann K. Morbihan's disease: treatment with CO2 laser blepharoplasty. J Dermatol 2004;31:113-5. https://doi.org/10.1111/j.1346-8138.2004.tb00518.x
  11. Hattori Y, Hino H, Niu A. Surgical lymphoedema treatment of Morbihan disease: a case report. Ann Plast Surg 2021;86:547-50. https://doi.org/10.1097/SAP.0000000000002797
  12. Aboutaam A, Hali F, Baline K, Regragui M, Marnissi F, Chiheb S. Morbihan disease: treatment difficulties and diagnosis: a case report. Pan Afr Med J 2018;30:226.
  13. Boparai RS, Levin AM, Lelli GJ Jr. Morbihan disease treatment: two case reports and a systematic literature review. Ophthalmic Plast Reconstr Surg 2019;35:126-32. https://doi.org/10.1097/IOP.0000000000001229
  14. Yamada Y. The studies on lymphatic venous anastomosis in lymphedema. Nagoya J Med Sci 1969;32:1-21.
  15. O'Brien BM, Chait LA, Hurwitz PJ. Microlymphatic surgery. Orthop Clin North Am 1977;8:405-24. https://doi.org/10.1016/S0030-5898(20)30703-3
  16. O'Brien BM, Sykes P, Threlfall GN, Browning FS. Microlymphaticovenous anastomoses for obstructive lymphedema. Plast Reconstr Surg 1977;60:197-211. https://doi.org/10.1097/00006534-197708000-00006
  17. Koshima I, Imai H, Yoshida S, Nagamatsu S, Yokota K, Mizuta H, et al. Lymphaticovenular anastomosis for persistent immunosuppressant-related eyelid edema. Int Microsurg J 2018;2:1. 
  18. Chen WF, Tuncer FB, Zeng W. Advanced technical pearls for successful supermicrosurgical lymphaticovenular anastomosis. Ann Plast Surg 2021;86(3S Suppl 2):S165-72.  https://doi.org/10.1097/SAP.0000000000002689