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Feasibility of dpFAMM flap in tongue reconstruction after facial vessel ligation and radiotherapy—case presentation

Feasibility of dpFAMM flap in tongue reconstruction after facial vessel ligation and... Background: Salvage surgery with reconstruction of the second and next primary tongue cancer remains diffi‑ cult, especially after earlier neck dissection and radiotherapy. In the current report, we describe the feasibility of the extended, double‑pedicled facial artery musculomucosal (dpFAMM) flap in the reconstruction of the patient with second primary tongue squamous cell carcinoma, after facial vessel ligation and radiotherapy. Case presentation: An 81‑ year‑ old female patient was operated on due to tongue squamous cell carcinoma (SCC) on the left side T3N1M0 in 2019. Bilateral selective neck dissection with tongue reconstruction was performed by island FAMM flap. The patient also suffered from synchronous mucinous breast carcinoma treated with tamoxifen. The second primary SCC of the tongue on the opposite (right) side was detected in 2020. The patient did not agree to surgical treatment; therefore, radiotherapy was performed. The local recurrence of the tongue cancer of the right side was treated surgically in 2021. Salvage surgery comprised hemiglossectomy and dpFAMM flap reconstruction with uneventful postoperative follow‑up. Conclusions: This case presentation proved that dpFAMM flap can be used in salvage surgery and reconstruction even in patients after ligation of facial vessels, irradiation, and in the course of hormone therapy. The flap is easy to handle, has good vascularity, and comprises a predictable method of reconstruction, especially for patients with severe comorbidities. Keywords: FAMM flap, Facial artery musculomucosal flap, Bozola flap, Tongue cancer, Reconstruction, Salvage surgery, Buccinator myomucosal flap, Tongue reconstruction, Tongue squamous cell carcinoma, dpFAMM flap Background of the tongue might be reconstructed by local flaps, such The incidence of synchronous and metachronous sec - as the facial artery musculomucosal (FAMM) flap and ond oral cancer is increasing due to a longer lifespan and its modifications [2–7]. The FAMM flap is an axial flap improvement of oncological therapy [1]. However, surgi- based on the facial artery and is useless in case of facial cal treatment and reconstruction of second and next pri- vessel ligation. In such cases, the reconstruction should mary cancer in the oral cavity remain difficult, especially be converted to a Bozola flap [8]. Extended, double- after previous neck dissection and radiotherapy. Defects pedicled FAMM (dpFAMM) flap owns modification of FAMM flap with facial and buccal vessel blood supply [3]. In the current report, we describe the feasibility of the *Correspondence: mgontarz@op.pl dpFAMM flap in the reconstruction in the patient with Department of Cranio‑Maxillofacial Surgery, Jagiellonian University second primary tongue squamous cell carcinoma, after Medical College, University Hospital, Jakubowskiego 2 Street, 30‑688 Cracow, Poland facial vessel ligation and radiotherapy. © The Author(s) 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. The Creative Commons Public Domain Dedication waiver (http:// creat iveco mmons. org/ publi cdoma in/ zero/1. 0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Gontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 2 of 6 Case presentation In November 2019, an 81-year-old female patient was admitted to the Department of Cranio-Maxillofacial Surgery of the Jagiellonian University in Cracow due to tongue squamous cell carcinoma (SCC) on the left side cT3N1M0. Clinical examination and computed tomog- raphy (CT) revealed a synchronous left breast tumor (Fig. 1). Biopsy from the breast tumor revealed mucinous carcinoma, and diagnostic imaging excluded dissemina- tion. After tumor board consultation, we decided to start the treatment with surgical excision of tongue SCC and bilateral neck dissection. Selective bilateral neck dissec- tion (level I–IV ipsilateral and I–III contralateral) was performed by a horizontal neck fold incision. During neck dissection, the facial vessels on the left side were preserved for an island FAMM (iFAMM) flap. How - ever, the facial vessels on the right side were ligated. Tongue cancer was excised with margins control by fro- zen section examination. Tongue reconstruction with an iFAMM flap was performed according to the technique described by Joseph et al. (Figs. 2, 3, and 4) [4]. The heal - ing process was uneventful. Histopathological examina- tion revealed squamous cell carcinoma G1 resected with inadequate distal margin (2mm) and metastasis in one Fig. 2 The harvested trilobed iFAMM flap lymph node (IIa cervical level) on the left side (pT3N1). The patient was qualified for postoperative radiotherapy and radical mastectomy with axillary node dissection. from the ulceration confirmed SCC G2. Clinical exami - However, the patient did not give her consent for the pro- nation and CT revealed only local advancement of the posed treatment. disease cT2N0M0. Breast cancer disease was stable. We In February 2020, the patient started hormone therapy proposed surgical treatment, but the patient again did with tamoxifen due to breast cancer and had oncologi- not give her consent. For that reason, the patient was cal controls in our outpatient department every month. In December 2020, after 4 months from the last visit, the patient was admitted to the outpatient clinic with suspi- cion of tongue cancer on the opposite, right side. Biopsy Fig. 1 CT axial view of the chest showing huge breast cancer on the Fig. 3 Transposition of the iFAMM flap with the facial vessel pedicle left side over the mandible G ontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 3 of 6 Fig. 5 CT with 3D reconstruction showing stump of ligated facial artery on the right side (blue arrow), preserved buccal artery on the right side (green arrow), and absence of the facial vessels after iFAMM harvesting on the left side (red arrow) earlier (Figs.  7 and 8) [3]. Histopathological examination Fig. 4 Immediate postoperative result of tongue reconstruction with revealed SCC G2 resected with adequate margins >5mm iFAMM flap (pT3). The healing process was uneventful (Fig.  9). Unfor- tunately, in August 2021, we observed enlarged cervical lymph nodes on the left side. Neck dissection in levels II and V was performed. Histopathological examination qualified for definitive radiotherapy. However, the total dose was reduced due to general health conditions. The patient received 30 Gy in 10 fractions on the second pri- mary and cervical region, and the treatment was finished in February 2021. In May 2021, 2 months after the last visit, the patient was admitted to our outpatient depart- ment with dysphagia and odynophagia. Clinical examina- tion and biopsy confirmed local recurrence of the tongue cancer on the right side. CT showed local recurrence with dimensions 21 × 42 × 29 mm and without suspicious neck lymph nodes (rT3N0M0) (Figs.  5 and 6). Definitive radiotherapy was impossible at this stage of the disease. The patients agreed to salvage surgery, hemiglossectomy with reconstruction and without neck dissection. Due to the fact that the right facial vessels were ligated in November 2019 and considering the extent of the defect, iFAMM was not possible. Also, the general condition of the patient was a contraindication to extensive, long pro- cedures such as free flap reconstruction. Another con - traindication for free flap reconstruction was a higher risk of early postoperative thrombosis of microvascular anastomoses due to continuous tamoxifen intake. Based Fig. 6 CT axial view of extensive local recurrence after radiotherapy on CT evaluation, we decided to use a dpFAMM flap of the tongue SCC on the right side (red arrow) for reconstruction, which was performed as described Gontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 4 of 6 confirmed three metastatic lymph nodes. The patient was disqualified from adjuvant radiotherapy and chemother - apy. The patient died 2 months following neck dissection due to pneumonia. Discussion and conclusions Soft tissue defects of the oral cavity are challenging in reconstructive surgery due to loss of motility, secretion, and sensory functions of the mucous membrane. Recon- struction especially of tongue defects should ensure proper patient’s speech and swallowing. Skin grafts are ineffective in the case of bone exposure and have a ten - Fig. 7 Salvage surgery. Surgical specimen of the tongue after right hemiglossectomy dency to keratinization and cicatrization, which addi- tionally decreases the movability of the remaining, healthy oral mucosa [8]. Also, most pedicled regional and free flaps containing skin islands are characterized by impaired sensitivity, keratinization, hair growth, and donor site morbidity. Local musculomucosal flaps are a good method of reconstruction of moderate tongue defects [3]. According to Massarelli et  al. [8], all buc- cinator musculomucosal flaps provide proper mucus secretion and sensitivity without shrinking tendency, especially after radiotherapy. The iFAMM flap in the tongue reconstruction is pos - sible in case of facial vessel preservation. Massarelli et al. [8] suggest that iFAMM flap pedicled solely on the facial artery with the surrounding fat tissue provides the cor- rect venous drainage, without flap congestion. However, Rahpeyma et  al. [9] in experimental studies on dogs observed iFAMM flap loss in each case of facial vein ligation. For that reason, iFAMM flap, pedicled only on the facial artery, is not acceptable for clinical usage [9]. Fig. 8 Immediate postoperative result of tongue reconstruction with dpFAMM flap In our case, the patient had facial vein and artery ligated during neck dissection  18 months earlier. This was the main contraindication for tongue reconstruction by iFAMM. However, the vascularization of the buccinator muscle is derived from the branches of the facial vessel in the anterior part and buccal vessels in the posterior region. Due to the fact that CT revealed preserved distal part of the facial artery in the buccal region (Fig.  5) and a large number of anastomoses between facial and buc- cal angiosome, which ensures good blood supply through the buccal vessels, we decided to used dpFAMM flap for this reconstruction [8]. The dpFAMM flap combines the advantages of both the FAMM and the Bozola flap, which allows its extension to be increased with sufficient venous drainage [3]. Another issue concerning reconstruction is preopera- tive radiotherapy. It should be pointed out that both the donor and recipient sites comprise the irradiation field, Fig. 9 Final result. Three months after tongue reconstruction with which implies a higher risk of healing process problems dpFAMM flap (green arrow) and iFAMM flap (blue arrow—21 months with potential flap necrosis. O’Leary and Bundgaard [10] after surgery) suggested that the FAMM flap is not suitable in patients, G ontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 5 of 6 Abbreviations who underwent the previous radiotherapy, due to the FAMM: Facial artery musculomucosal flap; dpFAMM: Double ‑pedicled facial risk of such complications as trismus, bleeding, osteo- artery musculomucosal flap; iFAMM: Island facial artery musculomucosal flap; radionecrosis, and impaired healing followed by flap SCC: Squamous cell carcinoma; CT: Computer tomography. necrosis. They observed partial flap necrosis in 75% (3 Acknowledgements from 4 patients) of cases. On the other hand, Ayad et al. We thank Dr. Krzysztof Śliwiński for technical assistance in clinical imaging. [11] did not notice the specific complication rate in the Authors’ contributions group of 10 patients previously irradiated. In our case, MG and GWP designed the study and drafted the manuscript. MG, JB, and KG we also did not detect problems with healing, bleeding, reviewed this case presentation and checked the patient’s treatments. MG, TM, and proper mouth opening. The short term of the follow- PS, and JZ analyzed the data and provided the clinical image and information. MG, JZ, and GWP developed the concept and edited the paper. The authors up did not allow for evaluation of possible mandibular have read and approved the final manuscript. osteoradionecrosis. The healing process can also be disturbed by hor- Funding None. mone therapy. According to Billon et  al. [12], hor- mone therapy, including tamoxifen intake, seems Availability of data and materials to be associated with a higher risk of postoperative All data during the study are included within the article. wound healing complications in patients with breast reconstruction. In addition, Parikh et  al. [13] in their Declarations meta-analysis concluded that perioperative tamox- Ethics approval and consent to participate ifen therapy may increase the risk of thrombotic flap Written informed consent was obtained from the patient prior to submission complications and flap loss in patients undergoing free of the case report. The study was conducted according to the guidelines of the Declaration of Helsinki and approved by the Institutional Review Board flap reconstruction due to breast cancer. They sug- of the Jagiellonian University (No: 1072.6120.229.2021, date of approval is on gested that short cessation of the tamoxifen therapy, September 29, 2021). about 4 weeks prior to reconstructive treatment, might Consent for publication decrease the risk of complications [13]. Our patient The patient has given her consent for the case report to be published. also suffered from synchronous mucinous carcinoma of the left breast treated with tamoxifen 15 months Competing interests The authors declare that they have no competing interests. before salvage surgery. This was one of the contrain- dications for free radial forearm flap application and Received: 3 November 2021 Accepted: 7 March 2022 choice of dpFAMM flap for tongue reconstruction. Transient discontinuation of tamoxifen before surgery was not recommended. However, the healing process was uneventful. References 1. Qaisi M, Vorrasi J, Lubek J, Ord R. Multiple primary squamous cell carcino‑ The extension of the dpFAMM flap in the anterior part mas of the oral cavity. J Oral Maxillofac Surg. 2014;72(8):1511–6. https:// of the buccinator muscle provided an ability for tongue doi. org/ 10. 1016/j. joms. 2014. 03. 012. reconstruction, even after hemiglossectomy. If the apex 2. Duranceau M, Ayad T. The facial artery musculomucosal flap: modification of the harvesting technique for a single‑stage procedure. Laryngoscope. of the tongue can be preserved during ablative surgery, 2011;121:2586–9. like in this case, the dpFAMM flap can be doubled. The 3. Gontarz M, Bargiel J, Gąsiorowski K, Marecik T, Szczurowski P, Zapała J, preserved apex of the tongue is rotated backward to et al. Extended, double‑pedicled facial artery musculomucosal (dpFAMM) flap in tongue reconstruction in edentulous patients: preliminary report obtain the best clinical result of the reconstruction. Flap and flap design. Medicina (Kaunas). 2021;57(8):758. https:// doi. org/ 10. harvesting takes around 30–50 min and can be done 3390/ medic ina57 080758. by one surgical team. The dpFAMM flap has a perfect 4. Joseph ST, Naveen BS, Mohan TM. Islanded facial artery musculomucosal flap for tongue reconstruction. Int J Oral Maxillofac Surg. 2017;46:453–5. color and structure, matching the surrounding tissues 5. Zhao Z, Zhang Z, Li Y, Li S, Xiao S, Fan X, et al. The buccinator muscu‑ without additional extraoral scars. This case presenta - lomucosal island flap for partial tongue reconstruction. J Am Coll Surg. tion only highlights that the dpFAMM flap can be used 2003;196:753–60. 6. Massarelli O, Gobbi R, Raho MT, Tullio A. Three‑ dimensional primary for reconstruction in salvage surgery, even, if the facial reconstruction of anterior mouth floor and ventral tongue using the vessels had been ligated previously and the patient was ‘trilobed’ buccinator myomucosal island flap. Int J Oral Maxillofac Surg. irradiated and treated with hormone therapy. However, 2008;37(10):917–22. https:// doi. org/ 10. 1016/j. ijom. 2008. 07. 020. 7. Massarelli O, Gobbi R, Soma D, Tullio A. The folded tunnelized‑facial artery further studies with more patients must be conducted myomucosal island flap: a new technique for total soft palate reconstruc‑ to prove the utility of the dpFAMM flap in such cases. tion. J Oral Maxillofac Surg. 2013;71(1):192–8. https:// doi. org/ 10. 1016/j. Other advantages of the dpFAMM flap include its feasi - joms. 2012. 03. 030. 8. Massarelli O, Baj A, Gobbi R, Soma D, Marelli S, De Riu G, et al. Cheek bility in the reconstruction of other areas, like the floor of mucosa: a versatile donor site of myomucosal flaps. Technical and func‑ the mouth, the alveolar ridge, the soft and hard palates, tional considerations. Head Neck. 2013;35(1):109–17. https:// doi. org/ 10. or the oropharynx. 1002/ hed. 22933. Gontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 6 of 6 9. Rahpeyma A, Khajehahmadi S, Sedigh HS. Facial artery myomucosal flap, pedicled solely on the facial artery: experimental design study on survival. J Craniofac Surg. 2016;27(7):e614–5. https:// doi. org/ 10. 1097/ SCS. 00000 00000 002947. 10. O’Leary P, Bundgaard T. Good results in patients with defects after intraoral tumour excision using facial artery musculo‑mucosal flap. Dan Med Bull. 2011;58(5):A4264. 11. Ayad T, Kolb F, De Monés E, Mamelle G, Temam S. Reconstruction of floor of mouth defects by the facial artery musculo‑mucosal flap following cancer ablation. Head Neck. 2008;30(4):437–45. https:// doi. org/ 10. 1002/ hed. 20722. 12. Billon R, Bosc R, Belkacemi Y, Assaf E, SidAhmed‑Mezi M, Hersant B, et al. Impact of adjuvant anti‑ estrogen therapies (tamoxifen and aromatase inhibitors) on perioperative outcomes of breast reconstruction. J Plast Reconstr Aesthet Surg. 2017;70(11):1495–504. https:// doi. org/ 10. 1016/j. bjps. 2017. 05. 046. 13. Parikh RP, Odom EB, Yu L, Colditz GA, Myckatyn TM. Complications and thromboembolic events associated with tamoxifen therapy in patients with breast cancer undergoing microvascular breast reconstruc‑ tion: a systematic review and meta‑analysis. Breast Cancer Res Treat. 2017;163(1):1–10. https:// doi. org/ 10. 1007/ s10549‑ 017‑ 4146‑3. Publisher’s Note Springer Nature remains neutral with regard to jurisdictional claims in pub‑ lished maps and institutional affiliations. Re Read ady y to to submit y submit your our re researc search h ? Choose BMC and benefit fr ? Choose BMC and benefit from om: : fast, convenient online submission thorough peer review by experienced researchers in your field rapid publication on acceptance support for research data, including large and complex data types • gold Open Access which fosters wider collaboration and increased citations maximum visibility for your research: over 100M website views per year At BMC, research is always in progress. Learn more biomedcentral.com/submissions http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png World Journal of Surgical Oncology Springer Journals

Feasibility of dpFAMM flap in tongue reconstruction after facial vessel ligation and radiotherapy—case presentation

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Copyright © The Author(s) 2022
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10.1186/s12957-022-02554-w
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Abstract

Background: Salvage surgery with reconstruction of the second and next primary tongue cancer remains diffi‑ cult, especially after earlier neck dissection and radiotherapy. In the current report, we describe the feasibility of the extended, double‑pedicled facial artery musculomucosal (dpFAMM) flap in the reconstruction of the patient with second primary tongue squamous cell carcinoma, after facial vessel ligation and radiotherapy. Case presentation: An 81‑ year‑ old female patient was operated on due to tongue squamous cell carcinoma (SCC) on the left side T3N1M0 in 2019. Bilateral selective neck dissection with tongue reconstruction was performed by island FAMM flap. The patient also suffered from synchronous mucinous breast carcinoma treated with tamoxifen. The second primary SCC of the tongue on the opposite (right) side was detected in 2020. The patient did not agree to surgical treatment; therefore, radiotherapy was performed. The local recurrence of the tongue cancer of the right side was treated surgically in 2021. Salvage surgery comprised hemiglossectomy and dpFAMM flap reconstruction with uneventful postoperative follow‑up. Conclusions: This case presentation proved that dpFAMM flap can be used in salvage surgery and reconstruction even in patients after ligation of facial vessels, irradiation, and in the course of hormone therapy. The flap is easy to handle, has good vascularity, and comprises a predictable method of reconstruction, especially for patients with severe comorbidities. Keywords: FAMM flap, Facial artery musculomucosal flap, Bozola flap, Tongue cancer, Reconstruction, Salvage surgery, Buccinator myomucosal flap, Tongue reconstruction, Tongue squamous cell carcinoma, dpFAMM flap Background of the tongue might be reconstructed by local flaps, such The incidence of synchronous and metachronous sec - as the facial artery musculomucosal (FAMM) flap and ond oral cancer is increasing due to a longer lifespan and its modifications [2–7]. The FAMM flap is an axial flap improvement of oncological therapy [1]. However, surgi- based on the facial artery and is useless in case of facial cal treatment and reconstruction of second and next pri- vessel ligation. In such cases, the reconstruction should mary cancer in the oral cavity remain difficult, especially be converted to a Bozola flap [8]. Extended, double- after previous neck dissection and radiotherapy. Defects pedicled FAMM (dpFAMM) flap owns modification of FAMM flap with facial and buccal vessel blood supply [3]. In the current report, we describe the feasibility of the *Correspondence: mgontarz@op.pl dpFAMM flap in the reconstruction in the patient with Department of Cranio‑Maxillofacial Surgery, Jagiellonian University second primary tongue squamous cell carcinoma, after Medical College, University Hospital, Jakubowskiego 2 Street, 30‑688 Cracow, Poland facial vessel ligation and radiotherapy. © The Author(s) 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. The Creative Commons Public Domain Dedication waiver (http:// creat iveco mmons. org/ publi cdoma in/ zero/1. 0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Gontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 2 of 6 Case presentation In November 2019, an 81-year-old female patient was admitted to the Department of Cranio-Maxillofacial Surgery of the Jagiellonian University in Cracow due to tongue squamous cell carcinoma (SCC) on the left side cT3N1M0. Clinical examination and computed tomog- raphy (CT) revealed a synchronous left breast tumor (Fig. 1). Biopsy from the breast tumor revealed mucinous carcinoma, and diagnostic imaging excluded dissemina- tion. After tumor board consultation, we decided to start the treatment with surgical excision of tongue SCC and bilateral neck dissection. Selective bilateral neck dissec- tion (level I–IV ipsilateral and I–III contralateral) was performed by a horizontal neck fold incision. During neck dissection, the facial vessels on the left side were preserved for an island FAMM (iFAMM) flap. How - ever, the facial vessels on the right side were ligated. Tongue cancer was excised with margins control by fro- zen section examination. Tongue reconstruction with an iFAMM flap was performed according to the technique described by Joseph et al. (Figs. 2, 3, and 4) [4]. The heal - ing process was uneventful. Histopathological examina- tion revealed squamous cell carcinoma G1 resected with inadequate distal margin (2mm) and metastasis in one Fig. 2 The harvested trilobed iFAMM flap lymph node (IIa cervical level) on the left side (pT3N1). The patient was qualified for postoperative radiotherapy and radical mastectomy with axillary node dissection. from the ulceration confirmed SCC G2. Clinical exami - However, the patient did not give her consent for the pro- nation and CT revealed only local advancement of the posed treatment. disease cT2N0M0. Breast cancer disease was stable. We In February 2020, the patient started hormone therapy proposed surgical treatment, but the patient again did with tamoxifen due to breast cancer and had oncologi- not give her consent. For that reason, the patient was cal controls in our outpatient department every month. In December 2020, after 4 months from the last visit, the patient was admitted to the outpatient clinic with suspi- cion of tongue cancer on the opposite, right side. Biopsy Fig. 1 CT axial view of the chest showing huge breast cancer on the Fig. 3 Transposition of the iFAMM flap with the facial vessel pedicle left side over the mandible G ontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 3 of 6 Fig. 5 CT with 3D reconstruction showing stump of ligated facial artery on the right side (blue arrow), preserved buccal artery on the right side (green arrow), and absence of the facial vessels after iFAMM harvesting on the left side (red arrow) earlier (Figs.  7 and 8) [3]. Histopathological examination Fig. 4 Immediate postoperative result of tongue reconstruction with revealed SCC G2 resected with adequate margins >5mm iFAMM flap (pT3). The healing process was uneventful (Fig.  9). Unfor- tunately, in August 2021, we observed enlarged cervical lymph nodes on the left side. Neck dissection in levels II and V was performed. Histopathological examination qualified for definitive radiotherapy. However, the total dose was reduced due to general health conditions. The patient received 30 Gy in 10 fractions on the second pri- mary and cervical region, and the treatment was finished in February 2021. In May 2021, 2 months after the last visit, the patient was admitted to our outpatient depart- ment with dysphagia and odynophagia. Clinical examina- tion and biopsy confirmed local recurrence of the tongue cancer on the right side. CT showed local recurrence with dimensions 21 × 42 × 29 mm and without suspicious neck lymph nodes (rT3N0M0) (Figs.  5 and 6). Definitive radiotherapy was impossible at this stage of the disease. The patients agreed to salvage surgery, hemiglossectomy with reconstruction and without neck dissection. Due to the fact that the right facial vessels were ligated in November 2019 and considering the extent of the defect, iFAMM was not possible. Also, the general condition of the patient was a contraindication to extensive, long pro- cedures such as free flap reconstruction. Another con - traindication for free flap reconstruction was a higher risk of early postoperative thrombosis of microvascular anastomoses due to continuous tamoxifen intake. Based Fig. 6 CT axial view of extensive local recurrence after radiotherapy on CT evaluation, we decided to use a dpFAMM flap of the tongue SCC on the right side (red arrow) for reconstruction, which was performed as described Gontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 4 of 6 confirmed three metastatic lymph nodes. The patient was disqualified from adjuvant radiotherapy and chemother - apy. The patient died 2 months following neck dissection due to pneumonia. Discussion and conclusions Soft tissue defects of the oral cavity are challenging in reconstructive surgery due to loss of motility, secretion, and sensory functions of the mucous membrane. Recon- struction especially of tongue defects should ensure proper patient’s speech and swallowing. Skin grafts are ineffective in the case of bone exposure and have a ten - Fig. 7 Salvage surgery. Surgical specimen of the tongue after right hemiglossectomy dency to keratinization and cicatrization, which addi- tionally decreases the movability of the remaining, healthy oral mucosa [8]. Also, most pedicled regional and free flaps containing skin islands are characterized by impaired sensitivity, keratinization, hair growth, and donor site morbidity. Local musculomucosal flaps are a good method of reconstruction of moderate tongue defects [3]. According to Massarelli et  al. [8], all buc- cinator musculomucosal flaps provide proper mucus secretion and sensitivity without shrinking tendency, especially after radiotherapy. The iFAMM flap in the tongue reconstruction is pos - sible in case of facial vessel preservation. Massarelli et al. [8] suggest that iFAMM flap pedicled solely on the facial artery with the surrounding fat tissue provides the cor- rect venous drainage, without flap congestion. However, Rahpeyma et  al. [9] in experimental studies on dogs observed iFAMM flap loss in each case of facial vein ligation. For that reason, iFAMM flap, pedicled only on the facial artery, is not acceptable for clinical usage [9]. Fig. 8 Immediate postoperative result of tongue reconstruction with dpFAMM flap In our case, the patient had facial vein and artery ligated during neck dissection  18 months earlier. This was the main contraindication for tongue reconstruction by iFAMM. However, the vascularization of the buccinator muscle is derived from the branches of the facial vessel in the anterior part and buccal vessels in the posterior region. Due to the fact that CT revealed preserved distal part of the facial artery in the buccal region (Fig.  5) and a large number of anastomoses between facial and buc- cal angiosome, which ensures good blood supply through the buccal vessels, we decided to used dpFAMM flap for this reconstruction [8]. The dpFAMM flap combines the advantages of both the FAMM and the Bozola flap, which allows its extension to be increased with sufficient venous drainage [3]. Another issue concerning reconstruction is preopera- tive radiotherapy. It should be pointed out that both the donor and recipient sites comprise the irradiation field, Fig. 9 Final result. Three months after tongue reconstruction with which implies a higher risk of healing process problems dpFAMM flap (green arrow) and iFAMM flap (blue arrow—21 months with potential flap necrosis. O’Leary and Bundgaard [10] after surgery) suggested that the FAMM flap is not suitable in patients, G ontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 5 of 6 Abbreviations who underwent the previous radiotherapy, due to the FAMM: Facial artery musculomucosal flap; dpFAMM: Double ‑pedicled facial risk of such complications as trismus, bleeding, osteo- artery musculomucosal flap; iFAMM: Island facial artery musculomucosal flap; radionecrosis, and impaired healing followed by flap SCC: Squamous cell carcinoma; CT: Computer tomography. necrosis. They observed partial flap necrosis in 75% (3 Acknowledgements from 4 patients) of cases. On the other hand, Ayad et al. We thank Dr. Krzysztof Śliwiński for technical assistance in clinical imaging. [11] did not notice the specific complication rate in the Authors’ contributions group of 10 patients previously irradiated. In our case, MG and GWP designed the study and drafted the manuscript. MG, JB, and KG we also did not detect problems with healing, bleeding, reviewed this case presentation and checked the patient’s treatments. MG, TM, and proper mouth opening. The short term of the follow- PS, and JZ analyzed the data and provided the clinical image and information. MG, JZ, and GWP developed the concept and edited the paper. The authors up did not allow for evaluation of possible mandibular have read and approved the final manuscript. osteoradionecrosis. The healing process can also be disturbed by hor- Funding None. mone therapy. According to Billon et  al. [12], hor- mone therapy, including tamoxifen intake, seems Availability of data and materials to be associated with a higher risk of postoperative All data during the study are included within the article. wound healing complications in patients with breast reconstruction. In addition, Parikh et  al. [13] in their Declarations meta-analysis concluded that perioperative tamox- Ethics approval and consent to participate ifen therapy may increase the risk of thrombotic flap Written informed consent was obtained from the patient prior to submission complications and flap loss in patients undergoing free of the case report. The study was conducted according to the guidelines of the Declaration of Helsinki and approved by the Institutional Review Board flap reconstruction due to breast cancer. They sug- of the Jagiellonian University (No: 1072.6120.229.2021, date of approval is on gested that short cessation of the tamoxifen therapy, September 29, 2021). about 4 weeks prior to reconstructive treatment, might Consent for publication decrease the risk of complications [13]. Our patient The patient has given her consent for the case report to be published. also suffered from synchronous mucinous carcinoma of the left breast treated with tamoxifen 15 months Competing interests The authors declare that they have no competing interests. before salvage surgery. This was one of the contrain- dications for free radial forearm flap application and Received: 3 November 2021 Accepted: 7 March 2022 choice of dpFAMM flap for tongue reconstruction. Transient discontinuation of tamoxifen before surgery was not recommended. However, the healing process was uneventful. References 1. Qaisi M, Vorrasi J, Lubek J, Ord R. Multiple primary squamous cell carcino‑ The extension of the dpFAMM flap in the anterior part mas of the oral cavity. J Oral Maxillofac Surg. 2014;72(8):1511–6. https:// of the buccinator muscle provided an ability for tongue doi. org/ 10. 1016/j. joms. 2014. 03. 012. reconstruction, even after hemiglossectomy. If the apex 2. Duranceau M, Ayad T. The facial artery musculomucosal flap: modification of the harvesting technique for a single‑stage procedure. Laryngoscope. of the tongue can be preserved during ablative surgery, 2011;121:2586–9. like in this case, the dpFAMM flap can be doubled. The 3. Gontarz M, Bargiel J, Gąsiorowski K, Marecik T, Szczurowski P, Zapała J, preserved apex of the tongue is rotated backward to et al. Extended, double‑pedicled facial artery musculomucosal (dpFAMM) flap in tongue reconstruction in edentulous patients: preliminary report obtain the best clinical result of the reconstruction. Flap and flap design. Medicina (Kaunas). 2021;57(8):758. https:// doi. org/ 10. harvesting takes around 30–50 min and can be done 3390/ medic ina57 080758. by one surgical team. The dpFAMM flap has a perfect 4. Joseph ST, Naveen BS, Mohan TM. Islanded facial artery musculomucosal flap for tongue reconstruction. Int J Oral Maxillofac Surg. 2017;46:453–5. color and structure, matching the surrounding tissues 5. Zhao Z, Zhang Z, Li Y, Li S, Xiao S, Fan X, et al. The buccinator muscu‑ without additional extraoral scars. This case presenta - lomucosal island flap for partial tongue reconstruction. J Am Coll Surg. tion only highlights that the dpFAMM flap can be used 2003;196:753–60. 6. Massarelli O, Gobbi R, Raho MT, Tullio A. Three‑ dimensional primary for reconstruction in salvage surgery, even, if the facial reconstruction of anterior mouth floor and ventral tongue using the vessels had been ligated previously and the patient was ‘trilobed’ buccinator myomucosal island flap. Int J Oral Maxillofac Surg. irradiated and treated with hormone therapy. However, 2008;37(10):917–22. https:// doi. org/ 10. 1016/j. ijom. 2008. 07. 020. 7. Massarelli O, Gobbi R, Soma D, Tullio A. The folded tunnelized‑facial artery further studies with more patients must be conducted myomucosal island flap: a new technique for total soft palate reconstruc‑ to prove the utility of the dpFAMM flap in such cases. tion. J Oral Maxillofac Surg. 2013;71(1):192–8. https:// doi. org/ 10. 1016/j. Other advantages of the dpFAMM flap include its feasi - joms. 2012. 03. 030. 8. Massarelli O, Baj A, Gobbi R, Soma D, Marelli S, De Riu G, et al. Cheek bility in the reconstruction of other areas, like the floor of mucosa: a versatile donor site of myomucosal flaps. Technical and func‑ the mouth, the alveolar ridge, the soft and hard palates, tional considerations. Head Neck. 2013;35(1):109–17. https:// doi. org/ 10. or the oropharynx. 1002/ hed. 22933. Gontarz et al. World Journal of Surgical Oncology (2022) 20:81 Page 6 of 6 9. Rahpeyma A, Khajehahmadi S, Sedigh HS. Facial artery myomucosal flap, pedicled solely on the facial artery: experimental design study on survival. J Craniofac Surg. 2016;27(7):e614–5. https:// doi. org/ 10. 1097/ SCS. 00000 00000 002947. 10. O’Leary P, Bundgaard T. Good results in patients with defects after intraoral tumour excision using facial artery musculo‑mucosal flap. Dan Med Bull. 2011;58(5):A4264. 11. Ayad T, Kolb F, De Monés E, Mamelle G, Temam S. Reconstruction of floor of mouth defects by the facial artery musculo‑mucosal flap following cancer ablation. Head Neck. 2008;30(4):437–45. https:// doi. org/ 10. 1002/ hed. 20722. 12. Billon R, Bosc R, Belkacemi Y, Assaf E, SidAhmed‑Mezi M, Hersant B, et al. Impact of adjuvant anti‑ estrogen therapies (tamoxifen and aromatase inhibitors) on perioperative outcomes of breast reconstruction. J Plast Reconstr Aesthet Surg. 2017;70(11):1495–504. https:// doi. org/ 10. 1016/j. bjps. 2017. 05. 046. 13. Parikh RP, Odom EB, Yu L, Colditz GA, Myckatyn TM. Complications and thromboembolic events associated with tamoxifen therapy in patients with breast cancer undergoing microvascular breast reconstruc‑ tion: a systematic review and meta‑analysis. Breast Cancer Res Treat. 2017;163(1):1–10. https:// doi. org/ 10. 1007/ s10549‑ 017‑ 4146‑3. Publisher’s Note Springer Nature remains neutral with regard to jurisdictional claims in pub‑ lished maps and institutional affiliations. Re Read ady y to to submit y submit your our re researc search h ? Choose BMC and benefit fr ? 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Journal

World Journal of Surgical OncologySpringer Journals

Published: Mar 12, 2022

Keywords: FAMM flap; Facial artery musculomucosal flap; Bozola flap; Tongue cancer; Reconstruction; Salvage surgery; Buccinator myomucosal flap; Tongue reconstruction; Tongue squamous cell carcinoma; dpFAMM flap

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