Open-access <b>Esophageal leiomyoma</b>: <b>Experience with nine surgical patients</b>

Abstracts

<b>Objective: </b>To inform about the experience acquired operating esophageal leiomyoma in the Thoracic Surgery Department of the Calderón Guardia Hospital. <b>&nbsp;</b> <b>Methods:</b> Fourteen patients with submucosal esophageal tumors were referred to the Thoracic Surgery Department at Hospital Calderón Guardia during the twelve-year period comprised between 1999 and 2011. The approach for four asymptomatic patients with a small mass was observation, and the remaining ten underwent surgical procedures, which confirmed the histologic diagnosis of leiomyoma. Once authorization was granted by the hospital&#8217;s Ethics Committee, clinical records were reviewed, considering personal characteristics, symptoms, methods for diagnosis, surgical treatment and follow-up for the patients involved. <b>&nbsp;</b> <b>Results:</b> Ten surgical patients were analyzed, eight were male and two female. Their age ranged from 38 to 71 years, with a mean of 56 years. Their primary complaint was dysphagia, but in three cases the tumor findings were incidental, while performing a routine upper endoscopy. In all cases the method used for detection was an upper endoscopy describing either a submucosal tumor or an extrinsic compression of the esophageal wall. The endoscopic ultrasound correlated in all cases that the tumor originated from the fourth layer of the esophagus, compatible with leiomyoma. In two cases the possibility of malignancy was questioned due to the dimensions of the lesion. Nine patients were operated through a thoracotomy and one through a laparotomy. In 7 cases enucleating was successful without perforation of the mucosa. In two patients partial esophagectomy with anastomosis and diaphragmatic patch was performed. In an exceptional case, the tumor extended from the cervical esophagus to the esophagogastric union, so a total esophagectomy was done with a gastric interposition. There was no mortality reported. One patient had complications with an small anastomotic leak that was resolved without intervention. Two patients had respiratory complications that prolonged their hospital stay. There has not been any relapse or other complication; the follow up ranges between 8 months and 12 years. <b>&nbsp;</b> <b>Conclusion:</b> The esophageal leiomyoma is an un common pathology. The surgery of submucosal esophageal tumors can be performed without mayor morbidity if there is an adequate preoperative evaluation and management. None of the cases of this series had complications or recurrence to the date the study was made

esophageal leiomyoma; esophagus; tumors; endoscopic ultrasound


<span name="style_bold">Objetivo: </span>informar la experiencia adquirida con la cirugía para leiomiomas esofágicos en el Servicio de Cirugía de Tórax del Hospital Dr. R. A. Calderón Guardia. <span name="style_bold"> </span> <span name="style_bold">Métodos: </span>durante el período de 12 años comprendido entre 1999 y 2011, fueron referidos 14 pacientes con tumores submucosos del esófago al Servicio de Cirugía de Tórax del Hospital Calderón Guardia. En cuatro pacientes asintomáticos con tumores pequeños se decidió observar, y diez fueron operados, confirmándose el diagnóstico histológico de leiomioma. Previa autorización del Comité de Ética del Hospital, se revisaron los expedientes clínicos, analizando las características personales, los síntomas, los métodos de diagnóstico, el tratamiento quirúrgico y la evolución de los pacientes operados. <span name="style_bold"> </span> <span name="style_bold">Resultados: </span>se encontró que de los diez pacientes operados, ocho fueron varones, en edades entre los 38 y 71 años, con un promedio de 56 años. El síntoma principal fue disfagia, pero en 3 casos el tumor fue un hallazgo incidental de una endoscopia de rutina. El método de detección en todos los casos fue la endoscopia en la que se describió un tumor submucoso o una compresión extrínseca de la pared esofágica. El ultrasonido endoscópico se correlacionó con un tumor originado en la cuarta capa, posible leiomioma. En dos casos, debido a las dimensiones de la lesión, se planteó la posibilidad de malignidad. La tumoración se intervino en nueve casos por toracotomía y en uno, por laparotomía. En 7 casos se enucleó sin que se abriera la mucosa, en dos se realizó esofagectomía parcial con anastomosis y parche de diafragma, y en un caso, excepcional, la tumoración se extendía desde el esófago cervical hasta la unión esófago-gástrica, de manera que se realizó una esofagectomía total, con ascenso gástrico y anastomosis cervical. No hubo ningún caso de mortalidad. Un paciente presentó una pequeña fuga de la anastomosis, la cual resolvió espontáneamente. Dos pacientes manifestaron complicaciones respiratorias que prolongaron su estancia. No se han evidenciado recidivas ni otras complicaciones, y el seguimiento de los pacientes oscila entre 8 meses y 12 años. <span name="style_bold"> </span> <span name="style_bold">Conclusión: </span>el leiomioma esofágico es una patología poco frecuente. La cirugía de los tumores submucosos del esófago, cuando se estudian y manejan adecuadamente en el preoperatorio, se puede realizar sin mayor morbilidad; ninguno de los casos de esta serie presentó complicaciones ni recidivas hasta la fecha de la revisiónel leiomioma esofágico es una patología poco frecuente. La cirugía de los tumores submucosos del esófago, cuando se estudian y manejan adecuadamente en el preoperatorio, se puede realizar sin mayor morbilidad; ninguno de los casos de esta serie presentó complicaciones ni recidivas hasta la fecha de la revisión

leiomioma esofágico; esófago; tumores; tumores benignos del esófago; ultrasonido endoscópico


<span name="style_bold">Objective: </span>To inform about the experience acquired operating esophageal leiomyoma in the Thoracic Surgery Department of the Calderón Guardia Hospital. <span name="style_bold"> </span> <span name="style_bold">Methods:</span> Fourteen patients with submucosal esophageal tumors were referred to the Thoracic Surgery Department at Hospital Calderón Guardia during the twelve-year period comprised between 1999 and 2011. The approach for four asymptomatic patients with a small mass was observation, and the remaining ten underwent surgical procedures, which confirmed the histologic diagnosis of leiomyoma. Once authorization was granted by the hospital’s Ethics Committee, clinical records were reviewed, considering personal characteristics, symptoms, methods for diagnosis, surgical treatment and follow-up for the patients involved. <span name="style_bold"> </span> <span name="style_bold">Results:</span> Ten surgical patients were analyzed, eight were male and two female. Their age ranged from 38 to 71 years, with a mean of 56 years. Their primary complaint was dysphagia, but in three cases the tumor findings were incidental, while performing a routine upper endoscopy. In all cases the method used for detection was an upper endoscopy describing either a submucosal tumor or an extrinsic compression of the esophageal wall. The endoscopic ultrasound correlated in all cases that the tumor originated from the fourth layer of the esophagus, compatible with leiomyoma. In two cases the possibility of malignancy was questioned due to the dimensions of the lesion. Nine patients were operated through a thoracotomy and one through a laparotomy. In 7 cases enucleating was successful without perforation of the mucosa. In two patients partial esophagectomy with anastomosis and diaphragmatic patch was performed. In an exceptional case, the tumor extended from the cervical esophagus to the esophagogastric union, so a total esophagectomy was done with a gastric interposition. There was no mortality reported. One patient had complications with an small anastomotic leak that was resolved without intervention. Two patients had respiratory complications that prolonged their hospital stay. There has not been any relapse or other complication; the follow up ranges between 8 months and 12 years. <span name="style_bold"> </span> <span name="style_bold">Conclusion:</span> The esophageal leiomyoma is an un common pathology. The surgery of submucosal esophageal tumors can be performed without mayor morbidity if there is an adequate preoperative evaluation and management. None of the cases of this series had complications or recurrence to the date the study was made The esophageal leiomyoma is an un common pathology. The surgery of submucosal esophageal tumors can be performed without mayor morbidity if there is an adequate preoperative evaluation and management. None of the cases of this series had complications or recurrence to the date the study was made

esophageal leiomyoma; esophagus; tumors; endoscopic ultrasound


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Esophageal

1, José-Alberto Mainieri-Hidalgo2


Thoracic Surgery Department, Dr. Rafael Ángel Calderón Guardia, Caja Costarricense de Seguro Social. Author’s Memberships: 1Gastric Cancer Early Detection Center. Hospital Max Peralta, Caja Costarricense de Seguro Social. 2 Thoracic Surgery Department, Hospital Calderón Guardia, Caja Costarricense de Seguro Social.

Aim: The aim of this study is to inform the experience acquired operating esophageal leiomyoma in the Thoracic Surgery Department of the Hospital Calderón Guardia.

Methods: Fourteen patients with submucosal esophageal tumors were referred to the Thoracic Surgery Department at Hospital Calderón Guardia during the twelve-year period comprised between 1999 and 2011. The approach for four asymptomatic patients with a small mass was

Results: Ten surgical patients were analyzed, eight were male and two female. Their age ranged from 38 to 71 years, with an average of 56 years. Their primary complaint was dysphagia. In three cases the tumor findings were incidental, while performing an upper endoscopy for digestive symptoms. In all cases the method used for detection was an upper endoscopy describing either a submucosal tumor or an extrinsic compression of the esophageal wall. The endoscopic ultrasound correlated in all cases that the tumor originated from the fourth layer of the esophagus, compatible with leiomyoma. In two cases the possibility of malignancy was questioned due to the dimensions of the lesion. Nine patients were operated through a thoracotomy and one through a laparotomy. In 7 cases enucleating was successful without perforation of the mucosa. In two patients partial esophagectomy with anastomosis and diaphragmatic patch was performed. In an exceptional case, the tumor extended from the cervical esophagus to the esophagogastric union, so a total esophagectomy was done with a gastric interposition. There was no mortality reported. One patient complicated with an small anastomotic leak that was resolved without intervention. Two patients had respiratory problems that prolonged their hospital stay. In the 12 years and 8 months of follow up, there has not been any relapse or complication.

Conclusion: The surgery of submucosal esophageal tumors can be performed without mayor morbidity if there is an adequate preoperative evaluation and Management is made. None of the cases of this series had complications or recurrence to the date the study was made.

Key words: esophageal leiomyoma, esophagus, tumors, endoscopic ultrasound.

1

2 Histologically, the tumors are comprised of smooth muscle tangles, well-demarcated by adjacent tissue or by a connective tissue capsule. Macroscopically, well-defined masses are visualized in the esophageal wall; cut surface is solid with a grayish white color. The majority occur as single lesions, of less than 5cm in diameter, but 5% may be multiple, especially patients with Alport’s Syndrome. Large tumors present themselves as posterior mediastinal masses that compressed adjacent organs and may be confused with neoplasias.3 The differential diagnosis is made with esophageal cancer, gastrointestinal stromal tumors (GIST) and other benign esophageal tumors.4 Leiomyomatosis is characterized by diffuse hypertrophy of all muscle layers of the esophagus and the presence of lymphocytic and plasma cell infiltration; usually accompanied by leiomyomas elsewhere, neuropathy, hearing problems, myopia or astigmatism (Alport’s Syndrome). The management in these cases consists of esophageal resection and replacement with the stomach or the colon. 5.7

8

1,3 By endoscopy, a mobile submucosal lesion can be seen, with a intact mucosa. If a leiomyoma is suspected, biopsy by any method should be avoided, the tearing of the mucosa difficult extramucosal resection (enucleation). If ulceration is present or there is suspicion of malignancy, a biopsy should be performed; a useful method is fine needle aspiration biopsy (FNAB). If a biopsy is performed, it is recommended to postponed surgery at least 2 weeks, to allow the esophageal mucosa to heal and diminished the risk of perforation. 9 Endoscopic ultrasound, demonstrate a homogeneous region of juxtaposed hypoechogenicity with the overlying mucosa. The radiologic findings of an

2,3

10 The recommended method of resection is enucleation, preserving the integrity of the mucosa. When the tumor is larger than 8cm, when adhered to the mucosa, or when there has been an extensive tearing during dissection, it may be necessary to remove part of the esophagus. The concomitant use of endoscopy helps to locate the lesion and detect perforations in the esophageal wall.11 In 1992, Everitt reported the first successful thoracoscopic esophageal enucleation, and in 2010, Wang et al reported 12-14 thoracoscopic resection of 42 esophageal leiomyomas or GISTs, with diameters up to 5cm. Thus, concluding that endoscopic surgery can be performed with good results, although there is a greater risk for perforation when comparing to conventional surgery. 15

2 and the described morbidity includes pain, atelectasia and pneumonia. The follow up consists in periodic esophagograms and endoscopies to detect recurrences.

56. In relation to gender, eight were men and two were women. Seven cases were detected at the CancerEarlyDetectionCenter of the Hospital Max Peralta.

table 1, and corresponds to less than one case per year, in a third level national reference center, attending a little more than a third of the adult population.

table 1

Fig. 1a). In one occasion, an ulceration of the mucosa was described.

Fig. 1a

Fig. 2a).

Fig. 2a

Fig. 1b).

Fig. 2b).

14

CancerEarlyDetectionCenter in Cartago. A generated doubt in three cases when the lesion was too large was the possible malignancy; in the department, no cases have been described of esophageal leiomyosarcoma, this lesion is much rarer.

References

References

  • 1. Punpale A, Rangole A, Bhambhani N, Karimundackal G, Desai N, de Souza A, Pramesh CS, Jambhekar N, Mistry R. Leiomyoma of Esophagus. Ann Torac Cardiovasc Surg2007; 13:78-81.

  • 2. Saleh W, Bamosa A, Al-Mutairi H, Al-Kattan K. Thoracoscopic enucleation of esophageal leiomioma in patient with MEN I syndrome. Ann Thoracic Med2010; 5:47-49.

  • 3. Yang P.S., Lee K.S., Lee S.J., Kim T.S., Choo I.W., Shim Y.M., Kim K., Kim Y. Esophageal Leiomyoma: Radiologic Findings in 12 Patients. Korean J Radiol 2001; 2:132-137.

  • 4. Loviscek L.F., Yun J.H., Park Y.S., Chiari A., Grillo C., Cenoz M.C. Leiomioma de esófago. Cir Esp. 2009; 85: 147-51.

  • 5. Boran C., Sengul N., Balaban Y.H., Gürel S. Multinodular leiomyoma of the esophagus with internodular hydropic degeneration and bulbous serosal protrusions similar to cotylednonoid uterine leiomyoma. Diseases of the Esophagus 2007; 20:187-189.

  • 6. Okugawa Y., Mohri Y., Toiyama Y., Yokoe T., Ohi M., Tanaka K., Uchida K., Shiraishi T., Kusunoki M. Multiple Solitary Leiomyomas in the Esophagus: Report of a Case. Surg Today. 2011; 41: 563-567.

  • 7.Obuchi T., Sasaki A., Nitta H., Koeda K., Ikeda K., Wakabayashi G.Minimally invasive surgical enucleation for esophageal leiomyoma: report of seven cases.Diseases of the Esophagus. 2010; 23: E1-E4.

  • 8. Asteriou C., Konstantinou D., Lalountas M., Kleontas A., Setzis K., Zafiriou G., Barbetakis N. Nine year experience in surgical approach of leiomyomatosis of esophagus. World J Surg Oncol 2009; 7: 102.

  • 9. Jiang G., Zhao H., Yang F., Li J., Li Y., Liu Y., Lui J., Wang J. Thoracoscopic enucleation of esophageal leiomyoma: a retrospective study on 40 cases. Diseases of the Esophagus. 2009; 22: 279-283.

  • 10. Schorlemmer G., Battaglini J., Murria G. The Cervical Approach to Esophageal Leiomyomas. Ann Thoracic Surg1983; 35: 469-472.

  • 11. Li Z.G., Chen H.Z., Jin H., Yang L.X., Xu Z.Y., Liu F., Yao F. Surgical treatment of esophageal leiomyoma located near or at the esophagogastric junction via a Thoracoscopic approach. Diseases of the Esophagus2009; 22: 185-189.

  • 12. Zaninotto G., Portale G., Costantini M., Rizzetto C., Salvador R., Rampado S., Pennelli G., Ancona E. Minimally invasive enucleation of esophageal leiomyoma. Surg Endosc 2006; 20: 1904-1908.

  • 13. Dapri G., Himpens J., Ntounda R., Alard S., Dereeper E., Cadiere G.B. Enucleation of a leiomyoma of the mid-esophagus through a right thoracoscopy with the patient in prone position. Surg Endosc. 2010; 24: 215-218.

  • 14.Wang L., Fan C.Q.,Ren W., Zhang X., Li Y.H., Zhao X.Y.Endoscopio dissection of large endogenous myogenic tumors in the esophagus and stomach is safe and feasible: A report of 42 cases. Scandinavian Journal of Gastroenterology.2010; 46:627-633.

  • 15. DeUgarte D., Teitelbaum D., Hirschl R., Geiger J.D. Robotic Extirpation of Complex Massive Esophageal Leiomyoma. J Laparoendoscopic & Advanced Surgical Techniques. 2008; 18: 286-289.

  • 16. Slesser A.A.P., Shaw I. A Large Esophageal Leiomyoma. International Journal of Surgical Pathology2009; 17: 401.

  • 17. Gupta V., Lal A., Sinha S.K., Nada R., GuptaN.M. Leiomyomatosis of the Esophagus: Experience over a Decade. J Gastrointest Surg. 2009; 13: 206-211.

en_bart7
Date Received: June 20th, 2011 Date Accepted: May 28th, 2012

References

  • 1. Punpale A, Rangole A, Bhambhani N, Karimundackal G, Desai N, de Souza A, Pramesh CS, Jambhekar N, Mistry R. Leiomyoma of Esophagus. Ann Torac Cardiovasc Surg2007; 13:78-81.

  • 2. Saleh W, Bamosa A, Al-Mutairi H, Al-Kattan K. Thoracoscopic enucleation of esophageal leiomioma in patient with MEN I syndrome. Ann Thoracic Med2010; 5:47-49.

  • 3. Yang P.S., Lee K.S., Lee S.J., Kim T.S., Choo I.W., Shim Y.M., Kim K., Kim Y. Esophageal Leiomyoma: Radiologic Findings in 12 Patients. Korean J Radiol 2001; 2:132-137.

  • 4. Loviscek L.F., Yun J.H., Park Y.S., Chiari A., Grillo C., Cenoz M.C. Leiomioma de esófago. Cir Esp. 2009; 85: 147-51.

  • 5. Boran C., Sengul N., Balaban Y.H., Gürel S. Multinodular leiomyoma of the esophagus with internodular hydropic degeneration and bulbous serosal protrusions similar to cotylednonoid uterine leiomyoma. Diseases of the Esophagus 2007; 20:187-189.

  • 6. Okugawa Y., Mohri Y., Toiyama Y., Yokoe T., Ohi M., Tanaka K., Uchida K., Shiraishi T., Kusunoki M. Multiple Solitary Leiomyomas in the Esophagus: Report of a Case. Surg Today. 2011; 41: 563-567.

  • 7.Obuchi T., Sasaki A., Nitta H., Koeda K., Ikeda K., Wakabayashi G.Minimally invasive surgical enucleation for esophageal leiomyoma: report of seven cases.Diseases of the Esophagus. 2010; 23: E1-E4.

  • 8. Asteriou C., Konstantinou D., Lalountas M., Kleontas A., Setzis K., Zafiriou G., Barbetakis N. Nine year experience in surgical approach of leiomyomatosis of esophagus. World J Surg Oncol 2009; 7: 102.

  • 9. Jiang G., Zhao H., Yang F., Li J., Li Y., Liu Y., Lui J., Wang J. Thoracoscopic enucleation of esophageal leiomyoma: a retrospective study on 40 cases. Diseases of the Esophagus. 2009; 22: 279-283.

  • 10. Schorlemmer G., Battaglini J., Murria G. The Cervical Approach to Esophageal Leiomyomas. Ann Thoracic Surg1983; 35: 469-472.

  • 11. Li Z.G., Chen H.Z., Jin H., Yang L.X., Xu Z.Y., Liu F., Yao F. Surgical treatment of esophageal leiomyoma located near or at the esophagogastric junction via a Thoracoscopic approach. Diseases of the Esophagus2009; 22: 185-189.

  • 12. Zaninotto G., Portale G., Costantini M., Rizzetto C., Salvador R., Rampado S., Pennelli G., Ancona E. Minimally invasive enucleation of esophageal leiomyoma. Surg Endosc 2006; 20: 1904-1908.

  • 13. Dapri G., Himpens J., Ntounda R., Alard S., Dereeper E., Cadiere G.B. Enucleation of a leiomyoma of the mid-esophagus through a right thoracoscopy with the patient in prone position. Surg Endosc. 2010; 24: 215-218.

  • 14.Wang L., Fan C.Q.,Ren W., Zhang X., Li Y.H., Zhao X.Y.Endoscopio dissection of large endogenous myogenic tumors in the esophagus and stomach is safe and feasible: A report of 42 cases. Scandinavian Journal of Gastroenterology.2010; 46:627-633.

  • 15. DeUgarte D., Teitelbaum D., Hirschl R., Geiger J.D. Robotic Extirpation of Complex Massive Esophageal Leiomyoma. J Laparoendoscopic & Advanced Surgical Techniques. 2008; 18: 286-289.

  • 16. Slesser A.A.P., Shaw I. A Large Esophageal Leiomyoma. International Journal of Surgical Pathology2009; 17: 401.

  • 17. Gupta V., Lal A., Sinha S.K., Nada R., GuptaN.M. Leiomyomatosis of the Esophagus: Experience over a Decade. J Gastrointest Surg. 2009; 13: 206-211.

en_bart7
Date Received: June 20th, 2011 Date Accepted: May 28th, 2012

Publication Dates

  • Publication in this collection
    07 May 2013
  • Date of issue
    Sept 2012

History

  • Received
    20 June 2011
  • Accepted
    28 May 2012
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