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  • 1. Aims and Scope

    Gut and Liver is an international journal of gastroenterology, focusing on the gastrointestinal tract, liver, biliary tree, pancreas, motility, and neurogastroenterology. Gut atnd Liver delivers up-to-date, authoritative papers on both clinical and research-based topics in gastroenterology. The Journal publishes original articles, case reports, brief communications, letters to the editor and invited review articles in the field of gastroenterology. The Journal is operated by internationally renowned editorial boards and designed to provide a global opportunity to promote academic developments in the field of gastroenterology and hepatology. +MORE

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    Yong Chan Lee Professor of Medicine
    Director, Gastrointestinal Research Laboratory
    Veterans Affairs Medical Center, Univ. California San Francisco
    San Francisco, USA

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    Deputy Editor
    Jong Pil Im Seoul National University College of Medicine, Seoul, Korea
    Robert S. Bresalier University of Texas M. D. Anderson Cancer Center, Houston, USA
    Steven H. Itzkowitz Mount Sinai Medical Center, NY, USA
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    All papers submitted to Gut and Liver are reviewed by the editorial team before being sent out for an external peer review to rule out papers that have low priority, insufficient originality, scientific flaws, or the absence of a message of importance to the readers of the Journal. A decision about these papers will usually be made within two or three weeks.
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Case Report

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Metastasectomy for Hepatic Metastases from Adenoid Cystic Carcinoma of the Trachea

Inkeun Park*, Sung-Nam Lim*, Dok Hyun Yoon*, Hyunjoo Park*, Byungjoo Sun*, Pil Hyung Lee*, Ilseon Hwang, and Dae Ho Lee*

*Department of Oncology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.

Department of Pathology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.

Correspondence to: Dae Ho Lee. Department of Oncology, University of Ulsan College of Medicine, Asan Medical Center, 388-1, Pungnap 2-dong, Songpagu, Seoul 138-736, Korea. Tel: +82-2-3010-3214, Fax: +82-2-3010-6961, leedaeho@amc.seoul.kr

Received: August 19, 2008; Accepted: January 30, 2009

Gut Liver 2009;3(2):127-129

Published online June 30, 2009, Published Date June 30, 2009 https://doi.org/10.5009/gnl.2009.3.2.127

Copyright © Gut and Liver.

The role of metastasectomy for recurrent disease in patients with adenoid cystic carcinoma (ACC) is not defined clearly yet. A 52-year-old woman found two hepatic metastatic nodules 3 years after the completion of treatment for primary ACC of the trachea. After confirming the absence of other lesions, metastasectomy was performed on the two metastatic nodules. Regular follow-up for more than 24 months showed no evidence of recurrent disease after the hepatic metastasectomy. Therefore, we suggest metastasectomy as an option for certain cases of metastatic ACC.

Keywords: Adenoid cystic carcinoma, Trachea, Metastasectomy

Adenoid cystic carcinomas (ACCs) are rare tumors, and mostly arise from salivary gland, especially minor salivary glands.1 They can also arise in various sites of the head and neck including the tracheobronchial tree and even in sites outside the head and neck, such as breast, uterine cervix, prostate, and the skin.2 They have "paradoxical" behaviors. They grow slowly but have relentless and progressive clinical course. They are usually feasible for surgical treatment at presentation but recur inevitably even after many years after complete resection. They seldom metastasize to regional lymph node but often metastasize to distant sites such as lung and bone. Therefore, 5-year survival rates are high but 10- to 20-year survival rates are dismally low.3 If metastasis occurs, watchful waiting, radiation therapy, palliative surgery, and systemic chemotherapy would be treatment options, but no standard palliative treatment modalities are exist.4 Despite the role of metastasectomy to control metastatic tumors in patients with different primary tumors of ACCs is uncertain and controversial, there are some series or anecdotal experiences of promising results of metastasectomy of pulmonary metastatic tumors or of hepatic metastatic tumors.5-9 We here present one case of metastasectomy of hepatic metastatic tumors of ACC of the trachea, in which the patient lives for more than 24 months without the evidence of progressive disease or further recurrence.

A 52-year-old woman came to Oncology Clinic to consult treatment of hepatic metastatic tumors. She had undergone laryngotrachectomy and thyoridectomy with mediastinal tracheostomy for ACC of the trachea followed by radiotherapy for residual tumors at surgical resection margin 3 years before the consultation. The pathology of the resected tumors, measuring 7×4×3 cm from the just below level of vocal cord to tracheal ring, had been reported to be ACC with predominantly tubular histology, extending to subglottic laryngeal wall and thyroid gland without lymphovascular tumor emboli. The tumor cells had uniform round-to-angulated basophilic nuclei, and did not demonstate notable pleomorphism or mitotic activity. They formed tubular structures with microcysts in the cord, with surrounding hyalinized stroma (Fig. 1). Although she had to undergo the radiotherapy for residual tumors, no distant metastasis had been identified on whole body positron emission tomography (PET) and bone scan at the time of the initial operation.

Her chest CT showed two nodules in the segment VII and VIII of liver (Fig. 2) while her abdomen/pelvis CT and whole body PET-CT showed no other metastatic lesions except the above two metastatic nodules (Fig. 3). Her performance status was good with ECOG performance status of 0 and her liver function test as well as other organ function tests was normal. She underwent the right lobectomy of the liver as metastasectomy. The final pathology confirmed that there were the two tumors only, measuring 5×4×4 cm in the segment VII and 3×3×2 cm in the segment VIII, respectively and there was no remnant tumor. The pathology of hepatic tumors was the same to that of the prior tumor of the trachea. No lymphovascular invasion and perineural invasion was identified in the metastatic tumors as well (Fig. 4). To date, she has been followed up regularly without evidence of progressive disease or further recurrence for more than 24 months.

The role of metastasectomy in ACCs is still debatable, but sometimes suggestive in a selected subset of patients. Usually, metastasectomy is often undergone when all metastatic tumors are potentially completely resectable under the control of the primary site and patients have an adequate performance status and a good cardiopulmonary function. Of interest, low tumor burden or less than 6 metastatic tumors and favorable disease-free interval of more than 3 years were sometimes suggested to be selection criteria for pulmonary metastasectomy of ACC.7 The lack of systemic therapeutic alternatives is also suggested one of important selection criteria. In the current case, she had disease-free interval of 3 years, potentially resectable 2 metastatic nodules, and good performance status and residual liver function as well as cardiopulmonary function.

Some argue that as tumor growth of metastatic tumors of ACCs is slow, watchful waiting can be more appropriate, especially when there are no or few symptoms. However, although metastatic tumors grow optimistically slowly, they have relentless and progressive disease course, and long-term outcomes are dismal. Whereas, the response rates for cytotoxic chemotherapy are extremely variable and inconsistent, and response duration was generally short.10 In addition, disease stabilization is more often seen after cytotoxic chemotherapy than objective response but it should be interpreted very cautiously because the ACC, as mentioned above, usually grow slowly. Therefore, systemic cytotoxic chemotherapy should be reserved for those who have symptomatic and rapidly progressive disease. If recurrent or metastatic tumors are totally resectable with conserving organ functions, metastasectomy could be considered as one of the treatment options, such as cytotoxic chemotherapy or watchful waiting.

In conclusion, metastasectomy in patients with ACCs can be resorted to in a very highly selected subset of patients, who have potentially completely resectable metastatic tumors under the control of primary tumors and adequate performance and organ functions.

Fig. 1.Pathology of the primary tumor. The tumor cells had uniform round-to-angulated basophilic nuclei, and did not demonstrate notable pleomorphism or mitotic activity. They appeared as tubular structures with microcysts in the cord, with surrounding hyalinized stroma. The tumor showed a mixed (A) but predominantly tubular (B) pattern (A, H&E stain, ×40; B, H&E stain, ×200).
Fig. 2.CT scan of the upper abdomen. There were two masses in liver segments VII and VIII. Segment VIII contained a poorly enhanced, round, 3-cm mass, and segment VII contained a peripherally enhanced, round, 4.5-cm cystic mass.
Fig. 3.Whole PET scan. Two hypermetabolic lesions were present in the liver (solid arrow and arrowhead).
Fig. 4.Metastatic adenoid cystic carcinoma (ACC) in the liver. (A) Gross findings showing two lesions. (B) Microscopic findings showing a metastatic ACC (H&E stain, ×100).
  1. Bradley, PJ. Adenoid cystic carcinoma of the head and neck: a review. Curr Opin Otolaryngol Head Neck Surg, 2004;12;127-132.
    Pubmed
  2. Spiro, RH, Huvos, AG. Stage means more than grade in adenoid cystic carcinoma. Am J Surg, 1992;164;623-628.
    Pubmed
  3. Westra, WH. The surgical pathology of salivary gland neoplasms. Otolaryngol Clin North Am, 1999;32;919-943.
    Pubmed
  4. Laurie, SA, Licitra, L. Systemic therapy in the palliative management of advanced salivary gland cancers. J Clin Oncol, 2006;24;2673-2678.
    Pubmed
  5. Bobbio, A, Copelli, C, Ampollini, L, et al. Lung metastasis resection of adenoid cystic carcinoma of salivary glands. Eur J Cardiothorac Surg, 2008;33;790-793.
    Pubmed
  6. Liu, D, Labow, DM, Dang, N, et al. Pulmonary metastasectomy for head and neck cancers. Ann Surg Oncol, 1999;6;572-578.
    Pubmed
  7. Locati, LD, Guzzo, M, Bossi, P, et al. Lung metastasectomy in adenoid cystic carcinoma (ACC) of salivary gland. Oral Oncol, 2005;41;890-894.
    Pubmed
  8. Qureshi, SS, Nadkarni, MS, Shrikhande, SV, et al. Hepatic resection for metastasis from adenoid cystic carcinoma of parotid gland. Indian J Gastroenterol, 2005;24;29-30.
    Pubmed
  9. Zeidan, BA, Hilal, MA, Al-Gholmy, M, El-Mahallawi, H, Pearce, NW, Primrose, JN. Adenoid cystic carcinoma of the lacrimal gland metastasising to the liver: report of a case. World J Surg Oncol, 2006;4;66.
    Pubmed
  10. Dodd, RL, Slevin, NJ. Salivary gland adenoid cystic carcinoma: a review of chemotherapy and molecular therapies. Oral Oncol, 2006;42;759-769.
    Pubmed

Article

Case Report

Gut Liver 2009; 3(2): 127-129

Published online June 30, 2009 https://doi.org/10.5009/gnl.2009.3.2.127

Copyright © Gut and Liver.

Metastasectomy for Hepatic Metastases from Adenoid Cystic Carcinoma of the Trachea

Inkeun Park*, Sung-Nam Lim*, Dok Hyun Yoon*, Hyunjoo Park*, Byungjoo Sun*, Pil Hyung Lee*, Ilseon Hwang, and Dae Ho Lee*

*Department of Oncology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.

Department of Pathology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.

Correspondence to: Dae Ho Lee. Department of Oncology, University of Ulsan College of Medicine, Asan Medical Center, 388-1, Pungnap 2-dong, Songpagu, Seoul 138-736, Korea. Tel: +82-2-3010-3214, Fax: +82-2-3010-6961, leedaeho@amc.seoul.kr

Received: August 19, 2008; Accepted: January 30, 2009

Abstract

The role of metastasectomy for recurrent disease in patients with adenoid cystic carcinoma (ACC) is not defined clearly yet. A 52-year-old woman found two hepatic metastatic nodules 3 years after the completion of treatment for primary ACC of the trachea. After confirming the absence of other lesions, metastasectomy was performed on the two metastatic nodules. Regular follow-up for more than 24 months showed no evidence of recurrent disease after the hepatic metastasectomy. Therefore, we suggest metastasectomy as an option for certain cases of metastatic ACC.

Keywords: Adenoid cystic carcinoma, Trachea, Metastasectomy

INTRODUCTION

Adenoid cystic carcinomas (ACCs) are rare tumors, and mostly arise from salivary gland, especially minor salivary glands.1 They can also arise in various sites of the head and neck including the tracheobronchial tree and even in sites outside the head and neck, such as breast, uterine cervix, prostate, and the skin.2 They have "paradoxical" behaviors. They grow slowly but have relentless and progressive clinical course. They are usually feasible for surgical treatment at presentation but recur inevitably even after many years after complete resection. They seldom metastasize to regional lymph node but often metastasize to distant sites such as lung and bone. Therefore, 5-year survival rates are high but 10- to 20-year survival rates are dismally low.3 If metastasis occurs, watchful waiting, radiation therapy, palliative surgery, and systemic chemotherapy would be treatment options, but no standard palliative treatment modalities are exist.4 Despite the role of metastasectomy to control metastatic tumors in patients with different primary tumors of ACCs is uncertain and controversial, there are some series or anecdotal experiences of promising results of metastasectomy of pulmonary metastatic tumors or of hepatic metastatic tumors.5-9 We here present one case of metastasectomy of hepatic metastatic tumors of ACC of the trachea, in which the patient lives for more than 24 months without the evidence of progressive disease or further recurrence.

CASE REPORT

A 52-year-old woman came to Oncology Clinic to consult treatment of hepatic metastatic tumors. She had undergone laryngotrachectomy and thyoridectomy with mediastinal tracheostomy for ACC of the trachea followed by radiotherapy for residual tumors at surgical resection margin 3 years before the consultation. The pathology of the resected tumors, measuring 7×4×3 cm from the just below level of vocal cord to tracheal ring, had been reported to be ACC with predominantly tubular histology, extending to subglottic laryngeal wall and thyroid gland without lymphovascular tumor emboli. The tumor cells had uniform round-to-angulated basophilic nuclei, and did not demonstate notable pleomorphism or mitotic activity. They formed tubular structures with microcysts in the cord, with surrounding hyalinized stroma (Fig. 1). Although she had to undergo the radiotherapy for residual tumors, no distant metastasis had been identified on whole body positron emission tomography (PET) and bone scan at the time of the initial operation.

Her chest CT showed two nodules in the segment VII and VIII of liver (Fig. 2) while her abdomen/pelvis CT and whole body PET-CT showed no other metastatic lesions except the above two metastatic nodules (Fig. 3). Her performance status was good with ECOG performance status of 0 and her liver function test as well as other organ function tests was normal. She underwent the right lobectomy of the liver as metastasectomy. The final pathology confirmed that there were the two tumors only, measuring 5×4×4 cm in the segment VII and 3×3×2 cm in the segment VIII, respectively and there was no remnant tumor. The pathology of hepatic tumors was the same to that of the prior tumor of the trachea. No lymphovascular invasion and perineural invasion was identified in the metastatic tumors as well (Fig. 4). To date, she has been followed up regularly without evidence of progressive disease or further recurrence for more than 24 months.

DISCUSSION

The role of metastasectomy in ACCs is still debatable, but sometimes suggestive in a selected subset of patients. Usually, metastasectomy is often undergone when all metastatic tumors are potentially completely resectable under the control of the primary site and patients have an adequate performance status and a good cardiopulmonary function. Of interest, low tumor burden or less than 6 metastatic tumors and favorable disease-free interval of more than 3 years were sometimes suggested to be selection criteria for pulmonary metastasectomy of ACC.7 The lack of systemic therapeutic alternatives is also suggested one of important selection criteria. In the current case, she had disease-free interval of 3 years, potentially resectable 2 metastatic nodules, and good performance status and residual liver function as well as cardiopulmonary function.

Some argue that as tumor growth of metastatic tumors of ACCs is slow, watchful waiting can be more appropriate, especially when there are no or few symptoms. However, although metastatic tumors grow optimistically slowly, they have relentless and progressive disease course, and long-term outcomes are dismal. Whereas, the response rates for cytotoxic chemotherapy are extremely variable and inconsistent, and response duration was generally short.10 In addition, disease stabilization is more often seen after cytotoxic chemotherapy than objective response but it should be interpreted very cautiously because the ACC, as mentioned above, usually grow slowly. Therefore, systemic cytotoxic chemotherapy should be reserved for those who have symptomatic and rapidly progressive disease. If recurrent or metastatic tumors are totally resectable with conserving organ functions, metastasectomy could be considered as one of the treatment options, such as cytotoxic chemotherapy or watchful waiting.

In conclusion, metastasectomy in patients with ACCs can be resorted to in a very highly selected subset of patients, who have potentially completely resectable metastatic tumors under the control of primary tumors and adequate performance and organ functions.

Fig 1.

Figure 1.Pathology of the primary tumor. The tumor cells had uniform round-to-angulated basophilic nuclei, and did not demonstrate notable pleomorphism or mitotic activity. They appeared as tubular structures with microcysts in the cord, with surrounding hyalinized stroma. The tumor showed a mixed (A) but predominantly tubular (B) pattern (A, H&E stain, ×40; B, H&E stain, ×200).
Gut and Liver 2009; 3: 127-129https://doi.org/10.5009/gnl.2009.3.2.127

Fig 2.

Figure 2.CT scan of the upper abdomen. There were two masses in liver segments VII and VIII. Segment VIII contained a poorly enhanced, round, 3-cm mass, and segment VII contained a peripherally enhanced, round, 4.5-cm cystic mass.
Gut and Liver 2009; 3: 127-129https://doi.org/10.5009/gnl.2009.3.2.127

Fig 3.

Figure 3.Whole PET scan. Two hypermetabolic lesions were present in the liver (solid arrow and arrowhead).
Gut and Liver 2009; 3: 127-129https://doi.org/10.5009/gnl.2009.3.2.127

Fig 4.

Figure 4.Metastatic adenoid cystic carcinoma (ACC) in the liver. (A) Gross findings showing two lesions. (B) Microscopic findings showing a metastatic ACC (H&E stain, ×100).
Gut and Liver 2009; 3: 127-129https://doi.org/10.5009/gnl.2009.3.2.127

References

  1. Bradley, PJ. Adenoid cystic carcinoma of the head and neck: a review. Curr Opin Otolaryngol Head Neck Surg, 2004;12;127-132.
    Pubmed
  2. Spiro, RH, Huvos, AG. Stage means more than grade in adenoid cystic carcinoma. Am J Surg, 1992;164;623-628.
    Pubmed
  3. Westra, WH. The surgical pathology of salivary gland neoplasms. Otolaryngol Clin North Am, 1999;32;919-943.
    Pubmed
  4. Laurie, SA, Licitra, L. Systemic therapy in the palliative management of advanced salivary gland cancers. J Clin Oncol, 2006;24;2673-2678.
    Pubmed
  5. Bobbio, A, Copelli, C, Ampollini, L, et al. Lung metastasis resection of adenoid cystic carcinoma of salivary glands. Eur J Cardiothorac Surg, 2008;33;790-793.
    Pubmed
  6. Liu, D, Labow, DM, Dang, N, et al. Pulmonary metastasectomy for head and neck cancers. Ann Surg Oncol, 1999;6;572-578.
    Pubmed
  7. Locati, LD, Guzzo, M, Bossi, P, et al. Lung metastasectomy in adenoid cystic carcinoma (ACC) of salivary gland. Oral Oncol, 2005;41;890-894.
    Pubmed
  8. Qureshi, SS, Nadkarni, MS, Shrikhande, SV, et al. Hepatic resection for metastasis from adenoid cystic carcinoma of parotid gland. Indian J Gastroenterol, 2005;24;29-30.
    Pubmed
  9. Zeidan, BA, Hilal, MA, Al-Gholmy, M, El-Mahallawi, H, Pearce, NW, Primrose, JN. Adenoid cystic carcinoma of the lacrimal gland metastasising to the liver: report of a case. World J Surg Oncol, 2006;4;66.
    Pubmed
  10. Dodd, RL, Slevin, NJ. Salivary gland adenoid cystic carcinoma: a review of chemotherapy and molecular therapies. Oral Oncol, 2006;42;759-769.
    Pubmed
Gut and Liver

Vol.16 No.5
September, 2022

pISSN 1976-2283
eISSN 2005-1212

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