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CORRESPONDENCE
Year : 2017  |  Volume : 13  |  Issue : 6  |  Page : 1070-1072

Small bowel and cecal Involvement: Unusual metastasis sites from adenoid cystic carcinoma of the minor salivary gland


1 Department of Medical Oncology, Ankara Numune Training and Research Hospital, Ankara, Turkey
2 Department of Surgery, Ankara Numune Training and Research Hospital, Ankara, Turkey
3 Department of Pathology, Ankara Numune Training and Research Hospital, Ankara, Turkey
4 Department of Radiation Oncology, Ankara Numune Training and Research Hospital, Ankara, Turkey
5 Department of Medical Oncology, Faculty of Medicine, Yıldırım Beyazıt University, Ankara, Turkey

Date of Web Publication13-Dec-2017

Correspondence Address:
Dr. Yakup Bozkaya
Department of Medical Oncology, Ankara Numune Training and Research Hospital, 06100 Sihhiye, Ankara
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.183549

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 > Abstract 

Adenoid cystic carcinoma (ACC) is a rare neoplasm and the most frequent pathology occurring in the minor salivary glands. Lung, bone, liver, and brain are the most frequent metastasis sites in ACC cases. To our knowledge, cecal and ileum metastasis from ACC has not been previously reported in literature. Herein, we reported a case of cecal and small bowel metastasis (ileum) in a patient with ACC. A 41-year-old male patient with ACC developed iron and Vitamin B12 deficiency anemia during further controls. Lower gastrointestinal (GI) endoscopy detected an ulcerated polypoid lesion with raised margins in cecum. Pathological evaluation of polyp biopsy was consistent with metastasis from ACC. The patient underwent surgery of right hemicolectomy. Pathological examinations revealed an ACC of cecum and small bowel (ileum) showing positive staining with CD117, CK7, and panCK. We aimed to emphasize that performing GI stromal screening in ACC patients presenting with iron deficiency is important for early diagnosis of metastasis.

Keywords: Adenoid cystic carcinoma, colon metastasis, distant metastasis, small bowel metastasis


How to cite this article:
Bozkaya Y, Çetinkaya E, Erdem GU, Çiftçi AY, Karahaliloğlu EA, Özdemir N. Small bowel and cecal Involvement: Unusual metastasis sites from adenoid cystic carcinoma of the minor salivary gland. J Can Res Ther 2017;13:1070-2

How to cite this URL:
Bozkaya Y, Çetinkaya E, Erdem GU, Çiftçi AY, Karahaliloğlu EA, Özdemir N. Small bowel and cecal Involvement: Unusual metastasis sites from adenoid cystic carcinoma of the minor salivary gland. J Can Res Ther [serial online] 2017 [cited 2019 Nov 14];13:1070-2. Available from: http://www.cancerjournal.net/text.asp?2017/13/6/1070/183549


 > Introduction Top


Adenoid cystic carcinoma (ACC) is a rare neoplasm and the most frequent pathology occurring in the minor salivary glands.[1] Despite slow-growing pattern, it has a tendency to local recurrence and perineural invasion (PNI). Hematogenous metastasis is more common than lymphogenous metastasis.[1],[2] Delayed distant metastasis may appear in up to 50% of ACC patients during the disease.[3] While lung, bone, liver, and brain are the most frequent metastasis sites in ACC cases, stomach, choroid, kidney, and skin metastases are relatively rare.[1],[4] Cecal and ileum metastasis from ACC has not been previously reported in literature. Herein, we present the first case of cecal and small bowel metastasis (ileum) from ACC to emphasize the rarity of this unexpected metastasis site.


 > Case Report Top


A 41-year-old male patient was admitted to hospital with the complaint of a slowly growing mass on lateral part of the upper lip in December 2008. Histopathological findings of the mass excision showed ACC. After a 5-year follow-up care without treatment, patient represented with the complaints of hemoptysis and chest pain. Thoracic computed tomography demonstrated a solid, lobulated, and irregular mass with peripherally spiculated extensions in size of 57 mm × 47 mm × 42 mm in the left hilar region. Bronchoscopic biopsy of hilar mass was compatible with ACC. The surgical resection was not appropriate for the patient due to the close relationship of the mass with vascular structure. The second primary tumor or any event of metastasis could not be ruled out. Since that the tumor is sole lesion with slow-growing pattern, curative chemoradiotherapy (with etoposide and cisplatin) was planned. No progression was observed in the mass during follow-up. However, iron and Vitamin B12 deficiency anemia were detected during further controls. Lower gastrointestinal (GI) endoscopy detected a ulcerated polypoid lesion (1.5 cm in size) with raised margins in cecum. Pathological evaluation of polyp biopsy was consistent with metastasis from ACC. In the laparoscopic exploration, we detected a mass in excess of serosa at ileum [Figure 1]. We decided right hemicolectomy and ileum resection [Figure 2]. We made extracorporel anastomosis. He tolerated oral intake and he discharged without any complication. Pathological examinations revealed an ACC of cecum and small bowel (ileum) showing positive staining with CD117, CK7, and panCK [Figure 3]. No residual disease was present after surgery and any further treatment was not indicated for the patient since the biological nature of tumor has a slow-growing pattern.
Figure 1: A mass in excess of serosa at ileum

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Figure 2: A specimen covers the ileal and cecum mass

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Figure 3: (a) The view of epithelial tumor in H and E stained sections, showing a tubular, cribriform and solid growth pattern in partly sclerotic and hyaline stroma (b) a malignant epithelial tumor appearance, invading through intestinal mucosa into submucosa (H and E, ×100)

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 > Discussion Top


Although locoregional control is possible in ACC, a delayed distant metastasis may occur during the disease. Therefore, a long-term follow-up and a high clinical suspicion are necessary to diagnose them early. Lung, bone, liver, and brain are the most frequent metastasis sites in ACC cases.[1] Colonic metastases from other solid tumors are uncommon in literature and usually occur secondary to malignant tumors of the stomach, breast, ovary, cervix, kidney, lung, prostate, or skin.[5] Unlike metastasis from other malignancies, ACC exhibits a slow-growing pattern with long-term survival despite multiple metastases.

The optimal treatment is generally considered to be surgery with postoperative radiotherapy when disease-free margins cannot be obtained surgically and when there is locally advanced disease or high-grade histological findings.[6],[7] Poor prognostic factors for ACC are solid growth pattern, positive resection margin, PNI, and higher tumor stages.[2] Chemotherapy is normally reserved for the palliative treatment of symptomatic locally recurrent or metastatic disease that is not suitable for further surgery or radiation.[6]

ACC may represent with metastasis in long-term follow-up. The implementation of local treatments as much as possible may increase patients' quality of life and overall survival.

We aimed to present this case since ACC rarely metastasized to small bowel and chaceum, and it is readily controlled with local treatments. As a result, ACC cases with colon metastasis may present with iron deficiency anemia due to occult GI bleeding. As in our case, performing GIS screening in ACC patients presenting with iron deficiency is important for early detection of metastatic disease.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

1.
Akhavan A, Binesh F, Navabii H. Adenoid cystic carcinoma of hard palate with coincidental metastases to lung and liver. BMJ Case Rep 2013;2013. pii:Bcr0120125658.  Back to cited text no. 1
    
2.
Shingaki S, Kanemaru S, Oda Y, Niimi K, Mikami T, Funayama A, et al. Distant metastasis and survival of adenoid cystic carcinoma after definitive treatment. J Oral Maxillofac Surg Med Pathol 2014;26:312-6.  Back to cited text no. 2
    
3.
Matsuba HM, Thawley SE, Simpson JR, Levine LA, Mauney M. Adenoid cystic carcinoma of major and minor salivary gland origin. Laryngoscope 1984;94:1316-8.  Back to cited text no. 3
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4.
Kumar V, Nair R, Upadhyaya S, Nayal B, Shetty A. Extradural spinal metastasis of adenoid cystic carcinoma (ACC): A case report. J Clin Diagn Res 2015;9:XD09-10.  Back to cited text no. 4
    
5.
Remo A, Zanella C, Pancione M, Astati L, Piacentini P, Cingarlini S, et al. Lung metastasis from TTF-1 positive sigmoid adenocarcinoma. Pitfalls and management. Pathologica 2013;105:69-72.  Back to cited text no. 5
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6.
Coca-Pelaz A, Rodrigo JP, Bradley PJ, Vander Poorten V, Triantafyllou A, Hunt JL, et al. Adenoid cystic carcinoma of the head and neck – An update. Oral Oncol 2015;51:652-61.  Back to cited text no. 6
[PUBMED]    
7.
Tincani AJ, Del Negro A, Araújo PP, Akashi HK, Martins AS, Altemani AM, et al. Management of salivary gland adenoid cystic carcinoma: Institutional experience of a case series. Sao Paulo Med J 2006;124:26-30.  Back to cited text no. 7
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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