Int J Clin Exp Pathol 2014;7(7):4516-4518 www.ijcep.com /ISSN:1936-2625/IJCEP0000821
Case Report Nasopharyngeal adenoid cystic carcinoma: a case report and review of the literature Yan-Fang Liang1,2, Bin Kong3, Wen-Yu Xiang3, Jian-Bo Ruan1,2, Ling-Mei Wang1,2, Can Chen1,2, Wei-Hua Xu4, Qiu-Liang Wu5, Jin-Cheng Zeng3, Jun-Fa Xu3 Department of Pathology, Taiping People’s Hospital of Dongguan, Dongguan 523905, China; 2Dongguan Hospital Affiliated to Medical College of Jinan University, Dongguan 523905, China; 3Guangdong Provincial Key Laboratory of Medical Molecular Diagnostics, Guandong Medical College, Dongguan 523808, China; 4Department of Otolaryngology, Affiliated Hospital of Guandong Medical College, Zhanjiang 524023, China; 5Sun Yat-sen University Cancer Center, Guanzhou 510060, China 1
Received May 18, 2014; Accepted June 12, 2014; Epub June 15, 2014; Published July 1, 2014 Abstract: ACC derived from nasopharyngeal epithelial cells is rare, usually benign. In this article, we reported a nasopharyngeal adenoid cystic carcinoma (NACC) in a 31-year-old woman with a symptom of hoarseness, headache, epistaxis slightly, diplopia, facial numbness and dysphagia near 3 months. A tumor on the right side of the nasopharynx was confirmed by laryngoscope check and MRI of the skull base. Histopathological findings showed that tumor cells were arranged in cord-like or acinar-like by atypical hyperplastic epithelial cells forming a cribriform and tubular pattern, and immunohistochemical findings showed that tumor tissues were immunopositive for p63 (+), CK7 (+), CK19 (+), CK8 (+), CK18 (+), SMA (+), CK (+), p53 (++), S-100 (+) and Ki-67 (5%+), and negative for CD34 (-), CK5/6 (-), CEA (-) and CD117 (-). Patient was treated by surgical operation and radiotherapy, and was followed-up near 10 months, no local recurrence and distant metastasis. Keywords: Adenoid cystic carcinoma, nasopharynx, immunohistochemistry
Introduction
Case report
Adenoid cystic carcinoma (ACC) was first described by the Billroth in 1895. Typical features of ACC shows a slow growth rate, high propensity for perineural spread, local recurrence and distant metastasis (like lung, liver, bone, et al) [1, 2]. Now, ACC is one of the most common malignant salivary gland neoplasms, but occurs rarely in the nasopharynx, accounting for 0.13~0.48% of all malignant nasopharyngeal tumors [3, 4]. ACC of the nasopharynx (NACC) has a characteristic with low lymphatic metastases rate, slow growth rate and high local invasiveness. And, partly NACC patients show perineural spread, intracranial involvement and cervical lymphadenopathy [5, 6]. So, the treatment of NACC should be combined by surgical operation and radiotherapy. Here, we reported a NACC in a 31-year-old woman with a symptom of hoarseness, headache, epistaxis slightly, diplopia, facial numbness and dysphagia near 3 months.
A 31-year-old woman had a symptom of hoarseness, headache, epistaxis slightly, diplopia, facial numbness and dysphagia both for liquid or solid food randomly last for 3 months. A nasopharynx tumor was observed by laryngoscope check. MRI of the skull base, signal uneven, high signal of T2W1, also confirmed an irregular mass in the right side of the nasopharynx with infiltration of right pterygopalatine fossa and cavernous. She was hospitalized in Department of Otolaryngology at Taiping People’s Hospital of Dongguan for endoscopic surgery. And, two irregularly gray mass measured 1 cm × 0.5 cm × 0.4 cm in volume from nasopharynx were taken out in succession. Wound healing was good after surgery one week, and radiation was used begin in another one week. Histopathological findings showed that tumor cells were arranged in cord-like or acinar-like by atypical hyperplastic epithelial cells forming a cribriform and tubular pattern,
Nasopharyngeal adenoid cystic carcinoma
Figure 1. The NACC tissues were stained with hematoxylin and eosin. A: Tumor cells were arranged in cord-like or acinar-like by atypical hyperplastic epithelial cells forming a cribriform and tubular pattern (40 ×). B: Some glass-like eosinophilic materials or mucilages were observed in tumor interstitials (100 ×).
Figure 2. NACC tissues were stained with immunohistochemistry. The tissues were immunopositive for CK (A), CK7 (B), CK8 (C), CK18 (D), CK19 (E), Ki-67 (F), P53 (G), P63 (H), S-100 (I), SMA (J) and immunonegative for CD34 (K), CD117 (L), CEA (M), CK5/6 (N) in NACC tissues (100 ×).
and some glass-like eosinophilic materials or mucilages were observed in tumor interstitials (Figure 1). Immunohistochemical findings showed that tumor tissues were immunopositive for p63 (+), CK7 (+), CK19 (+), CK8 (+), CK18 (+), SMA (+), CK (+), p53 (++), S-100 (+) and Ki-67 (5%+), and negative for CD34 (-), CK5/6 (-), CEA (-) and CD117 (-) (Figure 2). The 4517
patient was followed-up near 10 months, no local recurrence and distant metastasis. Discussion ACC can arise from parotid, submandibular, palate, intraoral site, buccal, tongue and nasopharynx glands [1-9]. Although most ACC arising from the head and neck glands, ACCs are Int J Clin Exp Pathol 2014;7(7):4516-4518
Nasopharyngeal adenoid cystic carcinoma common in uterine cervix, lungs, kidneys, prostate, skin, esophagus and breast [2, 4, 10, 11]. When ACC patients present high stage disease, skull base involvement, tumor recurrence, lymphovascular invasion, bone invasion and perineural invasion, suggest an increased incidence of recurrence or dying in patients [2-4].
of Dongguan, Dongguan 523905, China. E-mail: [email protected]
The best treatment for ACC is unanimously considered endoscopic surgery followed by radiotherapy [2, 4, 12]. MRI of the skull base is necessary for NACC patients, although there is no consensus on the imaging characteristics of NACC [2, 6, 13]. This approach ensures an optimal therapeutic outcome via accurate tumor mapping with special attention to the perineural spread. It is important to distinguish NACC from nasopharyngeal squamous cell carcinoma (NSCC). The skull base MRI of NSCC patients have a uniform signal on lesions. However, most NACC patients have a non-uniform signal on lesions.
[2]
Besides MRI, histological findings and immunohistochemical findings are very important in the diagnosis and distinguish of NACC. The cribriform and tubular pattern on tumor tissue and immunopositive for p63, CK7, CK19, CK8, CK18, SMA, CK, p53, S-100 and Ki-67 (5%), and negative for CD34, CK5/6, CEA and CD117, lead us to diagnose this patient as having a NACC. Cranial nerves invasion, stage and treatment approach might be important factors affecting the prognosis of NACC patients. Here, patient with no visible nerves invasion was treated by surgical operation following by radiotherapy, and followed-up showed no local recurrence and distant metastasis near 10 months after leaving hospital.
References [1]
[3] [4]
[5]
[6]
[7]
[8] [9]
Acknowledgements This work was supported by the National Natural Science Foundation of China (81273237), the Key Project of Science and Technology Innovation of Education Department of Guangdong Province (2012KJCX0059), the Science and Technology Project of Dongguan (20131051010006) and the Science and Technology Innovation Fund of Guangdong Medical College (STIF201110). Disclosure of conflict of interest None. Address correspondence to: Dr. Yan-Fang Liang, Department of Pathology, Taiping People’s Hospital
4518
[10]
[11]
[12] [13]
Alleyne CH, Bakay RA, Costigan D, Thomas B, Joseph GJ. Intracranial adenoid cystic carcinoma: case report and review of the literature. Surg Neurol 1996; 45: 265-71. Saâdi I, El Marfany M, Hadadi K, Amaoui B, Kebdani T, Errihani H, Mansouri A, Benjaafar N, El-Gueddari BK. Adenoid cystic carcinoma of the nasopharynx: a case report. Cancer Radiother 2003; 7: 190-4 Kuno H, Fujii S. A case of adenoid cystic carcinoma arising from the nasopharynx. Jpn J Clin Oncol 2013; 43: 942. Gormley WB, Sekhar LN, Wright DC, Olding M, Janecka IP, Snyderman CH, Richardson R. Management and long-term outcome of adenoid cystic carcinoma with intracranial extension: a neurosurgical perspective. Neurosurgery 1996; 38: 1105-12. Thompson LD, Penner C, Ho NJ, Foss RD, Miettinen M, Wieneke JA, Moskaluk CA, Stelow EB. Sinonasal tract and nasopharyngeal adenoid cystic carcinoma: a clinicopathologic and immunophenotypic study of 86 cases. Head Neck Pathol 2014; 8: 88-109. Liu XW, Xie CM, Li H, Zhang R, Geng ZJ, Mo YX, Zhao J, Cai MY, Lv YC, Wu PH. Nasopharyngeal adenoid cystic carcinoma: magnetic resonance imaging features in ten cases. Chin J Cancer 2012; 31: 19-28. Sa YJ, Sim SB, Kim TJ, Moon SW, Park CB. Latedeveloping tongue adenoid cystic carcinoma after pulmonary metastasectomy: a case report. World J Surg Oncol 2014; 12: 102. S V, R A. Adenoid cystic carcinoma of the buccal mucosa: a case report with review of literature. J Clin Diagn Res 2014; 8: 266-268. Terada T. Adenoid cystic carcinoma of the oral cavity: immunohistochemical study of four cases. Int J Clin Exp Pathol 2013; 6: 932-938. Canyilmaz E, Uslu GH, Memış Y, Bahat Z, Yildiz K, Yoney A. Adenoid cystic carcinoma of the breast: A case report and literature review. Oncol Lett 2014; 7: 1599-1601. Terada T. Pigmented adenoid cystic carcinoma of the ear skin arising from the epidermis: a case report with immunohistochemical studies. Int J Clin Exp Pathol 2012; 5: 254-259. Bradley PJ. Adenoid cystic carcinoma of the head and neck: a review. Curr Opin Otolaryngol Head Neck Surg 2004; 12: 127-32. Soprani F, Armaroli V, Venturini A, Emiliani E, Casolino D. A rare case of adenoid cystic carcinoma of the nasopharynx manifesting as Horner’s syndrome: discussion and review of the literature. Acta Otorhinolaryngol Ital 2007; 27: 216-9.
Int J Clin Exp Pathol 2014;7(7):4516-4518
Case Report Nasopharyngeal adenoid cystic carcinoma: a case report and review of the literature Yan-Fang Liang1,2, Bin Kong3, Wen-Yu Xiang3, Jian-Bo Ruan1,2, Ling-Mei Wang1,2, Can Chen1,2, Wei-Hua Xu4, Qiu-Liang Wu5, Jin-Cheng Zeng3, Jun-Fa Xu3 Department of Pathology, Taiping People’s Hospital of Dongguan, Dongguan 523905, China; 2Dongguan Hospital Affiliated to Medical College of Jinan University, Dongguan 523905, China; 3Guangdong Provincial Key Laboratory of Medical Molecular Diagnostics, Guandong Medical College, Dongguan 523808, China; 4Department of Otolaryngology, Affiliated Hospital of Guandong Medical College, Zhanjiang 524023, China; 5Sun Yat-sen University Cancer Center, Guanzhou 510060, China 1
Received May 18, 2014; Accepted June 12, 2014; Epub June 15, 2014; Published July 1, 2014 Abstract: ACC derived from nasopharyngeal epithelial cells is rare, usually benign. In this article, we reported a nasopharyngeal adenoid cystic carcinoma (NACC) in a 31-year-old woman with a symptom of hoarseness, headache, epistaxis slightly, diplopia, facial numbness and dysphagia near 3 months. A tumor on the right side of the nasopharynx was confirmed by laryngoscope check and MRI of the skull base. Histopathological findings showed that tumor cells were arranged in cord-like or acinar-like by atypical hyperplastic epithelial cells forming a cribriform and tubular pattern, and immunohistochemical findings showed that tumor tissues were immunopositive for p63 (+), CK7 (+), CK19 (+), CK8 (+), CK18 (+), SMA (+), CK (+), p53 (++), S-100 (+) and Ki-67 (5%+), and negative for CD34 (-), CK5/6 (-), CEA (-) and CD117 (-). Patient was treated by surgical operation and radiotherapy, and was followed-up near 10 months, no local recurrence and distant metastasis. Keywords: Adenoid cystic carcinoma, nasopharynx, immunohistochemistry
Introduction
Case report
Adenoid cystic carcinoma (ACC) was first described by the Billroth in 1895. Typical features of ACC shows a slow growth rate, high propensity for perineural spread, local recurrence and distant metastasis (like lung, liver, bone, et al) [1, 2]. Now, ACC is one of the most common malignant salivary gland neoplasms, but occurs rarely in the nasopharynx, accounting for 0.13~0.48% of all malignant nasopharyngeal tumors [3, 4]. ACC of the nasopharynx (NACC) has a characteristic with low lymphatic metastases rate, slow growth rate and high local invasiveness. And, partly NACC patients show perineural spread, intracranial involvement and cervical lymphadenopathy [5, 6]. So, the treatment of NACC should be combined by surgical operation and radiotherapy. Here, we reported a NACC in a 31-year-old woman with a symptom of hoarseness, headache, epistaxis slightly, diplopia, facial numbness and dysphagia near 3 months.
A 31-year-old woman had a symptom of hoarseness, headache, epistaxis slightly, diplopia, facial numbness and dysphagia both for liquid or solid food randomly last for 3 months. A nasopharynx tumor was observed by laryngoscope check. MRI of the skull base, signal uneven, high signal of T2W1, also confirmed an irregular mass in the right side of the nasopharynx with infiltration of right pterygopalatine fossa and cavernous. She was hospitalized in Department of Otolaryngology at Taiping People’s Hospital of Dongguan for endoscopic surgery. And, two irregularly gray mass measured 1 cm × 0.5 cm × 0.4 cm in volume from nasopharynx were taken out in succession. Wound healing was good after surgery one week, and radiation was used begin in another one week. Histopathological findings showed that tumor cells were arranged in cord-like or acinar-like by atypical hyperplastic epithelial cells forming a cribriform and tubular pattern,
Nasopharyngeal adenoid cystic carcinoma
Figure 1. The NACC tissues were stained with hematoxylin and eosin. A: Tumor cells were arranged in cord-like or acinar-like by atypical hyperplastic epithelial cells forming a cribriform and tubular pattern (40 ×). B: Some glass-like eosinophilic materials or mucilages were observed in tumor interstitials (100 ×).
Figure 2. NACC tissues were stained with immunohistochemistry. The tissues were immunopositive for CK (A), CK7 (B), CK8 (C), CK18 (D), CK19 (E), Ki-67 (F), P53 (G), P63 (H), S-100 (I), SMA (J) and immunonegative for CD34 (K), CD117 (L), CEA (M), CK5/6 (N) in NACC tissues (100 ×).
and some glass-like eosinophilic materials or mucilages were observed in tumor interstitials (Figure 1). Immunohistochemical findings showed that tumor tissues were immunopositive for p63 (+), CK7 (+), CK19 (+), CK8 (+), CK18 (+), SMA (+), CK (+), p53 (++), S-100 (+) and Ki-67 (5%+), and negative for CD34 (-), CK5/6 (-), CEA (-) and CD117 (-) (Figure 2). The 4517
patient was followed-up near 10 months, no local recurrence and distant metastasis. Discussion ACC can arise from parotid, submandibular, palate, intraoral site, buccal, tongue and nasopharynx glands [1-9]. Although most ACC arising from the head and neck glands, ACCs are Int J Clin Exp Pathol 2014;7(7):4516-4518
Nasopharyngeal adenoid cystic carcinoma common in uterine cervix, lungs, kidneys, prostate, skin, esophagus and breast [2, 4, 10, 11]. When ACC patients present high stage disease, skull base involvement, tumor recurrence, lymphovascular invasion, bone invasion and perineural invasion, suggest an increased incidence of recurrence or dying in patients [2-4].
of Dongguan, Dongguan 523905, China. E-mail: [email protected]
The best treatment for ACC is unanimously considered endoscopic surgery followed by radiotherapy [2, 4, 12]. MRI of the skull base is necessary for NACC patients, although there is no consensus on the imaging characteristics of NACC [2, 6, 13]. This approach ensures an optimal therapeutic outcome via accurate tumor mapping with special attention to the perineural spread. It is important to distinguish NACC from nasopharyngeal squamous cell carcinoma (NSCC). The skull base MRI of NSCC patients have a uniform signal on lesions. However, most NACC patients have a non-uniform signal on lesions.
[2]
Besides MRI, histological findings and immunohistochemical findings are very important in the diagnosis and distinguish of NACC. The cribriform and tubular pattern on tumor tissue and immunopositive for p63, CK7, CK19, CK8, CK18, SMA, CK, p53, S-100 and Ki-67 (5%), and negative for CD34, CK5/6, CEA and CD117, lead us to diagnose this patient as having a NACC. Cranial nerves invasion, stage and treatment approach might be important factors affecting the prognosis of NACC patients. Here, patient with no visible nerves invasion was treated by surgical operation following by radiotherapy, and followed-up showed no local recurrence and distant metastasis near 10 months after leaving hospital.
References [1]
[3] [4]
[5]
[6]
[7]
[8] [9]
Acknowledgements This work was supported by the National Natural Science Foundation of China (81273237), the Key Project of Science and Technology Innovation of Education Department of Guangdong Province (2012KJCX0059), the Science and Technology Project of Dongguan (20131051010006) and the Science and Technology Innovation Fund of Guangdong Medical College (STIF201110). Disclosure of conflict of interest None. Address correspondence to: Dr. Yan-Fang Liang, Department of Pathology, Taiping People’s Hospital
4518
[10]
[11]
[12] [13]
Alleyne CH, Bakay RA, Costigan D, Thomas B, Joseph GJ. Intracranial adenoid cystic carcinoma: case report and review of the literature. Surg Neurol 1996; 45: 265-71. Saâdi I, El Marfany M, Hadadi K, Amaoui B, Kebdani T, Errihani H, Mansouri A, Benjaafar N, El-Gueddari BK. Adenoid cystic carcinoma of the nasopharynx: a case report. Cancer Radiother 2003; 7: 190-4 Kuno H, Fujii S. A case of adenoid cystic carcinoma arising from the nasopharynx. Jpn J Clin Oncol 2013; 43: 942. Gormley WB, Sekhar LN, Wright DC, Olding M, Janecka IP, Snyderman CH, Richardson R. Management and long-term outcome of adenoid cystic carcinoma with intracranial extension: a neurosurgical perspective. Neurosurgery 1996; 38: 1105-12. Thompson LD, Penner C, Ho NJ, Foss RD, Miettinen M, Wieneke JA, Moskaluk CA, Stelow EB. Sinonasal tract and nasopharyngeal adenoid cystic carcinoma: a clinicopathologic and immunophenotypic study of 86 cases. Head Neck Pathol 2014; 8: 88-109. Liu XW, Xie CM, Li H, Zhang R, Geng ZJ, Mo YX, Zhao J, Cai MY, Lv YC, Wu PH. Nasopharyngeal adenoid cystic carcinoma: magnetic resonance imaging features in ten cases. Chin J Cancer 2012; 31: 19-28. Sa YJ, Sim SB, Kim TJ, Moon SW, Park CB. Latedeveloping tongue adenoid cystic carcinoma after pulmonary metastasectomy: a case report. World J Surg Oncol 2014; 12: 102. S V, R A. Adenoid cystic carcinoma of the buccal mucosa: a case report with review of literature. J Clin Diagn Res 2014; 8: 266-268. Terada T. Adenoid cystic carcinoma of the oral cavity: immunohistochemical study of four cases. Int J Clin Exp Pathol 2013; 6: 932-938. Canyilmaz E, Uslu GH, Memış Y, Bahat Z, Yildiz K, Yoney A. Adenoid cystic carcinoma of the breast: A case report and literature review. Oncol Lett 2014; 7: 1599-1601. Terada T. Pigmented adenoid cystic carcinoma of the ear skin arising from the epidermis: a case report with immunohistochemical studies. Int J Clin Exp Pathol 2012; 5: 254-259. Bradley PJ. Adenoid cystic carcinoma of the head and neck: a review. Curr Opin Otolaryngol Head Neck Surg 2004; 12: 127-32. Soprani F, Armaroli V, Venturini A, Emiliani E, Casolino D. A rare case of adenoid cystic carcinoma of the nasopharynx manifesting as Horner’s syndrome: discussion and review of the literature. Acta Otorhinolaryngol Ital 2007; 27: 216-9.
Int J Clin Exp Pathol 2014;7(7):4516-4518
