Extradural Spinal Metastasis of Adenoid Cystic Carcinoma (ACC): A Case Report

Kumar, Vinod

doi:10.7860/jcdr/2015/10644.5498

Cited by 6 publications

(6 citation statements)

References 9 publications

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“…The liver was reported in several studies as a single site of the systemic spread of the disease. Organs with less common metastasis include the breast and larynx, and those which are rarely involved in SACC metastasis include the pituitary gland, the sternum, the dorsal spine [ 31 ], the choroid, the toe bones, and the pericardium [ 31 – 38 ].…”

Section: Resultsmentioning

confidence: 99%

Adenoid Cystic Carcinoma of Salivary Glands: A Ten-Year Review and an Assessment of the Current Management, Surgery, Radiotherapy, and Chemotherapy

Saleh

Ukwas

2023

International Journal of Otolaryngology

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Adenoid cystic carcinoma (ACC) is a rare cancer that arises from the salivary glands and other sites in the body, such as the lung and breast. Although the tumor accounts for 10% of all salivary gland malignancies, it only accounts for 1% of head and neck malignancies. It can affect both major and minor salivary glands; here, it is called salivary gland adenoid cystic carcinoma or SACC, with a slight predilection to the latter, and commonly manifests between the 6th and 7th decades of life. The disease also shows a slight female predilection, with a reported female to male ratio of 3 : 2. Lesions of SACC are often insidious and slow-growing, and symptoms such as pain and altered sensation are frequently associated with advanced stages of the disease. Salivary adenoid cystic carcinoma is characterized by perineural invasion (PNI), a distinctive feature that potentially plays a significant role in the tumor’s relapse and recurrence, which is approximately 50%. The disease is not prevalent, and its etiopathogenesis is poorly understood, although several genetic patterns and biomarkers have been linked to its initiation and/or progression. The discovery of these mutations and biomarkers has encouraged several clinical studies to use therapeutic agents to target the specific receptors on the cancer cells to potentially prevent further proliferation of the tumor cells and metastasis of the disease. Diagnosis of SACC is often challenging and frequently requires a combination of clinical examination, imaging, and histopathology. Management of SACC is primarily surgical excision, while radiotherapy has shown to be effective in improving local control in cases with microscopic residual disease. However, treatment of recurrent or metastatic tumors by radiotherapy with or without chemotherapy has so far shown limited success. The aim of this thesis is to provide an update of literature on SACC with a particular focus on the latest management approaches and future trends.

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Section: Resultsmentioning

confidence: 99%

Adenoid Cystic Carcinoma of Salivary Glands: A Ten-Year Review and an Assessment of the Current Management, Surgery, Radiotherapy, and Chemotherapy

Saleh

Ukwas

2023

International Journal of Otolaryngology

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“…Multiple studies have also reported the liver as a primary site for systemic disease spread. While metastasis to the breast and larynx is less frequent, extremely rare instances of metastasis have been noted in areas such as the pituitary gland, sternum, dorsal spine [ 80 ], choroid, bones of the toe, and the pericardium [ 80 , 81 , 82 , 83 , 84 , 85 , 86 ]. Recurrences in ACC are frequent, and in post-initial treatment, the chances of reoccurrence can be up to 50% in particular cases [ 77 ].…”

Section: Discussionmentioning

confidence: 99%

Adenoid Cystic Carcinoma of the Minor Salivary Glands: A Systematic Review and Meta-Analysis of Clinical Characteristics and Management Strategies

Jaber,

Hassan,

Ingafou

et al. 2024

JCM

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Background: Intraoral adenoid cystic carcinoma (ACC) arising from minor salivary glands (MSG) is a rare malignancy associated with delayed diagnosis and unfavorable outcomes. This study aimed to comprehensively review ACC of MSGs, focusing on clinical characteristics, imaging modalities, treatment approaches, and long-term outcomes. Methods: A systematic search was conducted in PubMed, Web of Science, and MEDLINE databases to identify relevant articles reporting cases of ACC of MSGs between January 1997 and March 2023. The study was registered in PROSPERO (ID: CRD42023449478). A total of 10 studies that met the inclusion criteria were selected for critical review. In total, 902 patients were diagnosed with ACC of MSGs with an age range of 44.3 to 63 years, and an average age of 56.6 years. The female to male ratio ranges from 1:1 to 2.4:1. Regarding the primary site of ACC, the palate was the most common location, accounting for 30.5% to 83.3%, followed by the buccal mucosa, floor of the mouth, and lip and the retromolar area. For histology, the solid mass pattern was the most prevalent, seen in 95.2% of patients, followed by the cribriform pattern. Regarding treatment modalities, surgery was the most common approach, applied in 76.3% of cases, with a combination of surgery and radiotherapy used in 29.0% of cases. A smaller fraction, 3.2%, received a combination of surgery, chemotherapy, and radiotherapy, and 8.3% underwent radiotherapy alone. Local recurrence rates varied between 1% and 28.5%, and distant metastasis occurred in 18.2% to 33.3% of cases, predominantly to lymph nodes (14.5%). An analysis of overall survival across various stages and patient numbers indicated a 5-year survival rate of 68.0%. The findings of this study provide valuable insights for physicians in making treatment decisions and emphasize the need for ongoing research and collaborative clinical efforts to improve the management and outcomes of this challenging disease. Conclusion: ACC of MSGs is a multifaceted condition typically manifesting as asymptomatic enlargement and ulceration. This disease is marked by distinct histopathological patterns and perineural invasion (PNI). Recognizing these distinctive aspects is key in shaping the treatment plan, which can range from surgical procedures to radiation therapy, chemotherapy, and evolving targeted treatments. Continuous research and collaborative clinical efforts remain critical for ongoing progress in the treatment and management of this challenging condition.

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“… 3 , 10 , 11 A limited number of spine metastasis cases of ADCC from other gland origins of head and neck, such as parotid gland, submandibular gland, tongue base, and lacrimal gland have been reported. 12 - 14 However, spinal metastatic ADCC arising from ceruminous gland in the EAC is exceedingly rare. Marchesini et al 15 reported a male patient complaining of back pain and worsening myelopathy and was diagnosed with intramedullary spinal cord metastasis from ADCC of EAC.…”

Section: Discussionmentioning

confidence: 99%

Metastatic Ceruminous Adenoid Cystic Carcinoma of the Lumbar Spine Causing Neurological Compromise: A Case Report

Shen

Yang

Líu

et al. 2022

Geriatr Orthop Surg Rehabil

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Background Ceruminous glands are modified apocrine glands of the external auditory canal (EAC). Malignant tumours within the ceruminous glands are extremely rare, and the most common histological type is adenoid cystic carcinoma (ADCC), which has high recurrence and metastasis risks. Although a few cases of metastatic ADCC from other head and neck glands have been reported, metastatic ADCC originating from the ceruminous gland are extremely rare. Case presentation We present an unusual case of spinal metastases of ADCC from ceruminous glands. A 61-year-old woman complaining of low back pain and both lower limbs pain was referred to our department. The primary ceruminous tumour was resected 26 years ago and recurred 6 years later, which was treated by radiotherapy. Three years ago, she presented with low back pain and was diagnosed as multiple lungs and bone metastases. The patient underwent tumour excision, decompression and fusion. The biopsy revealed metastatic ADCC. The symptoms were alleviated after surgery. Conclusions ADCC of EAC is a pernicious malignant tumour that is characterized by slow-growing patterns and a high predisposition to recurrence and metastasis. Differential diagnoses of ADCC and benign tumours in the EAC are challenging, particularly at early stages. We report a rare case of ceruminous ADCC with a prolonged clinical history as well as spinal metastasis and highlight the significance of regular follow-ups for patients undergoing tumour excision in the EAC.

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Extradural Spinal Metastasis of Adenoid Cystic Carcinoma (ACC): A Case Report

Cited by 6 publications

References 9 publications

Adenoid Cystic Carcinoma of Salivary Glands: A Ten-Year Review and an Assessment of the Current Management, Surgery, Radiotherapy, and Chemotherapy

Adenoid Cystic Carcinoma of Salivary Glands: A Ten-Year Review and an Assessment of the Current Management, Surgery, Radiotherapy, and Chemotherapy

Adenoid Cystic Carcinoma of the Minor Salivary Glands: A Systematic Review and Meta-Analysis of Clinical Characteristics and Management Strategies

Metastatic Ceruminous Adenoid Cystic Carcinoma of the Lumbar Spine Causing Neurological Compromise: A Case Report

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