DOI: 10.1136/bcr-2018-225243

In a perplexing medical case recently reported in BMJ Case Reports, a 79-year-old man experienced an extraordinary recurrence of his previously treated salivary gland carcinoma. The case, led by a team of medical professionals at Ninewells Hospital and the University of Dundee, documents the evolution of his condition, which began with a lump in his left submandibular region and escalated to significant cranial nerve involvement.

Initially, the patient had undergone a radical excision of a carcinoma ex-pleomorphic adenoma from his left submandibular gland. However, he presented years later with a new 2cm lump in the same region, indicative of a neoplastic recurrence confirmed upon surgical excision. What followed was a surprising turn of events – during the workup for revision surgery, the patient developed a right VI cranial nerve palsy, impairing the lateral rectus muscle and leading to diplopia, commonly known as double vision. Although his history of transient ischemic attacks (TIAs) suggested a vascular etiology for the nerve palsy, the patient’s condition didn’t improve even after six months, prompting further investigation.

Subsequent MRI scans unraveled a new soft tissue area near the cavernous segment of the internal carotid artery. This discovery proved to be an unusual manifestation of the patient’s recurring cancer, particularly concerning since there was no local recurrence or nodal disease visible initially. Over time, repeated imaging revealed progressive growth of the abnormal tissue within the cavernous sinus and primary submandibular location. This involvement of multiple cranial nerves – IV, V, VI, VII, and XII – rendered surgical excision of the tumor impossible, marking a shift towards palliative care.

This case amplifies the myriad ways in which cancer can present and recur, demanding continuous vigilance from healthcare professionals even after the apparent successful treatment of a salivary gland carcinoma. According to Antony and colleagues (2012), “Carcinoma ex pleomorphic adenoma: a comprehensive review of clinical, pathological and molecular data” (DOI:10.1007/s12105-011-0281-z), tumor recurrence can manifest in various forms, with or without palpable masses. The absence of locoregional lytic lesions does not exclude the presence of neoplastic activity, as illustrated by this case where the neurologic deficits were the harbinger of recurrence. Similarly, Hu and team (2016) accentuated the significance of a broad range of prognostic factors—including local invasion when dealing with carcinoma ex pleomorphic adenoma (DOI:10.1016/j.oooo.2016.06.005).

Spiro’s 35-year review, “Salivary neoplasms: overview of a 35-year experience with 2,807 patients” (DOI:10.1002/hed.2890080309), underscores the diversity and unpredictability inherent in salivary gland neoplasms. This diversity can make diagnosis and subsequent therapeutic decisions challenging. Additionally, the World Health Organization classification of head and neck tumours (El-Naggar et al., 2017) aids in identifying malignancies but cannot predict all potential disease courses, as indicated by the progression and complexity of the case in question.

The Case Report serves as a crucial reminder of the importance of comprehensive follow-up in oncologic care, especially considering the difficulties in predicting cancer behavior. Katabi and colleagues (2010) explained the importance of understanding carcinoma histologic subtypes’ prognostic factors regarding recurrence and metastasis (DOI:10.1016/j.humpath.2009.12.011).

Moreover, the patient’s development of diplopia as a symptom of cancer recurrence brings focus to the necessity of interdisciplinary evaluation in cases of new-onset cranial nerve palsies amongst elderly patients. Dinkin (2014) posited the diagnostic approach to diplopia must be comprehensive, taking into account all potential etiologies from the benign to the life-threatening (DOI:10.1212/01.CON.0000453310.52390.58).

The implications of this case study extend beyond academic curiosity, serving as a reminder of the intricate nature of cancer recurrence and the necessity for a multi-faceted approach to patient care. Timely identification and intervention are paramount, as reiterated by the Royal College of Optometrists on the urgency of referrals in cases of new neuro-ophthalmic symptoms (http://guidance.college-optometrists.org/guidance-contents/communication-partnership-and-teamwork-domain/working-with-colleagues/urgency-of-referrals/).

In conclusion, the reported case underscores the complexity of recurrent salivary gland carcinomas and highlights the need for continuous monitoring for neoplastic recurrences, even in seemingly unrelated areas. The unusual presentation of diplopia as a symptom of tumor recurrence suggests the need for a detail-oriented diagnostic approach when encountering patients with similar histories.

Keywords

1. Salivary Gland Carcinoma Recurrence
2. Diplopia and Cancer
3. Cranial Nerve Palsy in Carcinoma
4. Carcinoma ex-Pleomorphic Adenoma
5. Tumor Recurrence and Imaging

References

1. Antony J, Gopalan V, Smith RA, et al. (2012). Carcinoma ex pleomorphic adenoma: a comprehensive review of clinical, pathological and molecular data. Head Neck Pathol, 6:1-9. DOI:10.1007/s12105-011-0281-z
2. Hu YH, Zhang CY, Xia RH, et al. (2016). Prognostic factors of carcinoma ex pleomorphic adenoma of the salivary glands, with emphasis on the widely invasive carcinoma: a clinicopathologic analysis of 361 cases in a Chinese population. Oral Surg Oral Med Oral Pathol Oral Radiol, 122:598-608. DOI:10.1016/j.oooo.2016.06.005
3. Spiro RH. (1986). Salivary neoplasms: overview of a 35-year experience with 2,807 patients. Head Neck Surg, 8:177–84. DOI:10.1002/hed.2890080309
4. Dinkin M. (2014). Diagnostic approach to diplopia. Continuum, 20:942–65. DOI:10.1212/01.CON.0000453310.52390.58
5. Royal College of Optometrists. (2017). Urgency of referrals. Retrieved from http://guidance.college-optometrists.org/guidance-contents/communication-partnership-and-teamwork-domain/working-with-colleagues/urgency-of-referrals/