Response to PD‐1 blockade in a patient with mucosal melanoma of the middle ear: Case report

Komatsuda, Hiroki; Kumai, Takumi; Ueda, Seigo; Hirata-Nozaki, Yui; Hayashi, Tatsuya; Harabuchi, Yasuaki

doi:10.1002/ccr3.3424

Cited by 5 publications

(5 citation statements)

References 14 publications

(33 reference statements)

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“…Recently, immunotherapy targeting CTLA-4 and IL-17 has been performed, 7 and a case report of primary MM from the middle ear treated with a PD-1 blockade reported a stable disease for more than 5 years. 15 However, most patients died of disease progression, with an average disease-free period of 1 year.…”

Section: Discussionmentioning

confidence: 99%

Mucosal Melanoma of Eustachian Tube: A Case Report

Kang¹,

Kim²,

Hong³

et al. 2022

Int Adv Otol

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Mucosal melanoma originating from the eustachian tube is very rare, and only 15 cases were reported so far. In this study, we report a case of mucosal melanoma from the eustachian tube which was surgically managed, followed by chemoradiotherapy. A 53-year-old man presented with a history of recurrent idiopathic hemotympanum and a dark red mass in the nasopharynx protruding from the eustachian tube orifice. Under an impression of mucosal melanoma from the eustachian tube, en-bloc surgical removal was performed using the infratemporal fossa approach type C combined with a transnasal endoscopic approach followed by postoperative chemoradiotherapy. However, the disease progressed to lung metastasis, and the patient died of the disease at 13 months postoperatively. The presenting case showed a poor progression despite a margin-free surgical resection followed by chemoradiotherapy. Additional trial of new treatment options is necessary to improve the poor prognosis.

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Section: Discussionmentioning

confidence: 99%

Mucosal Melanoma of Eustachian Tube: A Case Report

Kang¹,

Kim²,

Hong³

et al. 2022

Int Adv Otol

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“…Moreover, most patient-reported symptoms due to MM, including hearing loss, otalgia, or otorrhea, are common and may be secondary to a myriad of etiologies. Nevertheless, physicians should include temporal bone MM as a potential diagnosis in patients presenting with bloody otorrhea in the context of a chronic, recurrent serous otitis media, as observed in several of the patients in the current review (22,24–28). Furthermore, CN V, VI, and VII palsies should indicate the need for immediate further workup for potential temporal bone disease (8,20,21,28–31).…”

Section: Discussionmentioning

confidence: 99%

A Systematic Review of Primary Temporal Bone Mucosal Melanoma

et al. 2022

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Objective: Temporal bone mucosal melanomas (MMs) are rare, and patients may experience delays in diagnosis and treatment. Our objective was to better characterize the presentation, diagnosis, treatment modalities, and outcomes of this process. Data Sources: PubMed/Medline, CINAHL (EBSCOhost), and Web of Science databases were searched in all languages without restriction of publication dates. Study Selection: Inclusion criteria included that the article was either a case report or a case series with individual case data. All non-English articles were excluded if the corresponding abstract lacked data on demographics, initial presentation, and clinical management. Data Extraction: After full-text analysis, data pertaining to demographics, diagnosis, medical and surgical management modalities, and outcomes were extracted.Data Synthesis: Data were qualitatively synthesized, and means and averages were obtained for all continuous variables. Overall survival was measured by the Kaplan-Meier method, and significance was measured through log-rank testing. Conclusions: Clinicians should suspect temporal bone MM in the differential diagnosis of patients with bloody otorrhea in the context of a chronic serous otitis media or an associated cranial nerve palsy. If suspected, physicians should not delay the acquisition of a biopsy or imaging studies. Management is highly variable and must be decided on a case-by-case basis. Outcomes remain poor because of the high propensity for MM to metastasize.

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“…Mucosal melanoma of the middle ear and petrous temporal bone is remarkably rare, with only 13 cases identified in the literature (3)(4)(5)(6)(7)(8)(9)(10)(11)(12)(13)(14)(15). The mucosal surface of the Eustachian tube is also a described primary site and these lesions may extend into the middle ear cavity (3,18,19).…”

Section: Discussionmentioning

confidence: 99%

“…The group reported no evidence of disease at 9 months postoperatively. Komatsuda et al (4) described the use of a PD-1 inhibitor as a monotherapy for mucosal melanoma of the middle ear, reporting a partial response, stable for 60 months.…”

Section: Discussionmentioning

confidence: 99%

“…Though it makes up only 1% of all melanoma cases, mucosal melanoma most commonly presents within the head and neck (1,2). Primary lesions of the middle ear or petrous temporal bone are exceedingly rare with only 13 cases described (3)(4)(5)(6)(7)(8)(9)(10)(11)(12)(13)(14)(15). Surgical resection is the recommended primary treatment for mucosal melanoma (16,17).…”

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confidence: 99%

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Remission of Mucosal Melanoma of the Middle Ear and Petrous Temporal Bone and Reversal of Cranial Nerve Paresis Following Radiation and Single Agent Nivolumab: Clinical Capsule and Review of the Literature

Carlson¹,

Volsky²

2021

Otology &Amp; Neurotology

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Objective: We report disease remission and recovery of fifth and seventh nerve paresis in a case of primary mucosal melanoma of the middle ear and petrous temporal bone. Patient: A 74-year-old man developed sudden, profound, right sided sensorineural hearing loss, disequilibrium, otalgia, and cranial nerve V and VII dysfunction. Imaging demonstrated an unresectable, osteolytic lesion involving the middle ear and anterior petrous apex. Melanoma was diagnosed via in-office biopsy; whole-body metabolic imaging revealed no other primary site. Intervention: Multidisciplinary management included radiation therapy (30 Gy, 10 fractions) followed by induction (five cycles, q2w) and maintenance nivolumab (six cycles, q3w). Main Outcome Measure: Complete metabolic response of primary site and metastases on imaging, recovery of cranial neuropathies.Results: Following palliative radiation therapy and induction nivolumab, cranial neuropathies resolved. With maintenancedose nivolumab, primary site and metastases exhibited a complete response. Therapy was stopped at 16 months postdiagnosis. Complete remission was maintained until 22 months after diagnosis. The patient developed a solitary cerebral metastasis which was refractory to radiosurgery and biopsy confirmed melanoma. He expired 2 years, 8 months post-diagnosis. Conclusions: Mucosal melanoma of the middle ear and petrous temporal bone is exceedingly rare. Management is individualized and surgery is undertaken when possible. Key observations in this case are the complete metabolic response and reversal of cranial nerve neuropathies following radiation and anti-programed cell death receptor ligand 1 therapy. Non-surgical treatment is worthy of study as initial management for similar lesions.

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Response to PD‐1 blockade in a patient with mucosal melanoma of the middle ear: Case report

Abstract: This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.

Cited by 5 publications

References 14 publications

Mucosal Melanoma of Eustachian Tube: A Case Report

Mucosal Melanoma of Eustachian Tube: A Case Report

A Systematic Review of Primary Temporal Bone Mucosal Melanoma

Remission of Mucosal Melanoma of the Middle Ear and Petrous Temporal Bone and Reversal of Cranial Nerve Paresis Following Radiation and Single Agent Nivolumab: Clinical Capsule and Review of the Literature

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