Abstract
Background. Primary malignant melanoma of the lacrimal sac is rare. A patient with lacrimal sac melanoma was presented, and 14 Japanese patients with lacrimal sac melanoma in the literature were reviewed. Case Presentation. A 78-year-old Japanese man was presented with painless swelling of the lacrimal sac on the left side. Dacryocystectomy revealed diffuse infiltration with large epithelioid cells, sometimes with pigments, which were positive for cocktail mix of antibodies to tyrosinase, melan A (MART-1), and HMB45, leading to pathological diagnosis of melanoma. One month later, positron emission tomography (PET) revealed 2 high-uptake sites (SUVmax = 10.29 and 15.38) at the levels of medial canthus and nasolacrimal duct, but no abnormal uptake in the other site of the body. The lesion had the BRAF V600E mutation. He began to take daily oral dabrafenib (BRAF inhibitor) and trametinib (MEK inhibitor), leading to no abnormal uptake on PET in half a year. He had stable disease in good physical status with small and weak uptake sites of lymph nodes on PET 1 year later. Results. In the review of 15 Japanese patients, including this patient, local recurrence was noted in 4 patients, regional lymph node metastasis only in 3, distant metastasis in 6, and no metastasis in 6. Five patients died within 2 years and the others were alive in short follow-up periods. Conclusions. Chemotherapy was the standard for local recurrence or metastasis. Emerging molecular target drugs, as shown in the present patient, would change the strategy for management of lacrimal sac melanoma.
| Original language | English |
|---|---|
| Journal | Journal of Investigative Medicine High Impact Case Reports |
| Volume | 7 |
| DOIs | |
| Publication status | Published - Jan 1 2019 |
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Keywords
- BRAF inhibitor
- BRAF mutation
- dabrafenib
- lacrimal sac
- malignant melanoma
- MEK inhibitor
- PET/CT
- trametinib
ASJC Scopus subject areas
- Epidemiology
- Safety, Risk, Reliability and Quality
- Safety Research
Cite this
Lacrimal Sac Malignant Melanoma in 15 Japanese Patients : Case Report and Literature Review. / Matsuo, Toshihiko; Tanaka, Takehiro; Yamasaki, Osamu.
In: Journal of Investigative Medicine High Impact Case Reports, Vol. 7, 01.01.2019.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Lacrimal Sac Malignant Melanoma in 15 Japanese Patients
T2 - Case Report and Literature Review
AU - Matsuo, Toshihiko
AU - Tanaka, Takehiro
AU - Yamasaki, Osamu
PY - 2019/1/1
Y1 - 2019/1/1
N2 - Background. Primary malignant melanoma of the lacrimal sac is rare. A patient with lacrimal sac melanoma was presented, and 14 Japanese patients with lacrimal sac melanoma in the literature were reviewed. Case Presentation. A 78-year-old Japanese man was presented with painless swelling of the lacrimal sac on the left side. Dacryocystectomy revealed diffuse infiltration with large epithelioid cells, sometimes with pigments, which were positive for cocktail mix of antibodies to tyrosinase, melan A (MART-1), and HMB45, leading to pathological diagnosis of melanoma. One month later, positron emission tomography (PET) revealed 2 high-uptake sites (SUVmax = 10.29 and 15.38) at the levels of medial canthus and nasolacrimal duct, but no abnormal uptake in the other site of the body. The lesion had the BRAF V600E mutation. He began to take daily oral dabrafenib (BRAF inhibitor) and trametinib (MEK inhibitor), leading to no abnormal uptake on PET in half a year. He had stable disease in good physical status with small and weak uptake sites of lymph nodes on PET 1 year later. Results. In the review of 15 Japanese patients, including this patient, local recurrence was noted in 4 patients, regional lymph node metastasis only in 3, distant metastasis in 6, and no metastasis in 6. Five patients died within 2 years and the others were alive in short follow-up periods. Conclusions. Chemotherapy was the standard for local recurrence or metastasis. Emerging molecular target drugs, as shown in the present patient, would change the strategy for management of lacrimal sac melanoma.
AB - Background. Primary malignant melanoma of the lacrimal sac is rare. A patient with lacrimal sac melanoma was presented, and 14 Japanese patients with lacrimal sac melanoma in the literature were reviewed. Case Presentation. A 78-year-old Japanese man was presented with painless swelling of the lacrimal sac on the left side. Dacryocystectomy revealed diffuse infiltration with large epithelioid cells, sometimes with pigments, which were positive for cocktail mix of antibodies to tyrosinase, melan A (MART-1), and HMB45, leading to pathological diagnosis of melanoma. One month later, positron emission tomography (PET) revealed 2 high-uptake sites (SUVmax = 10.29 and 15.38) at the levels of medial canthus and nasolacrimal duct, but no abnormal uptake in the other site of the body. The lesion had the BRAF V600E mutation. He began to take daily oral dabrafenib (BRAF inhibitor) and trametinib (MEK inhibitor), leading to no abnormal uptake on PET in half a year. He had stable disease in good physical status with small and weak uptake sites of lymph nodes on PET 1 year later. Results. In the review of 15 Japanese patients, including this patient, local recurrence was noted in 4 patients, regional lymph node metastasis only in 3, distant metastasis in 6, and no metastasis in 6. Five patients died within 2 years and the others were alive in short follow-up periods. Conclusions. Chemotherapy was the standard for local recurrence or metastasis. Emerging molecular target drugs, as shown in the present patient, would change the strategy for management of lacrimal sac melanoma.
KW - BRAF inhibitor
KW - BRAF mutation
KW - dabrafenib
KW - lacrimal sac
KW - malignant melanoma
KW - MEK inhibitor
KW - PET/CT
KW - trametinib
UR - http://www.scopus.com/inward/record.url?scp=85075611483&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85075611483&partnerID=8YFLogxK
U2 - 10.1177/2324709619888052
DO - 10.1177/2324709619888052
M3 - Article
C2 - 31747798
AN - SCOPUS:85075611483
VL - 7
JO - Journal of Investigative Medicine High Impact Case Reports
JF - Journal of Investigative Medicine High Impact Case Reports
SN - 2324-7096
ER -