Sinonasal Leiomyosarcoma: Clinicopathological Analysis of Nine Cases with Emphasis on Common Association with Other Mali
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ORIGINAL PAPER
Sinonasal Leiomyosarcoma: Clinicopathological Analysis of Nine Cases with Emphasis on Common Association with Other Malignancies and Late Distant Metastasis Abbas Agaimy1 · Sabine Semrau2 · Michael Koch3 · Lester D. R. Thompson4 Received: 5 November 2017 / Accepted: 8 December 2017 © Springer Science+Business Media, LLC, part of Springer Nature 2017
Abstract Sinonasal tract (SNT) leiomyosarcoma (LMS) is exceedingly rare with 10 mitoses/10 high power fields (Fig. 1d)] and areas of tumor necrosis, two
43 F
61 M 28 M
26 F
27 M
66 F
66 M
3
4 5
6
7
8
9
Wide excision
Wide excision
Wide excision
Radical maxillect-omy
Surgery Surgery
Excision and radiochemotherapy
M male, F female, GVHD graft-versus-host-disease
Maxillary sinus
Nasal cavity
Maxillary sinus
Maxillary sinus
Nasal cavity Maxillary sinus, infiltrating dura
Right nasal cavity
Left nasal cavity + sinuses
Hodgkin Lymphoma (Stage IIIB in 3/2003) Allogenic stem cell transplantation after recurrent Hodgkin (2011) Bad GVHD Retinoblastoma (bilateral) at 18 months (bilateral blindness) Macrocytosis Intellectual disability with epilepsy/seizures Grade 1, Stage 1A uterine endometrioid adenocarcinoma (1999) Polyostotic fibrous dysplasia
Not known Not known
Yes: irradiation for fibrous No dysplasia at a younger age
Brain extension resulted in death from disease at 0.5 years
Alive without evidence of disease at 6.6 years
No
No
Local recurrence at 2 years which resulted in brain invasion Dead of disease at 2.4 years
No
Chemotherapy then stem cell transplant in 2004
55 months later, there was resection of multiple lung metastases of both rectal cancer and leiomyosarcoma Currently, alive with disease at 88 months Local recurrence middle turbinate (26 months) Two metastases to the right lower lung lobe (13 years) Alive with no evidence of disease at 17 years At 16 months, imaging showed solitary lung metastasis, cerebral metastasis and local recurrence (not verified histologically) Lost to follow-up thereafter Not known Multiple recurrences over 16 months, then lost to follow-up Lung metastases at 20 months, treated with surgery Alive without evidence of disease at 8.2 years
Follow up and outcome
Mother had retinoblaYes: for retinoblastoma; 2 stoma and rhabdo-myotimes: at initial and then sarcoma at age 2 after second eye developed retinoblastoma
Not known Not known
Not known Not known
No
No
No
Anti-androgen therapy for prostate cancer
No
Family history
Previous irradiation or chemotherapy?
Retinoblastoma of right eye Radiotherapy for retinoat age 3 blastoma
Tubulovillous rectal adenoma, 13 years later
48 F
2
Excision and adjuvant radiochemo-therapy
Prostatic carcinoma at age 73 Sinonasal leiomyosarcoma at age 77 Rectal carcinoma at age 81
Excision and radiotherapy
77 M
1
Right nasal cavity + maxillary sinus
Associated tumors
Treatment
No. Age sex Site
Table 1 Clinicopathological features of sinonasal leiomyosarcomas (n = 9)
Head and Neck Pathology
13
Head and Neck Pathology
Fig. 1 Sinona
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