Biphenotypic Sinonasal Sarcoma: Literature Review of a Peculiar Pathological Entity—The Neurosurgical Point of View
Simple Summary
Abstract
1. Introduction
2. Methods
Statistical Analysis
3. Results
3.1. Demographic, Clinical, Neuroradiological and Pathological Data (Table 1 and Table 3, Figure 2)
3.2. Treatment and Outcome Data (Table 2 and Table 4, Figure 2)
4. Discussion
- Slow growth: mean time from clinical onset to treatment is 12 months (even if cases up to 3 years are described (Table 2)); nevertheless, considering the small sizes and the function of the common sites of origin of the tumor—such as nasal cavity and paranasal sinuses—as well as of the adjacent structures usually involved—such as the orbit—it is easy to understand that a tumor becomes symptomatic quite early;
- Local aggressiveness: the tumor invades and destructs adjacent structures, both bony and soft tissues, including the medial wall, floor and roof of the orbit, cribriform plate, and orbital fat; therefore, a prompt and proper diagnosis and treatment are mandatory to prevent neuro-ophthalmological complications, such as CSF leak, meningitis, meningocele, seizures, pneumocephalus, anosmia, proptosis, and diplopia;
- Infiltrative pattern of growth: this makes it hard to achieve clear margins after surgical excision despite the high rates of gross total resection and low rate of peri- and post-operative complications;
- Long time to and very low frequency of malignant transformation;
- Tendency to locally recur: Recurrence was observed in patients regardless of the extent of tumor resection and the administration of adjuvant radiotherapy; there is no significant evidence to support the need for concomitant radiotherapy or surgical excision alone. Post-operative RT is mainly adopted when the examination of the surgical margins is found to be positive or inconclusive [6]. Therefore, it is important to collect further studies with large case series and long follow-up to analyze the main risk factors for recurrence.
- No distant metastasis.
Limitations and Advantages of This Study
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
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Studies | Demographic and Clinical Data | Radiological Data | Diagnosis | |||||
---|---|---|---|---|---|---|---|---|
Authors/Year | Num of Cases | Sex, Mean Age (Years) | Presenting Symptoms | Anatomical Origin | Skull Base Involvement | Orbit Involvement | ||
1 | Lewis et al. [1], 2012 | 28 | 21 F 7 M (52 years) | Breath difficulty, congestion, facial pressure | 19 ES, 8 NC, 1 SS. | 3 YES (ACF) | 7 YES | Immunohistochemical |
2 | Powers et al. [7] 2015 | 1 | M, 59 | Sinusitis, congestion, facial pressure, anosmia, dysgeusia | ES-NC | YES (ACF) | None | Immunohistochemical |
3 | Rooper et al. [8] 2016 | 11 | 8 F 3 M (44 years) | n.a. | 4 ES 3 FS 3 NC 1 ES-NC | None | 2 YES | Immunohistochemical molecular |
4 | Wong et al. [9] 2016 | 1 | M, 33 | Recurrent brisk epistaxis | NC-SS | None | None | Immunohistochemical molecular |
5 | Huang et al. [10] 2016 | 7 | 4 M 3 F (52 years) | n.a. | 2 FS 2 ES-NC 2 NC 1 ES | None | None | Immunohistochemical molecular |
6 | Cannon et al. [11] 2017 | 3 | 3 F (67.6 years) | Diplopia, facial discomfort, supraorbital swelling nasal obstruction, facial pressure | 3 FS-ES | 3 YES | 3 Lamina papiracea | Immunohistochemical molecular |
7 | Lin et al. [12] 2017 | 1 | F, 67 | Nasal obstruction | ES-FS-SS-MS | YES | None | Immunohistochemical |
8 | Hockstein et al. [36] 2018 | 1 | F, 79 | Asymptomatic | FS | YES | Roof | Immunohistochemical |
9 | Andreasen et al. [13] 2018 | 3 | 2 F, 1 M (59.6 years) | Nasal obstruction and midfacial pressure | 2 ES 1 ES-NC | None | None | Immunohistochemical |
10 | Koszewski et al. [35] 2018 | 1 | M, 53 | Unilateral nasal obstruction and epiphora | NC | YES (ACF) | Lamina papiracea | Immunohistochemical |
11 | Kakkar et al. [14] 2018 | 6 | 5 F, 1 M (51 years) | Nasal obstruction | 1 NC 1 NC, MS 1 NC, MS, ES 1 NC, MS, ES 1 NC, MS, ES, FS 1 NC, ES | 1 YES | None | Immunohistochemical |
12 | Quadros et al. 2019 | 1 | F, 55 | Obstruction of the left nasal cavity | NC | None | None | Immunohistochemical |
13 | Chitguppi et al. [6] 2019 | 1 | M, 53 | n.a. | ES-NC | YES | YES | Immunohistochemical molecular |
14 | Alkhudher et al. [16] 2019 | 1 | F, 35 | Nasal obstruction, epistaxis | NC, MS, ES | None | Lamina papiracea | Immunohistochemical |
15 | Miglani et al. [34] 2019 | 5 | 4 F, 1 M (56 years) | n.a. | 5 NC-ES | 5 YES (ACF) | 5 Lamina Papiracea | Immunohistochemical |
16 | Fudaba et al. [15] 2019 | 1 | M, 70 | Loss of consciousness and vomiting | ES | YES | None | Immunohistochemical molecular |
17 | Le Loarer et al. [17] 2019 | 41 | 16 M, 25 F (51 years) | n.a. | 14 NC 11 ES 10 ES-FS 6 n.a. | 4 YES | 4 YES | Immunohistochemical molecular |
18 | Kuhn et al. [33] 2019 | 1 | n.a. | Worsening nasal obstruction, rhinorrhea, left orbital pain, proptosis and blurry vision | NC-ES | YES (ACF) | Lamina papiracea | Immunohistochemical molecular |
19 | Okafor et al. [32] 2020 | 1 | M, 54 | Left-sided nasal airway obstruction and anosmia | NC-MS-ES-FS | YES (ACF) | Lamina papiracea | Immunohistochemical |
20 | Okuda et al. [31] 2020 | 1 | F, 64 | Nasal obstruction | NC-MS-ES pterygopalatine fossa | YES (MCF) | YES | Immunohistochemical |
21 | Sethi et al. [18] 2021 | 3 | 3 F (56 years) | Left-sided nasal congestion and headaches/right nasal obstruction/rhinorrhea and left-sided nasal congestion | 3 ES-MS-FS-NC | 1 YES (ACF) | 2 YES | Immunohistochemical |
22 | Hanbazazh et al. [19] 2021 | 1 | M, 50 | Orbital pain and pressure, diplopia, blurred vision, lateral gaze restriction | ES | YES | Lamina papiracea | Immunohistochemical molecular |
23 | Bell et al. [20] 2022 | 1 | M, 66 | Swelling of left eyelid, vertical diplopia and purulent nasal discharge | NC | YES (ACF) | YES | Immunohistochemical molecular |
24 | Hasnie et al. [21] 2022 | 1 | F, 72 | Nasal obstruction, episodic epistaxis and facial pressure/headaches, decreased sense of smell | MS-ES-Bilateral FS-NC | YES (ACF) | Lamina papiracea | Immunohistochemical molecular |
25 | Turri-Zanoni et al. [22] 2022 | 15 | 3 M, 12 F (54 years) | 14 nasal airway obstruction 9 epistaxis, 6 olfactory disfunction3 facial pain | 13 ES 2 FS | None | None | Immunohistochemical molecular |
26 | Nichols et al. [23] 2023 | 1 | M, 54 | Persistent headaches, postnasal drip, thickened nasal secretions, and epistaxis after sneezing | ES-SS | None | None | Immunohistochemical molecular |
27 | Ingle et al. [24] 2023 | 1 | F, 47 | Swelling eyelid, proptosis | NC, FS, ES, MS | None | Lamina papiracea | Immunohistochemical |
28 | Meyer et al. [25] 2023 | 1 | M, 67 | Nasal congestion and epiphora, right-sided ocular proptosis | ES-MS-FS | None | YES | Immunohistochemical molecular |
29 | Kominsky et al. [26] 2023 | 2 | 2 M (65 years) | Bilateral nasal congestion and blurry vision | ES-NC-FS | 2 YES | 2 Lamina papiracea | Immunohistochemical molecular |
30 | Bhele et al. [27] 2023 | 1 | F,22 | Vision loss, headache, hyposmia, facial pressure | NC-ES-SS-MS | YES (ACF) | Lamina papiracea | Immunohistochemical |
31 | Viramontes et al. [28] 2023 | 1 | F, 40 | Progressive obstruction of the right nasal cavity, | NC | None | None | Immunohistochemical molecular |
32 | Muraoka et al. [29] 2023 | 1 | F, 73 | Purulent nasal discharge and dull pain in the left cheek area | NC-ES-FS | YES (ACF) | None | Immunohistochemical molecular |
33 | Anastasiadou et al. [30] 2023 | 3 | 3 F (43 years) | Exophthalmos, headaches | NC-MS | 1 YES | 2 YES | Immunohistochemical molecular |
34 | Corvino et al. [37] 2024 | 1 | M, 46 | l. proptosis, upward gaze restriction | FS-ES | YES (ACF) | Roof | Immunohistochemical |
Studies | Treatment Data | Outcome Data at Last Follow Up | ||||||||
---|---|---|---|---|---|---|---|---|---|---|
Authors/Year | Num of Cases | Time to Treatment | Type of Treatment | Type of Surgical Approach | EOR | Peri-Post Operative Complications | Clinics | Recurrence | Status | |
1 | Lewis et al. [1] 2012 | 28 | n.a. | n.a. | n.a. | n.a. | n.a. | n.a. | 7/16 (range 12–118 mo.) | (mean 8.3 years) 14 alive 2 dead due to other causes |
2 | Powers et al. [7] 2015 | 1 | n.a. | S | EEA | GTR | CSF leak | n.a. | None | Alive 10 mo. |
3 | Rooper et al. [8] 2016 | 11 | n.a. | n.a. | n.a. | n.a. | n.a. | n.a. | 2/7 (range 1–26 mo.) | (mean 4 years) 1/7 dead due to tumor |
4 | Wong et al. [9] 2016 | 1 | n.a. | S Ad-CHT-RT | EEA | GTR | n.a. | n.a. | None | Alive 5 mo. |
5 | Huang et al. [10] 2016 | 7 | n.a. | 6 S 1 S + Ad-CHT-RT | n.a. | 4 GTR | n.a. | n.a. | 1/4 (36 mo) | (mean 8 years) 4 alive |
6 | Cannon et al. [11] 2017 | 3 | n.a | 2 S 1 Biopsy | 1 EEA − 1 EEA + TCA1 EEA Biopsy | 2 GTR 1 STR | n.a. | n.a. | 1/3 (17 mo.) | (mean 25 mo.) 3 alive |
7 | Lin et al. [12] 2017 | 1 | n.a | S | EEA | GTR | Subarachnoid hemorrhage; brain herniation | n.a. | n.a. | Dead due to surgery |
8 | Hockstein et al. [36] 2018 | 1 | 12 mo. | S | EEA + TCA | GTR | n.a. | n.a. | None | Alive |
9 | Andreasen et al. [13] 2018 | 3 | n.a | 1S 2 S + Ad.RT | n.a. | 3 GTR | None | n.a. | 1/3 (11, 21 and 24 mo) | (mean 67.3 mo.) 3 alive |
10 | Koszewski et al. [35] 2018 | 1 | 4 mo. | S + Ad.RT | n.a. | STR | n.a. | n.a. | None | Alive |
11 | Kakkar et al. [14] 2018 | 6 | n.a. | 3 S 3 Biopsy | 3 EEA | 4 STR | n.a. | n.a. | None | 1/6 dead due to other causes |
12 | Quadros et al. 2019 | 1 | n.a | S | EEA | n.a. | n.a. | n.a. | n.a. | n.a. |
13 | Chitguppi et al. [6] 2019 | 1 | n.a. | S + Ad-RT | TCA + ETOA | STR | n.a. | n.a. | None | Alive |
14 | Alkhudher et al. [16] 2019 | 1 | 2 mo. | S | EEA | GTR | n.a. | Improved | None | Alive 2 years |
15 | Miglani et al. [34] 2019 | 5 | n.a | 4 S 1 S + Ad-RT | 3 TCA 2 EEA | 4 GTR 1 STR | n.a. | n.a. | 2/5 (mean 31.4 mo.) | (mean 31.4 mo.) 5 alive |
16 | Fudaba et al. [15] 2019 | 1 | REC after 11 years | S | EEA + TCA | GTR | n.a. | n.a. | No further | Alive |
17 | Le Loarer et al. [17] 2019 | 41 | n.a. | 20 S 8 S + RT 2 S + RT + CHT 1 RT+ CHT 2 S + CHT | n.a. | n.a. | n.a. | n.a. | 8/25 (range 9–95 mo.) | (mean 45 mo.) |
18 | Kuhn et al. [33] 2019 | 1 | n.a | S | TCA | GTR | None | n.a. | n.a. | n.a. |
19 | Okafor et al. [32] 2020 | 1 | 5 mo. | 2 S | EEA | 1 STR 1 GTR | None | n.a. | n.a. | n.a. |
20 | Okuda et al. [31] 2020 | 1 | REC after 2 mo. | S + Ad.CHT | TCA | GTR | None | n.a. | YES (after 2 mo) | Dead 8 mo., death due to tumor progression |
21 | Sethi et al. [18] 2021 | 3 | n.a | 1 S + Ad.RT 2 S | 3 EEA | 3 GTR | None | n.a. | None | 2 alive (mean 22 mo) |
22 | Hanbazazh et al. [19] 2021 | 1 | 36 mo | 1 Biopsy 1 S 1 S + Ad.RT | Biopsy EEA TO TCA | STR | None | Improved | None | Alive |
23 | Bell et al. [20] 2022 | 1 | REC after 15 years | 1 S + Ad.RT | TCA | GTR | None | Stable | No further | Alive 10 mo. |
24 | Hasnie et al. [21] 2022 | 1 | 24 mo. | S | EEA + TCA | GTR | Infection, pneumocephal | n.a. | None | Death due to other causes |
25 | Turri-Zanoni et al. [22] 2022 | 15 | n.a | 13 S 2S + RT | 7 EEA 8 EEA + TCA | 13 GTR2 STR | n.a. | n.a. | 1/15 (after 35 and 47 mo) | (27.3 months) 15 alive |
26 | Nichols et al. [23] 2023 | 1 | n.a | S | EEA | n.a. | n.a. | Improved | None | Alive 3 mo. |
27 | Ingle et al. [24] 2023 | 1 | 2 mo. | S | EEA + TCA | GTR | n.a. | n.a. | None | Alive 3 mo. |
28 | Meyer et al. [25] 2023 | 1 | 36 mo. | Biopsy, RT, CHT | EEA | Biopsy | n.a. | n.a. | Progression | Dead 15 mo., death due to tumor progression |
29 | Kominsky et al. [26], 2023 | 2 | 3 weeks (1) | 2 S | 2 EEA | 2 GTR | n.a. | n.a. | None | 2 alive (mean 13 mo.) |
30 | Bhele et al. [27] 2023 | 1 | 8 mo. | Biopsy, Neo-CHT, S, Ad-PB | TCA + EEA | STR | n.a. | n.a. | None | Alive, 10 mo. |
31 | Viramontes et al. [28], 2023 | 1 | n.a | S | EEA | GTR | n.a. | n.a. | None | Alive, 16 mo. |
32 | Muraoka et al. [29] 2023 | 1 | n.a | S | TCA + EEA | GTR | n.a. | n.a. | None | Alive |
33 | Anastasiadou et al. [30], 2023 | 3 | n.a | 1 S, 2 S + Ad.RT | 3 EEA | 3 GTR | 1 CSF leak | n.a. | None | Alive 7 years |
34 | Corvino et al. [37] 2024 | 1 | 2 mo. | S | TCA + EEA | GTR | None | Improved | None | Alive, 10 mo. |
Covariates | Overall Sample 149 (%) | Statistical Analysis (p Value) |
---|---|---|
Demographic and clinical data | ||
Sex -F -M | 148/149 * (99.3%) 99/148 (66.9%) 49/148 (33.1%) | p = 0.6 |
Age range (median) | 22–79 years (54.88 y.o.) | p = 0.04 |
Main presenting symptoms -Nasal obstruction -Facial pressure/pain/discomfort -Epistaxis -Ocular impairment | 84/149 * (56.3%) 68/84 (81%) 37/84 (44%) 13/84 (15.5%) 12/84 (14.3%) | p = 0.46 |
Radiological data | ||
Anatomical Origin -NC -ES -FS -MS -SS | 143/149 * (96%) 65/143 (45.4%) 97/143 (67.8%) 34/143 (23.7%) 18/143 (12.6%) 5/143 (3.5%) | p = 0.32 |
Skull Base involvement -Yes -Not | 143/149 * (96%) 35/143 (24.5%) 108/143 (75.5%) | p = 0.22 |
Orbit involvement -Yes -Not | 143/149 * (96%) 41/143 (28.7%) 102/143 (71.3%) | p = 0.26 |
Pathological Diagnosis | ||
Diagnostic method -immunohistochemical alone -immunohistochemical and molecular | 149/149 * (100%) 56/149 (37.6%) 93/149 (62.4%) | p = 0.55 |
Covariates | Overall Sample 149 (%) | Statistical Analysis (p Value) |
---|---|---|
Treatment Data | ||
Time to treatment (mean in months) | 11/149 (7.3%) 12 months | p = 0.11 |
Type of treatment -S -S + RT -Biopsy alone -S + CHT -S + RT + CHT -RT + CHT | 104/149 * (69.8%) 69/104 (66.3%) 20/104 (19.2%) 5/104 (4.8%) 3/104 (2.9%) 5/104 (4.8%) 2/104 (1.9%) | p = 0.43 |
Type of surgical approach -EEA -TCA -TOA -Combined | 58/149 * (39%) 33/58 (56.9%) 7/58 (12%) 1/58 (1.7%) 17/58 (29.3%) | p = 0.1 |
EOR -GTR -STR | 62/149 * (41.6%) 49/62 (79%) 13/62 (21%) | p = 0.45 |
Peri- and post-operative complications -Yes -None | 12/149 * (8%) 4/12 (33.3%) 8/12 (66.7%) | |
Outcome | ||
Clinical -Improved -Stable -Worsened | 5/149 * (3.3%) 4/5 (80%) 1/5 (20%) --- | |
Recurrence -Yes -Not | 84/149 * (56.3%) 22/84 (26.2%) 62/84 (73.8%) | p = 0.6 |
Status -Alive -Dead | 85/149 * (57%) 77/85 (91.8%) 8/85 (8.2%) | p = 0.87 |
Follow-up | Mean 4.6 years | St. Dev = 3.05 |
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Corvino, S.; Corazzelli, G.; Mariniello, G.; Iuliano, A.; Altieri, R.; Pontillo, G.; Strianese, D.; Barbarisi, M.; Elefante, A.; de Divitiis, O. Biphenotypic Sinonasal Sarcoma: Literature Review of a Peculiar Pathological Entity—The Neurosurgical Point of View. Cancers 2024, 16, 3747. https://doi.org/10.3390/cancers16223747
Corvino S, Corazzelli G, Mariniello G, Iuliano A, Altieri R, Pontillo G, Strianese D, Barbarisi M, Elefante A, de Divitiis O. Biphenotypic Sinonasal Sarcoma: Literature Review of a Peculiar Pathological Entity—The Neurosurgical Point of View. Cancers. 2024; 16(22):3747. https://doi.org/10.3390/cancers16223747
Chicago/Turabian StyleCorvino, Sergio, Giuseppe Corazzelli, Giuseppe Mariniello, Adriana Iuliano, Roberto Altieri, Giuseppe Pontillo, Diego Strianese, Manlio Barbarisi, Andrea Elefante, and Oreste de Divitiis. 2024. "Biphenotypic Sinonasal Sarcoma: Literature Review of a Peculiar Pathological Entity—The Neurosurgical Point of View" Cancers 16, no. 22: 3747. https://doi.org/10.3390/cancers16223747
APA StyleCorvino, S., Corazzelli, G., Mariniello, G., Iuliano, A., Altieri, R., Pontillo, G., Strianese, D., Barbarisi, M., Elefante, A., & de Divitiis, O. (2024). Biphenotypic Sinonasal Sarcoma: Literature Review of a Peculiar Pathological Entity—The Neurosurgical Point of View. Cancers, 16(22), 3747. https://doi.org/10.3390/cancers16223747