Table I.
Case series of lacrimal pathway tumours.
| Authors | No. of cases | Histological diagnosis | Surgical strategy | RT/CTx | Neck dissection | Recurrence |
|---|---|---|---|---|---|---|
| Wakasaki (2023) 27 | 25 | Benign
1 inverted papilloma 2 exophytic papilloma Malignant 6 SCC 2 adenoid cystic carcinoma 2 sebaceous carcinoma 1 mucoepidermoid carcinoma 11 lymphoma |
The initial treatment of benign (n = 3) and epithelial malignancies (n = 22) was surgery. Extended total maxillary resection with eyeball resection was performed in 2 SCC and one sebaceous carcinoma.
11 of the 14 patients underwent free-flap mandibular reconstructions following resection |
3 RT
5 CTx |
2 SCC underwent neck dissection.
One patient pN+ (submental and submandibular lymph nodes, no extranodal extension) |
3 of 22 patients with malignant tumour had locoregional recurrence, but only one patient with sebaceous carcinoma died of locoregional and brain/bone metastases. Two patients survived with distant metastases (one basaloid SCC and one adenoid cystic carcinoma) |
| Bai (2023) 42 | 15 | 15 lymphoma | 15 tumour resections
2 combined with DCR |
12 post-CTx | - | - |
| Sun (2022) 43 | 8 | 8 mucoepidermoid carcinoma | 6 tumours were managed with radical resection and 2 patients underwent orbital exenteration | 5 RT
1 RT+CTx |
- | 1 recurrence
2 metastases |
| Næser (2022) 44 | 1 | 1 angioleiomyoma | External DCT combined with endonasal, endoscopic anterior turbinectomy, and nasal mucosal resection | - | - | - |
| Meng (2022) 45 |
8 | 8 lymphoma | 3 Incisional biopsy
4 DCT 1 Medial maxillectomy |
5 RT
3 CTx |
- | - |
| Kim (2022) 46 | 1 | 1 large B-cell lymphoma | Lacrimal sac excision | - | - | Recurrence after 2 months, CTx |
| Kadir (2022) 1 | 10 | 1 papilloma
1 RLH 1 SFT 4 NHL 1 SCC 1 adenocarcinoma 1 mucoepidermoid carcinoma |
5 DCT
2 Incisional biopsy |
5 RT
2 CTx |
- | - |
| Amjad (2022) 47 | 1 | 1 small cell neuroendocrine carcinoma | - | CTx-RT | - | - |
| Almutairi (2022) 48 | 1 | 1 oncocytoma | DCT | - | - | Recurrence at 3 years |
| Turri-Zanoni (2022) 36 | 1 | 1 ectopic olfactory neuroblastoma | DCR, total ethmoidectomy, and medial maxillectomy | RT | - | No after 12 months |
| Howden (2021) 49 | 1 | 1 oncocytoma | Extended right Lynch incision | - | - | - |
| Azhdam (2021) 34 | 1 | 1 angioleiomyoma | 1 endoscopic excision of the lesion along with medial maxillectomy and DCR | - | - | - |
| Song (2020) 50 | 17 | 17 SCC | - | 17 RT
11 CTx |
- | 1 recurrence |
| Sharma (2020) 51 | 1 | 1 epithelial-myoepithelial carcinoma | Combined endoscopic endonasal and open approach | - | - | No after 20 months |
| Morawala (2020) 52 | 3 | 3 SFT | External and endoscopic approaches were used to assess the orbital clearance and manage the sinonasal extension and the nasolacrimal duct excision up to the inferior meatus | RT | - | - |
| Miller (2020) 53 | 1 | 1 transitional cell carcinoma | Lacrimal sac excision and medial maxillectomy | RT | - | No after 5 years |
| Cheang (2020) 54 | 1 | 1 inverted papilloma | Combined endonasal and external approach | - | - | - |
| Chang (2020) 33 | 1 | 1 SCC | Image-guided surgery, navigation-assisted transnasal endoscopic-assisted excision | RT+CTx | - | Recurrence after 3 months |
| Alam (2020) 55 | 1 | 1 apocrine adenocarcinoma | Orbital exenteration | RT | - | No after 1 year |
| Zhang (2019) 56 | 18 | 9 SCC
7 poorly differentiated carcinoma 1 transitional cell carcinoma 1 adenoid cystic carcinoma |
En bloc resection of the lacrimal sac malignancy was performed in all patients with concurrent partial ethmoidectomy in 8 patients and medial maxillectomy in 5 patients | 18 CTx+RT | - | - |
| Wu (2019) 57 | 1 | 1 sebaceous carcinoma | Combined external and endoscopic approach | - | - | No after 3 months |
| Ucgul (2019) 58 | 2 | 2 MALT lymphoma | Lacrimal sac excision within the tumour, canalicular DCR with bicanalicular silicone intubation | 1 CTx+RT
1 RT |
- | 1 recurrence |
| Purser (2019) 59 | 1 | 1 inverted papilloma | Endoscopic DCR | - | - | - |
| Morawala (2019) 60 | 1 | 1 adenocarcinoma | Combined endonasal and external approach | RT | - | No after 10 months |
| Matsuo (2019) 25 | 15 | 15 melanoma | 14 DCT
1 exenteratio orbitae |
7 CTx
2 RT 1 Immunotherapy |
3 (1 submandibular and pre-auricular nodes; 1 laterocervical nodes; 1 submandibular nodes) | 4 recurrences |
| Gervasio (2019) 61 | 1 | 1 inverted papilloma | Combined endonasal and external approach | - | - | - |
| Bowen (2019) 62 | 1 | 1 adenoid cystic carcinoma | DCT, anterior ethmoidectomy, and medial maxillectomy | RT | - | No after 5 years |
| Song (2018) 21 | 90 | 69 SCC
7 lymphoma 3 adenocarcinoma 3 adenoid cystic carcinoma 2 melanoma 2 transitional cell carcinoma 1 mucoepidermoid carcinoma 1 malignant neurilemmoma 1 haemangioendothelioma 1 Merkel cell carcinoma |
40 local tumour resections
26 extended tumour resections 3 orbital exenterations |
21 RT definitive
26 post-RT 43 pre-RT 50 RT+CTx |
Positive lymph nodes status at diagnosis significantly associated with a worse overall survival and progression-free survival (the number of patients undergoing neck dissection is not reported) | 22 recurrences |
| Nomura (2018) 63 | 1 | 1 adenocarcinoma and transitional cell carcinoma | Lateral rhinotomy | - | - | Recurrence, RT |
| Khanna (2018) 64 | 1 | 1 melanoma | Lateral rhinotomy approach, including the nasolacrimal duct, part of the lateral wall of the nose, the lacrimal sac, distal upper and lower canaliculi, and inferomedial orbital fat | RT | - | - |
| Haft (2018) 35 | 1 | 1 pleomorphic adenoma | Combined endonasal and external approach | - | - | - |
| Curragh (2018) 65 | 1 | 1 SCC | Combined external and endoscopic prelacrimal approach | - | - | - |
| Subramaniam (2017) 26 | 1 | 1 melanoma | Maxillectomy, exenteratio orbitae with radial forearm free flap reconstruction | RT | Supra-omohyoid neck dissection (pN+) | - |
| Mikhail (2017) 66 | 1 | 1 extranodal NK/T-cell lymphoma | External DCR | RT+CTx | - | - |
| Krishna (2017) 8 | 3 | 3 SLL | Incisional biopsy by DCR | CTx+RT | - | - |
| Jakobiec (2017) 67 | 1 | 1 lymphoepithelial carcinoma | DCR | - | - | Recurrence at 2 years: combined transnasal and transconjunctival diagnostic debulking of the tumour + RT-CTx for regional cervical lymph nodes metastases |
| Huggins (2017) 68 | 1 | 1 basaloid adenocarcinoma | DCT | RT | - | - |
| Grumbine (2017) 69 | 1 | 1 smooth muscle tumour of uncertain malignant potential | DCT | - | - | - |
| Dave (2017) 70 | 1 | 1 transitional cell papilloma | DCT | - | - | - |
| Zarrabi (2016) 71 | 1 | 1 large B cell lymphoma | External excisional biopsy | RT+CTx | - | - |
| Watanabe (2016) 72 | 2 | 2 haemangiopericytoma | DCT | - | - | One case recurred 3 times during an 18-year period. The other did not recur during 51 months of follow-up |
| Roos (2016) 29 | 1 | 1 mucoepidermoid carcinoma | Excision of the lacrimal sac, nasolacrimal duct, paranasal sinuses | - | Radical neck dissection + parotidectomy | Metastasis in the right cavernous sinus at 6 months,
gamma knife RT |
| Ramos (2016) 38 | 1 | 1 adenoid cystic carcinoma | Lateral rhinotomy, osteotomy from the canthal ligament to the orbital floor. The orbital floor was reconstructed | RT | - | - |
| Park (2016) 73 | 1 | 1 adenocarcinoma | Right medial maxillectomy including removal of the inferior and middle turbinates and partial orbital exenteration | RT | - | No after 25 months |
| Moriyama (2016) 74 | 1 | 1 SFT | Combined approach with endoscopic-modified medial maxillectomy and external incision | - | - | - |
| Marunaka (2016) 75 | 1 | 1 large B-cell lymphoma | Endoscopic biopsy | RT+CTx | - | No after 3 months |
| Janakiram (2016) 37 | 1 | 1 mucoepidermoid carcinoma | External maxillectomy and forehead flap reconstruction | RT+CTx | - | - |
| Bousaadani (2016) 76 | 1 | 1 epidermoid carcinoma | Combined approach with endoscopic-modified medial maxillectomy and external incision | RT | - | No after 2 years |
| Belliveau (2016) 77 | 1 | 1 ectopic olfactory neuroblastoma | Resection of the lateral nasal wall and medial maxilla en bloc, endonasal DCR and DCT | RT | - | No after 17 months |
| Walijee (2015) 78 | 1 | 1 inverted papilloma | Combined external and endoscopic approach | - | - | - |
| Wada (2015) 31 | 1 | 1 adenoid cystic carcinoma | - | Proton beam therapy | - | - |
| Vagia (2015) 79 | 1 | 1 adenocarcinoma | Maxillectomy and exenteratio orbitae | RT+CTx | Neck dissection + parotidectomy | Recurrence at 2 years: second line CTx, then third line CTx and then anti-androgens drugs (abiraterone) with good clinical response |
| Tsao (2015) 80 | 1 | 1 large B cell lymphoma | Endoscopic biopsy | RT+CTx | - | - |
| Satchi (2015) 81 | 5 | 5 melanomas | Case 1: orbital exenteration and lateral rhinotomy with en bloc excision of the orbital contents and entire lacrimal pathway
Case 2: orbital exenteration and DCT with a combined external and endonasal procedure. Case 3: orbital exenteration with en bloc excision of the entire lacrimal system within the medial wall of the orbit and maxilla. Case 4: orbital exenteration. Case 5: orbital exenteration |
- | - | Case 1: died within 8 months for metastases.
Case 2: developed metastasis in the parotid gland 4 years after exenteration and underwent a superficial parotidectomy and radical neck dissection. Case 3: local recurrence in the maxilla and lateral nasal wall 4 years after exenteration: resection of the lesion and RT. Case 4: en bloc excision of the medial orbit and medial maxilla for recurrence after 2 years. Case 5: recurrence after 3 years: en bloc resection of medial orbital wall, lateral nasal wall and medial maxilla, radial forearm free flap reconstruction and postoperative RT. |
| Papastefanou (2015) 82 | 1 | 1 small lymphocytic lymphoma | Endoscopic biopsy | CTx | - | - |
| Litschel (2015) 83 | 4 | 4 small cell lymphocytic lymphomas | Biopsy by endoscopic DCR | RT+CTx | - | - |
| Lee (2015) 84 | 2 | 2 SCC | Wide resection of lacrimal sac and nasolacrimal duct via anterior orbitotomy and medial maxillectomy via lateral rhinotomy | RT | - | - |
| Lee (2015) 85 | 1 | 1 benign mixed tumour | DCT | - | - | - |
| Iordanous (2015) 86 | 1 | 1 mucoepidermoid carcinoma associated to squamous/transitional cell papilloma | Maxillectomy | - | - | - |
| Higashi (2015) 24 | 1 | 1 melanotic neuroectodermal tumour | External maxillectomy | RT+CTx | - | - |
| Hardy (2015) 87 | 1 | 1 inverted papilloma | Maxillectomy | - | - | - |
| Abdelkhalek (2015) 88 | 2 | 2 MALT lymphomas | - | RT | - | - |
| Neffendorf (2014) 89 | 1 | 1 rhabdomyosarcoma | - | RT+CTx | - | - |
| Linxweiler (2014) 90 | 1 | 1 exophytic Schneiderian carcinoma | DCT+DCR | - | - | - |
| Kurdi (2014) 91 | 1 | 1 SFT | DCT+DCR | - | - | - |
| Guo (2014) 92 | 5 | 5 lymphomas | - | RT+CTx | - | - |
| Erickson (2014) 18 | 1 | 1 transitional cell-type papillary carcinoma | Medial maxillectomy, inferior turbinectomy, ethmoidectomy, and partial rhinectomy, reconstruction by a paramedian forehead flap and myocutaneous cheek advancement flap | RT | - | No after 1 year |
| Yuksel (2013) 93 | 1 | 1 mucoepidermoid carcinoma | The tumour, lacrimal sac, and nasolacrimal duct were removed en bloc with the frontal process of the maxilla, lateral nasal wall, lacrimal fossa, ethmoids, and anterior part of the medial orbital wall | RT | - | - |
| Mulay (2013) 94 | 1 | 1 oncocytoma | DCT | - | - | - |
| Maegawa (2013) 95 | 1 | 1 melanoma | Lateral rhinotomy, en bloc excision, including the lacrimal apparatus, medial wall of the orbit, and medial upper and lower lids, and medial maxillectomy. A T-shaped, median forehead flap repaired the defect | RT+CTx | - | - |
| Kim (2013) 32 | 1 | 1 osteoma | Endoscopic DCR | - | - | - |
| Jung (2013) 96 | 1 | 1 oncocytic carcinoma | Combined approach: DCT+ endoscopic inferior medial maxillectomy, including medial wall of maxillary sinus, inferior turbinate, and lateral wall of nasal cavity | RT | - | - |
| Islam (2013) 30 | 1 | 1 bilateral metachronous SCC arising within inverted papilloma | Orbital exenteration, medial
maxillectomy and radial forearm free flap reconstruction |
- | Selective left (I-III) neck dissection (pN0) | After 2 years, excision of the contralateral lacrimal sac, lamina papyracea and anterior and posterior ethmoids for a SCC arising within an IP extended to the orbit. Died for systemic metastasis after 1 year |
| Ishida (2013) 28 | 1 | 1 adenocarcinoma | DCT and exenteratio orbitae | - | Submandibular, and upper-, mid-, and lower-internal deep cervical lymph nodes (pN+) | - |
| Hodgson (2013) 97 | 1 | 1 bilateral SCC | Medial maxillectomy and DCT through a lateral rhinotomy approach | - | - | No after 6 months |
| Gustafson (2013) 98 | 1 | 1 synchronous verrucous carcinoma and inverted papilloma | Endoscopic debulking and DCT | - | - | - |
| Eweiss (2013) 99 | 20 | 10 transitional cell papillomas
10 transitional cell carcinomas |
Lateral rhinotomy | 7 RT | - | 4 recurrences |
| Donnadieu (2013) 100 | 1 | 1 oncocytoma | DCT | - | - | - |
| Chai (2013) 101 | 1 | 1 lymphoma | - | RT+CTx | - | No after 1 year |
| Azari (2013) 102 | 1 | 1 transitional cell carcinoma | DCT | - | - | - |
| Original case report | 1 | 1 SCC arising within inverted papilloma | DCR, medial maxillectomy and anterior ethmoidectomy | - | - | No after 6 months |
| RLH: reactive lymphoid hyperplasia; SFT: solitary fibrous tumour; NHL: non-Hodgkin’s lymphoma; SCC: squamous cell carcinoma; DCT: dacryocystectomy; CTx: chemotherapy; RT: radiotherapy; SLL: small lymphocytic lymphoma; DCR: dacryocystorhinostomy; IP: inverted papilloma. | ||||||