NK/T-Cell Lymphoma of the Nasal Cavity Causing Contralateral Dacryoadenitis

Luemsamran, Panitee; Pornpanich, Kanograt; Uiprasertkul, Mongkol; Sakolsatayadorn, Natta; Vangveeravong, Sumalee

doi:10.3109/01676830.2013.788665

Cited by 9 publications

(11 citation statements)

References 5 publications

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“…Staphyloccus aureus ( 30 ), Nocardia farcinica ( 31 ), Mycobacterium fortuitum, M. marinum ( 32 ), Leishmania infantum ( 33 ), and EBV ( 9 – 11 , 34 ) are among the suspected causative agents. Mucoralean fungi have been reported to cause infections with highly similar clinical features ( 1 , 24 – 26 ). Particularly, M. irregularis ( 28 , 29 ) shows resemblance because this species causes chronic infection in patients without an apparent underlying disease, in contrast to other Mucoralean members which are usually acute in patients with leukemia or ketoacidotic diabetes.…”

Section: Discussionmentioning

confidence: 99%

“…Some LMG patients received chemotherapy, but post-mortem , the cause of their disorder was shown to be fungal ( 24 , 25 ). In patients primarily diagnosed with ROCM, the detection of NK cells and/or T cells in the tissue led to a change of diagnosis to from ROCM to LMG, with a fatal outcome ( 1 , 26 ). In another paper, two ROCM cases were reported with NK/T cells and angiocentric characteristics.…”

Section: Introductionmentioning

confidence: 99%

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Case Report: Rhizopus arrhizus Rhino-Orbital-Cerebral Mycosis and Lethal Midline Granuloma: Another Fungal Etiological Agent

Lun

et al. 2021

Front. Med.

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Objective: Both rhino-orbital-cerebral mycosis and lethal midline granuloma (LMG) may result in midline destruction. LMG has now been generally considered as a natural killer/T cell lymphoma, nasal type (ENKTL-NT) with an association of EBV. Fungi have been detected from the diseased tissues now and then but are often considered as lymphoma-associated infections. We previously reported an ENKTL-NT case with Mucor irregularis, which played a causal role in the disease and was involved in the overexpression of Ki67 and CD56 in the mouse experiment. The present study describes a chronic Rhizopus arrhizus infection with immunological parameters that are closely similar to LMG. We aim to explore the relationship of another Mucorales fungus, R. arrhizus, and LMG in a patient and in mice.Methods: Case study and mouse infection modules were designed for our observation. A 35-year-old man with midline face ulcers which was clinically suspected as LMG was selected. Biopsy specimens were sent for lymphoma diagnosis and microbiological detection. The isolated fungus was tested in an ICR mouse model for mycological and histological analyses.Results: Five tissue samples yielded Rhizopus arrhizus. In the pathology, characteristic inflammation, necrosis, and granulation with thin-walled hyphae are observed. Immunohistochemistry showed NK/T cell infiltration (CD3+, CD8+, TIA1+, GZMB+, PRF+, individual CD56+) with hyperplasia (Ki67+) and angioinvasion. The patient recovered completely with amphotericin B. In the murine experiment, R. arrhizus caused angioinvasion with NK/T cell infiltration (CD3+, CD56+, TIA1+, GZMB +, PRF+) with proliferation (Ki67+) and was re-isolated from the infected host.Conclusions: We here describe a mid-face destruction patient, which was diagnosed by the top pathologists in China according to the current criteria of NK/T cell lymphoma, with a negative result for EBV and positive result for R. arrhizus. With a then developed mouse experiment, the R. arrhizus in the diseased lesions was responsible for the NK/T cell infiltration (CD3+, CD8+, CD56+, TIA1+, GZMB+, PRF+), proliferation (Ki67+), and angioinvasion, suggesting another fungal etiological agent for LMG, which could be eradicated with amphotericin B.Limitations: The sample size is not sufficient for statistical analysis. However, our findings are suggestive for the role fungus plays in LMG.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Case Report: Rhizopus arrhizus Rhino-Orbital-Cerebral Mycosis and Lethal Midline Granuloma: Another Fungal Etiological Agent

Lun

et al. 2021

Front. Med.

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“…The sample should be forwarded to an experienced pathologist for assessment while fresh [ 1 , 24 ]. Establishing the diagnosis is difficult because NK/T-cell lymphoma displays extensive necrosis and a small number of cancer cells in the affected area [ 15 , 25 ].…”

Section: Discussionmentioning

confidence: 99%

“…An infection caused by P. Aeruginosa can mimic NK/T-cell lymphoma [ 5 ], as was the case with our patient. Cases suggestive of periorbital cellulitis were also reported before establishing the definitive diagnosis of NK/T-cell lymphoma [ 5 , 18 , 25 , 27 ].…”

Section: Discussionmentioning

confidence: 99%

Natural Killer (NK)//T-Cell Lymphoma, Nasal Type, with Periorbital Involvement: A Case Report and Literature Review

et al. 2020

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Patient: Female, 36-year-old Final Diagnosis: NK/T-cell lymphoma Symptoms: Nasal congestion • rhinorrea • sinus congestion Medication: — Clinical Procedure: — Specialty: Otolaryngology Objective: Rare disease Background: Extranodal natural killer (NK)/T-cell lymphomas are rare neoplasms that result in the destruction of mid-facial tissues. Infrequently seen in ear, nose, and throat clinical practice, they are often misdiagnosed and treated as chronic sinusitis, which delays proper diagnosis. This study aimed to describe the insidious course of NK/T-cell lymphomas, nasal type. Case Report: A 36-year-old woman with a 2-year history of recurrent exacerbation of chronic sinusitis and allergies was admitted to our clinic. Multiple endoscopic sinus surgical procedures were performed. Repeated histopathological tissue examinations revealed extranodal NK/T-cell lymphoma, nasal type. Positron emission tomography combined with computed tomography (PET-CT) was performed to assess the extent of the disease. The patient was treated with antibiotics, steroids, and antifungal drugs for many months before the definitive diagnosis was made. The patient was eligible for modified SMILE chemotherapy; however, the patient died suddenly from septic shock several days before the beginning of treatment. Conclusions: Chronic rhinosinusitis with progressive necrosis of sinonasal tissue that persists despite adequate antibacterial and antifungal treatment should prompt further investigation. In such cases, it is important to consider the diagnosis of NK/T-cell lymphomas, nasal type. Early diagnosis increases the opportunity for successful treatment.

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“…Many were described with rapid disease progression and death 5 6. Two cases were associated with dacrocystitis or orbital cellulitis,7 8 but no previous cases have described orbital complications to the extent of the formation of an extraconal subperiosteal phlegmon. NK/T cell lymphoma is known to cause palatal perforation9 and naso-oral fistulae in some cases 10.…”

Section: Discussionmentioning

confidence: 99%

Extranodal NK/T cell lymphoma, nasal type, with retrobulbar extraconal phlegmon and naso-oral fistula

Wong¹,

Parry

Bloch

et al. 2019

BMJ Case Rep

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Extranodal natural killer (NK-)/T cell lymphoma, nasal type (ENKL), 1 is a rare disease that often mimics rheumatological and infectious conditions and can therefore be difficult to diagnose. The authors present a case of a 55-year-old Vietnamese woman who was misdiagnosed with severe atrophic rhinitis and chronic sinus osteitis. Over a period of 8 months from initial referral, she underwent multiple biopsies and was treated with various antimicrobial regimens until the histopathological diagnosis of ENKL was finally made. Her presentation was complicated by bacterial dacrocystitis, preseptal cellulitis and a retrobulbar extraconal phlegmon requiring surgical drainage. She also subsequently developed a naso-oral fistula on treatment. This case highlights the importance of repeated biopsies, in particular from non-necrotic regions of the sinonasal tract when a patient does not respond to therapy and clinical suspicion of neoplastic pathology remains. This is the first case of ENKL to describe significant orbital complication.

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NK/T-Cell Lymphoma of the Nasal Cavity Causing Contralateral Dacryoadenitis

Abstract: Dacryoadenitis can be a clinical manifestation of NK/T-cell lymphoma. To our knowledge, this is the first reported case of nasal NK/T-cell lymphoma with contralateral dacryoadenitis.

Cited by 9 publications

References 5 publications

Case Report: Rhizopus arrhizus Rhino-Orbital-Cerebral Mycosis and Lethal Midline Granuloma: Another Fungal Etiological Agent

Case Report: Rhizopus arrhizus Rhino-Orbital-Cerebral Mycosis and Lethal Midline Granuloma: Another Fungal Etiological Agent

Natural Killer (NK)//T-Cell Lymphoma, Nasal Type, with Periorbital Involvement: A Case Report and Literature Review

Extranodal NK/T cell lymphoma, nasal type, with retrobulbar extraconal phlegmon and naso-oral fistula

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