Spontaneous regression of an anaplastic large cell lymphoma in the oral cavity: first reported case and review of the literature

doi:10.1016/S1368-8375(99)00034-2

Oral Oncology

Volume 35, Issue 6, November 1999, Pages 609-613

https://doi.org/10.1016/S1368-8375(99)00034-2 Get rights and content

Abstract

Lymphomas account for 2–5% of all oral malignancies and are the third most common in this site. This case report appears to be the first in the world literature describing spontaneous regression in the oral cavity of a subset of non-Hodgkins lymphomas known as Ki-1 anaplastic large cell lymphomas (ALCL). Ki-1 ALCL account for 2–7% of all non-Hodgkins lymphomas and the clinical presentation is variable; they may arise de novo or in the setting of a separate primary lymphoma and commonly present in the extra-nodal location. Disease severity is also variable with waxing and waning lesions at one extreme which may spontaneously regress to bone marrow involvement in around 12% of cases. This case is especially interesting since the patient is a farmer, given the recent evidence that there may be a link between non-Hodgkins lymphoma and this occupation.

Section snippets

Case report

A 77-year-old gentleman was referred to the Department of Oral Medicine at Glasgow Dental Hospital and School by his general dental practitioner for investigation of a swelling affecting the left side of his soft palate. The patient reported that the palatal swelling had been present for some 3 weeks and he related this to previous dental treatment. He had attended 3 weeks previously to his general dental practitioner for the placement of a small restoration in his upper left canine tooth. This

Discussion

This interesting case would appear to be the first in the world literature reporting the spontaneous regression of an ALCL in the oral cavity. Disease relapse with rapidly progressive cervical lymphadenopathy occurred after a disease-free interval of 12 months from primary remission but without any recurrence at the primary site in the oral cavity.

Malignant oral lymphomas are rare, accounting for 2–5% of all oral malignancies [1], [2], although they are the third most common malignancy in the

Clinical

Since Stein et al. [12] reported diffuse large cell non-Hodgkin's type lymphoma positive for Ki-1 antigen in 1985 there have been numerous reports of similar cases. It is now regarded as a separate clinical entity in the updated Kiel classification and the REAL classification of non-Hodgkin's lymphomas. Anaplastic large cell lymphoma accounts for 2–7% of all non-Hodgkin's lymphomas [13], [14]. Most cases arise de novo (primary type); others arise in the setting of other lymphomas (secondary)

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Cited by (39)

Self-regressing oral CD30-positive, EBV-negative, T-cell lymphoproliferative lesions. A poorly understood process highlighted by ominous clinicopathologic features and indolent behavior
2021, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Intraoral, primary, CD30-positive (CD30⁺) T-cell lymphoproliferative disorders (TLPDs) are uncommon, and their clinicopathologic presentation and management can vary and may be challenging. Herein, we present a retrospective study of 4 examples of self-regressing primary CD30⁺ TLPD affecting the gingiva.
Archived files were retrospectively reviewed for oral CD30⁺ TLPDs featuring (1) proper immunohistochemical documentation, (2) Epstein-Barr virus negativity, (3) adequate follow-up information corroborating regression, and (4) no history of hematopoietic malignancy or related-mucocutaneous disease.
Three women and 1 man (age range, 55-82 years; mean, 68.3 years) presented with rapidly growing gingival ulcers. Microscopic evaluation revealed diffuse infiltration by sheets of large, atypical cells admixed with lymphocytes and eosinophils, showing angiocentric distribution, focal neurotropism, and muscle infiltration. The lesional cells consistently stained for CD3 and CD30 and were variably immunoreactive against CD2, CD4, CD5, CD7, and CD8, but were negative for ALK1 and EBV-encoded small RNA. TCR-γ gene rearrangement studies revealed a monoclonal T-cell population in 1 case. All lesions showed complete regression 2 to 8 weeks postoperatively (mean follow-up, 4.5 weeks).
Notwithstanding their alarming clinicopathologic appearance, there are CD30⁺ TLPDs confined to the oral cavity that have an indolent course. However, clinical staging is essential to exclude aggressive systemic malignancy.
Necrotic mucosal CD30-positive ulcer on the oral mucosa: a self-healing lymphoma
2017, British Journal of Oral and Maxillofacial Surgery
An elderly man with a gingival mass that spontaneously regressed
2016, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Citation Excerpt :
Regression often appears to follow some nonspecific mechanical event that is accompanied by a reduction of tumor load, such as biopsy or incomplete excision, as was the case with our patient. Additional events that have been associated with regression are local infection, autoimmune illness, irradiation of metastases other than those that regress, transfusion reaction, hemodialysis, and pregnancy termination.15,21-30 Patients with gingival enlargements are seen very commonly in daily clinical practice, but treatment planning is essential to differentiate neoplastic lesions from nonneoplastic lesions.
A case of spontaneous regression of lymphoma in the mandibular gingiva after biopsy
2015, Oral and Maxillofacial Surgery Cases
We report a case of lymphoma that developed on the mandibular gingiva and spontaneously regressed after biopsy was performed. The patient was an 87-year-old man who visited our hospital with a complaint of gingival swelling in the left mandibular molar region. We observed an elastic, soft, well-demarcated, granuloma-like mass measuring 30 × 25 mm in size and comprising partially white pseudomembranous areas. We conducted a biopsy, which confirmed a pathologic diagnosis of a diffuse large B-cell lymphoma. Because of age constraints, the patient was monitored without being administered aggressive therapy, and the mass disappeared 20 days after biopsy. We then performed a follow-up biopsy, which revealed the absence of tumor cells. There was no recurrence of lymphoma during the 2.5-year follow-up period.
Spontaneous remission of a squamous cell carcinoma of the floor of the mouth
2014, Journal of Cranio-Maxillofacial Surgery
Citation Excerpt :
In addition, there are several reports in the literature describing the regression of a variety of cancers, including HPV-induced cervical intraepithelial neoplasia (Kadish et al., 2002), cholangiocarcinoma (Yoshimitsu et al., 1996), hepatocellular carcinoma (Misawa et al., 1999; Harada et al., 2010) metastatic renal cell carcinoma (Crisci et al., 2008), oesophageal carcinoma (Kubota et al., 2003) and Merkel cell carcinoma (Brown et al., 1999; Torroni et al., 2007; Vesely et al., 2008; Karkos et al., 2010). In the oral cavity, spontaneous regression of lymphomas (Savarrio et al., 1999; Koga et al., 2003; Heibel et al., 2004), as well as oropharyngeal (Kurita et al., 2007) and recurrent tongue (Oya and Ikemura, 2004) carcinomas has been reported. Here, we present a novel case of a patient with a primary squamous cell carcinoma on the floor of the mouth whose tumour regressed spontaneously in three months, without any treatment.
Spontaneous remission is a rare, but well recognized event in oncology. Certain tumours, such as melanomas, hypernephromas and neuroblastomas, are known for showing spontaneous regression. Similarly, spontaneous regression of oral lymphomas, as well as oropharyngeal and recurrent tongue carcinomas, has been reported. Here, we present a novel case of a patient with a primary squamous cell carcinoma on the floor of the mouth whose tumour regressed spontaneously in three months, without any treatment. We also review of the literature on the spontaneous remission of oral cancer and discuss possible mechanisms for this phenomenon.
The spectrum of primary mucosal CD30-positive T-cell lymphoproliferative disorders of the head and neck
2014, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Citation Excerpt :
Similar to primary cutaneous ALCL (C-ALCL), ALCL limited to mucosal sites of the head and neck is characterized by large atypical neoplastic cells with diverse morphology, CD30 expression, and ALK negativity.4,7 Moreover, some studies have described that ALCL limited to mucosal sites may show indolent behavior,4,6,10 a prominent feature of C-ALCL. This representation has led to speculation by some authors that ALCLs primarily arising in the mucosa of the head and neck (M-ALCLs) may be “counterparts” of C-ALCL and that primary cutaneous CD30+ T-cell lymphoproliferative disorders may extend to mucosal sites of the head and neck.4
To retrospectively investigate the clinicopathologic spectrum of primary mucosal CD30-positive T-cell lymphoproliferative disorders (PTCLDs) of the head and neck.
Archives of PTCLDs primarily arising in head and neck mucosa were reviewed. Immunostaining of CD20, CD3, CD4, CD8, CD30, CD56, anaplastic lymphoma kinase (ALK), epithelial membrane antigen (EMA), cytotoxic molecules (TIA-1, granzyme B, or perforin), and Ki67; in situ hybridization for Epstein-Barr virus; and T-cell receptor gene rearrangement analysis were performed.
Fourteen cases of primary mucosal anaplastic large cell lymphoma (M-ALCL) were identified, and no lymphomatoid papulosis (LyP) cases were found. All cases demonstrated atypical mononuclear neoplastic cells with diverse histology and cytomorphology. The typical immunophenotype of neoplastic cells was CD3-positive, CD4-positive, CD8-negative, CD30-positive, ALK-negative, and cytotoxic molecules–positive. Infiltration of inflammatory cells was common. All cases presented an indolent course, regardless of therapy.
PTCLDs of the head and neck provisionally included M-ALCL alone.

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Case reportSpontaneous regression of an anaplastic large cell lymphoma in the oral cavity: first reported case and review of the literature

Abstract

Section snippets

Case report

Discussion

Clinical

Eur J Cancer Clin Oncol

Oral Surg Oral Med Oral Pathol

Oral Surg Oral Med Oral Pathol

Pathol Res Pract

Oral Surg Oral Med Oral Pathol

Lancet

Blood

Blood

Ann Oncol

Blood

Int J Oral Maxillofac Surg

Oral Oncol

Occurrence and prognosis of extranodal lymphomas

Cancer

Malignant lymphoma of the oral cavity: clinicopathological analysis of 20 cases

J Oral Pathol

Spontaneous regression of diffuse large cell lymphoma associated with Hashimoto's thyroiditis (letter)

Am J Haematol

Meta-analyses of non-Hodgkin's lymphoma and farming

Scan J Work, Envir and Health

Post-transplant CD 30 (Ki-1) positive anaplastic large cell lymphoma. Report of a case with presentation as apleural effusion

Acta Cytol

A case of Ki-1 positive anaplastic large cell lymphoma transformed from mycosis fungoides

J Dermatology

Case report
Spontaneous regression of an anaplastic large cell lymphoma in the oral cavity: first reported case and review of the literature