American journal of ophthalmology case reports · 2026
Окулярная аднексальная плазмоцитома, представляющая собой повреждение пличного полулунариса и кранка у 21-летнего мужчины.
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Al-Kaylani Hend M, Torres Guadalupe, Holman Carol J, Binkley Elaine, Greiner Mark A, Goins Kenneth M, Smith Mark C, Syed Nasreen A, Matharu Kanwal S
Аннотация
ЦЕЛЬ: Одиночная экстрамульгированная плазменная цитома (SEMP) составляет 2-5% плазменных клеточных дискразий и определяется как плазменная опухоль клеток в отсутствие системной множественной миеломы или другой дискразии. Редким подтипом SEMP является плазмоцитома, расположенная в окулярной аднексе, чаще всего в конъюнктуре и орбитальных мягких тканях. Мы представляем и обсуждаем случай изолированной массы сосуда и пилика полулунариса у здорового 21-летнего мужчины. ЗАМЕЧАНИЯ: 21-летний мужчина, не имеющий прошлой медицинской или окулярной истории и имеющий расширяющуюся, раздражающую массу в левой медиальной канте. У него была 20/15 зрительная акуратность на двусторонней основе и не было недостатка в экстраокульной мощи. Магнитный резонансный снимок выявил Т2-гиперинтенсию, хорошо обрезанное повреждение без проникновения в синусы или другие ткани. После обрезания гистопатологический анализ подтвердил наличие плазмоцитомы. Пациент прошел системный анализ на множественные миеломы, включая биопсию костного мозга и электрофорез иммунной фексации сыворотки, что было непримечательным. После подтверждения отсутствия каких-либо других мест участия был диагностирован окулярно-аднексальная плазмоцитома, и пациент прошел локальное излучение. ВЫВОДЫ И ВАЖНОСТЬ: Хотя окулярная аднексальная плазмоцитома рассматривалась в предыдущих тематических исследованиях, такая новообразование у здорового молодого человека редко выявляется. Биопсия, как указано, позволяет диагностировать и направлять лечение. Не существует никаких клинических испытаний для лечения окулярной аднексальной плазматомы; в предыдущих докладах было показано, что случаи хорошо реагируют на локальную радиационную терапию или, если их можно полностью исключить, эксцизии лишь при условии тщательного контроля за их повторением. В то время как окулярная аднексальная плазмоцитома сопряжена с меньшим риском продвижения к миеломе, чем плазма-цитома, произведенная костным мозгом, пациенты должны регулярно контролироваться после лечения их офтальмологом, врачом первичной медико-санитарной помощи или онколономом.
Оригинал публикации
Ocular adnexal plasmacytoma presenting as a lesion of the plica semilunaris and caruncle in a 21-year-old male.
PURPOSE: Solitary extramedullary plasmacytoma (SEMP) makes up 2-5% of plasma cell dyscrasias and is defined as a plasma cell neoplasm in the absence of systemic multiple myeloma or other dyscrasia. A rare subset of SEMP is plasmacytoma located in the ocular adnexa, most commonly in the conjunctiva and orbital soft tissues. We present and discuss the case of an isolated mass of the caruncle and plica semilunaris in an otherwise healthy 21-year-old male. OBSERVATIONS: A 21-year-old male with no past medical or ocular history presented with an enlarging, irritating mass in the left medial canthus. He had 20/15 visual acuity bilaterally and no deficits in extraocular motility. Magnetic resonance imaging revealed a T2-hyperintense, well-circumscribed lesion with no infiltration into the sinuses or other tissues. After excision, histopathological analysis confirmed plasmacytoma. The patient underwent a systemic workup for multiple myeloma, including bone marrow biopsy and serum immunofixation electrophoresis, that was unremarkable. After confirmation of no other sites of involvement, a diagnosis of ocular adnexal plasmacytoma was made and the patient underwent localized radiation. CONCLUSIONS AND IMPORTANCE: While ocular adnexal plasmacytoma has been reviewed in previous case studies, it is rare to identify this neoplasm in a healthy young adult. Biopsy is indicated to establish the diagnosis and guide treatment. No clinical trials exist on the treatment of ocular adnexal plasmacytoma; previous reports have demonstrated that cases respond well to localized radiation therapy or, if fully excisable, excision alone with close monitoring for recurrence. While ocular adnexal plasmacytoma has a lower risk for progression to multiple myeloma than bone marrow-derived plasmacytomas, patients should be monitored regularly after treatment by their ophthalmologist, primary care physician, or oncologist.
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Introduction
Plasmacytoma of the ocular adnexa is a rare neoplasm in which plasma cells are identified on histopathology of the conjunctiva or orbital soft tissues in the absence of systemic involvement. 1 Histopathology shows sheets of plasma cells, characterized by eccentric nuclei and abundant cytoplasm. 1 , 2 Because biopsy-confirmed plasmacytoma is associated with multiple myeloma in over 50% of cases, diagnosis requires a thorough systemic evaluation to exclude other sites of involvement, including lymph node assessment, PET CT, skeletal survey, and bone marrow biopsy. 3 , 4 We describe a case of a solitary extramedullary plasmacytoma presenting as a growth of the caruncle and plica semilunaris in an otherwise healthy 21-year-old male.
Case presentation
Patient consent for this report was obtained verbally in clinic. This study adhered to the tenets of the Declaration of Helsinki and the Health Insurance Portability and Accountability Act.
A healthy 21-year-old man of Latinx origin was referred to our clinic for a growing lesion of the bulbar conjunctiva in the left eye with associated tearing, dryness and irritation. He first noticed the lesion seven months prior. He had no ocular history; family history was significant for “blood cancer” in a maternal cousin.
Visual acuity was 20/15 bilaterally, intraocular pressure was within normal limits, and pupils were equal, round, and reactive to light. On exam, a thick, fleshy lesion of the bulbar conjunctiva was present near the plica semilunaris ( Fig. 1 ). The patient chose to defer biopsy at that time. He had concurrent findings of allergic and vernal conjunctivitis, for which he was started on tacrolimus 0.3% ointment once daily, topical prednisolone acetate four times daily, and topical olopatadine twice daily. One month later, there was no change in examination findings; prednisolone was ceased at that visit to avoid confounding of biopsy results. Magnetic resonance imaging (MRI) of the orbit with and without contrast was obtained ( Fig. 2 ). Patient again deferred biopsy at that time.
Slit lamp examination of the left eye at initial presentation demonstrated a fleshy, well-circumscribed lesion of the left caruncle and plica semilunaris (white arrows).
MRI of the orbit without contrast showed a T2 hyperintense 1.2 × 0.7 cm lesion within the medial aspect of the left globe (yellow arrow) with circumscribed margins and no infiltration to adjacent structures.
One month later, the patient agreed to undergo biopsy. The tumor was unable to be completely excised. Histopathology is presented in Fig. 3 . Flow cytometry showed a 21% population of plasma cells with the following immunophenotype: dim CD45 + , bright CD38 + , CD319+, cytoplasmic kappa +, cytoplasmic lambda-, CD117-, CD27 − , CD56 − , CD81 + , dim CD19 + , CD20 − . The immunophenotype was consistent with a kappa-restricted plasma cell neoplasm which is aberrantly CD27 negative.
(A) Photomicrograph illustrating round cells with perinuclear halos (arrows) containing immunoglobulin characteristic of plasma cells (H&E, original magnification = 300x). (B) Immunohistochemical staining for CD20 was negative, (C) CD3 staining was negative, (D) CD138 was positive, indicating plasma cell origin, (E) Kappa light chain was positive throughout most of the lesion, and (F) Lamda light chain was largely negative, in keeping with a kappa restricted proliferation as seen on flow cytometry. (DAB with hematoxylin counterstain; original magnification = 100x for images B-F).
Serum infectious workup was positive for Epstein-Barr virus (EBV) antibodies and negative for human immunodeficiency virus (HIV) antibody testing. Bone marrow analysis, karyotype, fluorescence in situ hybridization, and serum immunofixation electrophoresis were unremarkable; there was no evidence for active multiple myeloma. Lastly, full body positron emission tomography (PET) showed no other sites of involvement.
Three months after biopsy, the lesion demonstrated stability in size and characteristics ( Fig. 4 ). The patient was referred to radiation oncology for localized external beam radiotherapy (40 Gy, 20 fractions) given confirmation of the lesion as a solitary plasmacytoma without active multiple myeloma or other sites of growth.
Slit lamp examination of the left eye three months after biopsy and before radiation (Panels A and B) and at the end of external beam radiation (Panels C and D). The full tumor was unable to be removed through an anterior approach. At 3 months, the lesion extended 8 mm from the medial canthus, with a height of 20 mm. After radiation, treatment sequelae include conjunctival hyperemesis, madarosis, and focal scleral thinning (yellow arrow).
After completion of radiotherapy, the lesion significantly decreased in size. He had 2+ punctate epithelial erosions on slit lamp exam and symptoms of ocular surface dryness related to radiation which were managed with artificial tears and ointment. Visual acuity was 20/20-1 with pinhole. He continues to follow in our clinic for surveillance.
Discussion
Ocular adnexal plasmacytoma is a subset of solitary extramedullary plasmacytoma, defined as a plasma cell neoplasm in the absence of systemic disease or medullary lesions. 4 , 5
Cases of plasmacytoma have been reported in the bulbar 1 , 5 and palpebral conjunctiva 2 , 6 as well as the posterior orbit. 1 One reported case of conjunctival plasmacytoma had corneal involvement at the site of a main incision for cataract surgery seven months prior. 7 In patients with previously diagnosed multiple myeloma, ocular plasmacytoma can signify insufficiently treated or recurrent disease; previous cases include a mass beneath subconjunctival hemorrhage. 8 Other manifestations of multiple myeloma in the eye include corneal crystals, ciliary body/pars plana cysts, and hyperviscosity of the retinal vasculature; thus, posterior segment evaluation is crucial to a thorough physical examination. 5
Our patient presented with ocular adnexal plasmacytoma involving the caruncle and plica semilunaris. Neoplasms in this region have a wide differential; imaging and biopsy are crucial to guide management. Benign neoplasms of the ocular adnexa include nevus (most common), sebaceous gland adenoma, squamous papilloma, pyogenic granuloma, capillary hemangioma, and reactive lymphoid hyperplasia; malignant and pre-malignant lesions include conjunctival keratoacanthoma, oncocytoma, epithelial dysplasia, follicular lymphoma, ocular surface squamous neoplasia, and basal cell carcinoma 9 , 10 , 11 , 12 . For our patient, biopsy was essential to establish the diagnosis and pursue systemic workup for plasma cell dyscrasia.
Given its rarity, risk factors for ocular adnexal plasmacytoma have not been clearly established. In recent work, EBV has been theorized as a causal factor in the development of multiple myeloma 13 but its association with ocular adnexal plasmacytoma has not been investigated to date. Our patient had serum anti-EBV antibodies. Hower, given greater than 90% of the population also has serum anti-EBV antibodies, an association between EBV exposure and plasmacytoma in our patient is very unlikely.
No clinical trials have been conducted to date to establish guidelines for plasmacytomas of the orbit. Previous cases have responded well to external beam radiation. 6 , 14 , 15 Excision can help alleviate space-occupying symptoms or proptosis, and chemotherapy can be considered if the patient has risk factors for transformation to multiple myeloma. 16 Periodic monitoring after radiotherapy is important to ensure the absence of systemic multiple myeloma, especially in the six months after initial biopsy. 2 , 5 , 17 Alternatively, if complete excision is successful, the area can be monitored closely for recurrence without radiation treatment. 5 , 7 Our patient was young and otherwise healthy with no risk factors for systemic disease, but the lesion was unable to be excised completely. So, he was offered 40 Gy of localized radiation therapy. While highly effective at controlling the tumor, there was also radiation-associated exacerbation of ocular surface dryness in our patient. Patients undergoing radiation should be counseled on the risks for ocular surface dryness and radiation retinopathy/optic neuropathy.
Although ocular adnexal plasmacytomas are rare, ophthalmologists should be aware that plasma cell dyscrasias can present in the eye and orbit, even in patients with no risk factors. In patients with multiple myeloma, regular ophthalmologic exams are a method of surveillance for disease progression or recurrence. 8 Strong relationships with oncology and pathology colleagues allow accurate diagnosis, management, and monitoring of patients with plasmacytomas of the ocular adnexa.
CRediT authorship contribution statement
Hend M. Al-Kaylani: Writing – original draft, Visualization. Guadalupe Torres: Writing – review & editing, Investigation. Carol J. Holman: Visualization, Formal analysis. Elaine Binkley: Writing – review & editing, Investigation. Mark A. Greiner: Writing – review & editing. Kenneth M. Goins: Writing – review & editing. Mark C. Smith: Writing – review & editing, Investigation. Nasreen A. Syed: Writing – original draft, Visualization, Investigation. Kanwal S. Matharu: Writing – review & editing, Visualization, Investigation.
Patient consent
We obtained informed verbal consent from the patient to publish this report.
Authorship
All authors attest that they meet the current ICMJE criteria for authorship.
Artificial intelligence statement
Artificial intelligence was not used for any component of the preparation of this manuscript.
Funding
There were no private, public, or non-profit sources of funding for this project.
Declaration of competing interest
The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: Dr. Elaine Binkley reports a relationship with the American Journal of Ophthalmology Case Reports that includes: board membership. Given her role as editor on the board of the American Journal of Ophthalmology Case Reports, Dr. Elaine Binkley had no involvement in the peer review of this article and had no access to information regarding its peer review. Full responsibility for the editorial process for this article was delegated to another journal editor. Author: EB.
Dr. Elaine Binkley received research support from Castle Biosciences and the Gilbert Family Foundation that is not relevant to the content of this manuscript. Author: EB.
Dr. Kanwal Singh Matharu previously served as an editor for the American Journal of Ophthalmology Case Reports. He had no involvement in the peer review of this article and had no access to information regarding its peer review. Full responsibility for the editorial process for this article was delegated to another journal editor. Author: KSM.
Authors Hend M. Al-Kaylani, Guadalupe Torres, Carol J. Holman, Mark A. Greiner, Kenneth M. Goins, Mark C. Smith, and Nasreen A. Syed have no relevant conflicts of interest to disclose. If there are other authors, they declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Acknowledgements
We would like to acknowledge our patient for his commitment to medical education, the care team for this patient, and our clinic photographers.
References
Plasmacytoma of the ocular adnexa is a rare neoplasm in which plasma cells are identified on histopathology of the conjunctiva or orbital soft tissues in the absence of systemic involvement. 1 Histopathology shows sheets of plasma cells, characterized by eccentric nuclei and abundant cytoplasm. 1 , 2 Because biopsy-confirmed plasmacytoma is associated with multiple myeloma in over 50% of cases, diagnosis requires a thorough systemic evaluation to exclude other sites of involvement, including lymph node assessment, PET CT, skeletal survey, and bone marrow biopsy. 3 , 4 We describe a case of a solitary extramedullary plasmacytoma presenting as a growth of the caruncle and plica semilunaris in an otherwise healthy 21-year-old male.
Case presentation
Patient consent for this report was obtained verbally in clinic. This study adhered to the tenets of the Declaration of Helsinki and the Health Insurance Portability and Accountability Act.
A healthy 21-year-old man of Latinx origin was referred to our clinic for a growing lesion of the bulbar conjunctiva in the left eye with associated tearing, dryness and irritation. He first noticed the lesion seven months prior. He had no ocular history; family history was significant for “blood cancer” in a maternal cousin.
Visual acuity was 20/15 bilaterally, intraocular pressure was within normal limits, and pupils were equal, round, and reactive to light. On exam, a thick, fleshy lesion of the bulbar conjunctiva was present near the plica semilunaris ( Fig. 1 ). The patient chose to defer biopsy at that time. He had concurrent findings of allergic and vernal conjunctivitis, for which he was started on tacrolimus 0.3% ointment once daily, topical prednisolone acetate four times daily, and topical olopatadine twice daily. One month later, there was no change in examination findings; prednisolone was ceased at that visit to avoid confounding of biopsy results. Magnetic resonance imaging (MRI) of the orbit with and without contrast was obtained ( Fig. 2 ). Patient again deferred biopsy at that time.
Slit lamp examination of the left eye at initial presentation demonstrated a fleshy, well-circumscribed lesion of the left caruncle and plica semilunaris (white arrows).
MRI of the orbit without contrast showed a T2 hyperintense 1.2 × 0.7 cm lesion within the medial aspect of the left globe (yellow arrow) with circumscribed margins and no infiltration to adjacent structures.
One month later, the patient agreed to undergo biopsy. The tumor was unable to be completely excised. Histopathology is presented in Fig. 3 . Flow cytometry showed a 21% population of plasma cells with the following immunophenotype: dim CD45 + , bright CD38 + , CD319+, cytoplasmic kappa +, cytoplasmic lambda-, CD117-, CD27 − , CD56 − , CD81 + , dim CD19 + , CD20 − . The immunophenotype was consistent with a kappa-restricted plasma cell neoplasm which is aberrantly CD27 negative.
(A) Photomicrograph illustrating round cells with perinuclear halos (arrows) containing immunoglobulin characteristic of plasma cells (H&E, original magnification = 300x). (B) Immunohistochemical staining for CD20 was negative, (C) CD3 staining was negative, (D) CD138 was positive, indicating plasma cell origin, (E) Kappa light chain was positive throughout most of the lesion, and (F) Lamda light chain was largely negative, in keeping with a kappa restricted proliferation as seen on flow cytometry. (DAB with hematoxylin counterstain; original magnification = 100x for images B-F).
Serum infectious workup was positive for Epstein-Barr virus (EBV) antibodies and negative for human immunodeficiency virus (HIV) antibody testing. Bone marrow analysis, karyotype, fluorescence in situ hybridization, and serum immunofixation electrophoresis were unremarkable; there was no evidence for active multiple myeloma. Lastly, full body positron emission tomography (PET) showed no other sites of involvement.
Three months after biopsy, the lesion demonstrated stability in size and characteristics ( Fig. 4 ). The patient was referred to radiation oncology for localized external beam radiotherapy (40 Gy, 20 fractions) given confirmation of the lesion as a solitary plasmacytoma without active multiple myeloma or other sites of growth.
Slit lamp examination of the left eye three months after biopsy and before radiation (Panels A and B) and at the end of external beam radiation (Panels C and D). The full tumor was unable to be removed through an anterior approach. At 3 months, the lesion extended 8 mm from the medial canthus, with a height of 20 mm. After radiation, treatment sequelae include conjunctival hyperemesis, madarosis, and focal scleral thinning (yellow arrow).
After completion of radiotherapy, the lesion significantly decreased in size. He had 2+ punctate epithelial erosions on slit lamp exam and symptoms of ocular surface dryness related to radiation which were managed with artificial tears and ointment. Visual acuity was 20/20-1 with pinhole. He continues to follow in our clinic for surveillance.
Discussion
Ocular adnexal plasmacytoma is a subset of solitary extramedullary plasmacytoma, defined as a plasma cell neoplasm in the absence of systemic disease or medullary lesions. 4 , 5
Cases of plasmacytoma have been reported in the bulbar 1 , 5 and palpebral conjunctiva 2 , 6 as well as the posterior orbit. 1 One reported case of conjunctival plasmacytoma had corneal involvement at the site of a main incision for cataract surgery seven months prior. 7 In patients with previously diagnosed multiple myeloma, ocular plasmacytoma can signify insufficiently treated or recurrent disease; previous cases include a mass beneath subconjunctival hemorrhage. 8 Other manifestations of multiple myeloma in the eye include corneal crystals, ciliary body/pars plana cysts, and hyperviscosity of the retinal vasculature; thus, posterior segment evaluation is crucial to a thorough physical examination. 5
Our patient presented with ocular adnexal plasmacytoma involving the caruncle and plica semilunaris. Neoplasms in this region have a wide differential; imaging and biopsy are crucial to guide management. Benign neoplasms of the ocular adnexa include nevus (most common), sebaceous gland adenoma, squamous papilloma, pyogenic granuloma, capillary hemangioma, and reactive lymphoid hyperplasia; malignant and pre-malignant lesions include conjunctival keratoacanthoma, oncocytoma, epithelial dysplasia, follicular lymphoma, ocular surface squamous neoplasia, and basal cell carcinoma 9 , 10 , 11 , 12 . For our patient, biopsy was essential to establish the diagnosis and pursue systemic workup for plasma cell dyscrasia.
Given its rarity, risk factors for ocular adnexal plasmacytoma have not been clearly established. In recent work, EBV has been theorized as a causal factor in the development of multiple myeloma 13 but its association with ocular adnexal plasmacytoma has not been investigated to date. Our patient had serum anti-EBV antibodies. Hower, given greater than 90% of the population also has serum anti-EBV antibodies, an association between EBV exposure and plasmacytoma in our patient is very unlikely.
No clinical trials have been conducted to date to establish guidelines for plasmacytomas of the orbit. Previous cases have responded well to external beam radiation. 6 , 14 , 15 Excision can help alleviate space-occupying symptoms or proptosis, and chemotherapy can be considered if the patient has risk factors for transformation to multiple myeloma. 16 Periodic monitoring after radiotherapy is important to ensure the absence of systemic multiple myeloma, especially in the six months after initial biopsy. 2 , 5 , 17 Alternatively, if complete excision is successful, the area can be monitored closely for recurrence without radiation treatment. 5 , 7 Our patient was young and otherwise healthy with no risk factors for systemic disease, but the lesion was unable to be excised completely. So, he was offered 40 Gy of localized radiation therapy. While highly effective at controlling the tumor, there was also radiation-associated exacerbation of ocular surface dryness in our patient. Patients undergoing radiation should be counseled on the risks for ocular surface dryness and radiation retinopathy/optic neuropathy.
Although ocular adnexal plasmacytomas are rare, ophthalmologists should be aware that plasma cell dyscrasias can present in the eye and orbit, even in patients with no risk factors. In patients with multiple myeloma, regular ophthalmologic exams are a method of surveillance for disease progression or recurrence. 8 Strong relationships with oncology and pathology colleagues allow accurate diagnosis, management, and monitoring of patients with plasmacytomas of the ocular adnexa.
CRediT authorship contribution statement
Hend M. Al-Kaylani: Writing – original draft, Visualization. Guadalupe Torres: Writing – review & editing, Investigation. Carol J. Holman: Visualization, Formal analysis. Elaine Binkley: Writing – review & editing, Investigation. Mark A. Greiner: Writing – review & editing. Kenneth M. Goins: Writing – review & editing. Mark C. Smith: Writing – review & editing, Investigation. Nasreen A. Syed: Writing – original draft, Visualization, Investigation. Kanwal S. Matharu: Writing – review & editing, Visualization, Investigation.
Patient consent
We obtained informed verbal consent from the patient to publish this report.
Authorship
All authors attest that they meet the current ICMJE criteria for authorship.
Artificial intelligence statement
Artificial intelligence was not used for any component of the preparation of this manuscript.
Funding
There were no private, public, or non-profit sources of funding for this project.
Declaration of competing interest
The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: Dr. Elaine Binkley reports a relationship with the American Journal of Ophthalmology Case Reports that includes: board membership. Given her role as editor on the board of the American Journal of Ophthalmology Case Reports, Dr. Elaine Binkley had no involvement in the peer review of this article and had no access to information regarding its peer review. Full responsibility for the editorial process for this article was delegated to another journal editor. Author: EB.
Dr. Elaine Binkley received research support from Castle Biosciences and the Gilbert Family Foundation that is not relevant to the content of this manuscript. Author: EB.
Dr. Kanwal Singh Matharu previously served as an editor for the American Journal of Ophthalmology Case Reports. He had no involvement in the peer review of this article and had no access to information regarding its peer review. Full responsibility for the editorial process for this article was delegated to another journal editor. Author: KSM.
Authors Hend M. Al-Kaylani, Guadalupe Torres, Carol J. Holman, Mark A. Greiner, Kenneth M. Goins, Mark C. Smith, and Nasreen A. Syed have no relevant conflicts of interest to disclose. If there are other authors, they declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Acknowledgements
We would like to acknowledge our patient for his commitment to medical education, the care team for this patient, and our clinic photographers.
References