Letters to Editor
Figure 1: Cervical smear showing HPV changes. Few developing granulomas are also seen (arrows). Inset (left) shows epithelioid giant cell. Inset (right) shows well‑developed HPV changes in another area (Pap, ×400)
could be responsible for the development of superimposed tubercular lesion in cervix. A diminished immunity due to local or systemic effects of tumor may lead to reactivation of dormant tuberculosis.[4] Radiation or chemotherapeutic agents administered to cure or prevent relapse of malignancy may also exacerbate pre‑existing tuberculous lesion.[4] In our opinion, despite the rare incidence, tuberculosis of the cervix should be looked for while evaluating a case for cervical carcinoma and vice versa because of possibility of high morbidity and mortality in patients with co‑existent disease. A simple Pap smear examination can be a stepping‑stone to achieve it. Seth Ankit, Kudesia Madhur, Gupta Kusum, Pant Leela1, Mathur Anjali Departments of Pathology, Kasturba Hospital, Daryaganj, 1Hindurao Hospital, MalkaGanj, Delhi, India
Correspondence to:
Dr. Ankit Seth, E-mail: [email protected]
Respiratory symptoms as first manifestation in an occult alveolar soft part sarcoma
Sir, A 23‑years‑old male presented in chest diseases out‑door patient department with complaints of cough and occasional breathlessness for the last 4 weeks. X‑ray chest showed nodular opacities in lower halves of both lungs [Figure 1]. All his hematological investigations including erythrocyte sedimentation rate and sputum examination were normal. CT scan of chest showed multiple nodules in both lungs with possibility of the diagnosis of metastasis [Figure 2]. After suspicion of the multiple metastases in lungs, CT scan of the abdomen and pelvis was performed to find out the possible primary site and/or the metastasis in the abdominal organs. During this investigation, a soft tissue mass in the left gluteal region was detected. There was an ill‑defined heterogeneous mass involving the left gluteal medius and minimus muscle [Figure 3]. MRI of this region showed the heterogeneous mass of size 6.5 × 5.5 × 5.0 cm with Indian Journal of Cancer | July–September 2014 | Volume 51 | Issue 3
a
b
Figure 2: (a) HPV changes in ectocervical epithelium (H and E, ×100), (b) tuberculous cervicitis (H and E, ×100)
References 1. Samantaray S, Parida G, Rout N, Giri SK, Kar R. Cytologic detection of tuberculous cervicitis: A report of 7 cases. Acta Cytol 2009;53:594‑6. 2. Misch KA, Smithies A, Twomey D, O’Sullivan JC, Onuigbo W. Tuberculosis of the cervix: Cytology as an aid to diagnosis. J Clin Pathol 1976;29:313‑6. 3. Castelo‑Branco C, Mallofre C, Torné A, Gratacós E, Iglesias Guiu X. Primary adenocarcinoma of the endometrium associated with genital tuberculosis. A case report. J Reprod Med 1995;40:673‑5. 4. Karnak D, Kayacan O, Beder S. Reactivation of pulmonary tuberculosis in malignancy. Tumori 2002;88:251‑4. Access this article online Quick Response Code:
Website: www.indianjcancer.com DOI: 10.4103/0019-509X.146773 PMID: *****
poor capsulation and increased vascularity [Figure 4]. Tc‑99 methylenediphosphonate (MDP) bone scan showed no skeletal metastasis. Fine needle aspiration cytology showed dyscohesive cells with an eccentrically located nucleus, nuclear pleomorphism, and variable nuclear chromatin. These neoplastic cells showed a single cell population of epithelioid to plasmacytoid morphology [Figure 5]. The core needle biopsy confirmed the diagnosis of alveolar soft part sarcoma and showed a characteristic morphology with large tumoral cells in nests separated by delicate fibrovascular septae creating pseudo‑alveolar pattern confirming the diagnosis of alveolar soft part sarcoma [Figure 6]. The tumor was widely resected and measured 6 × 5 × 5 cm in dimensions. This was poorly circumscribed. The cut surface was grey‑white to grey‑yellow and friable with areas of hemorrhage and necrosis. There were large tortuous vessels in the surrounding normal tissue [Figure 7]. The patient was treated with chemotherapy using doxorubicin and ifosfamide. However, the patient died within 18 months of treatment due to extensive metastasis and bilateral malignant pleural effusion. 393
Letters to Editor
Figure 1: X‑ray chest showing nodular opacities in both lungs
Figure 3: Computed tomography scan abdomen and pelvis detecting mass in left gluteal region
Figure 2: Computed tomography scan thorax confirming multiple nodular opacities in both lungs
Figure 4: Magnetic resonance imaging T‑2 weighted image showing the tumor in left gluteal muscles
Figure 5: Fine needle aspiration cytology showing round to polygonal cells
ASPS is a rare soft tissue tumor commonly involving the extremities of young adults and less commonly the trunk, head and neck, retroperitoneum, lung, mediastinum, stomach, and the female genital tract. This accounts for less than 1% of all soft tissue sarcomas. [1] This usually presents as a slowly‑growing, painless mass, which can remain undetected due to lack of symptoms. Early metastasis is a characteristic feature of this tumor and, in a good number of cases, metastasis to the lungs or brain is the 394
Figure 6: Histopathology showing pseudo‑alveolar pattern
first presentation of the disease.[1‑4] This young male patient being reported also first developed symptoms related to respiratory system, and the primary lesion remained asymptomatic, which could only be detected during the screening CT scan of abdomen and pelvis. During this imaging, the lesion in the gluteal region was detected and proved to be alveolar soft part sarcoma on histopathology. Indian Journal of Cancer | July–September 2014 | Volume 51 | Issue 3
Letters to Editor Departments of Orthopedic, and 1Pathology, Pt BD Sharma PGIMS, Rohtak, Haryana, India
Correspondence to:
Dr. Zile Singh Kundu, E‑mail: [email protected]
References 1. 2. 3. 4. Figure 7: The widely excised specimen
We conclude that ASPS can remain undetected for a long period and can be easily overlooked due to relative lack of symptoms of the primary tumor. Early metastasis is a characteristic feature of this poor prognostic tumor and unfortunately, in large number of patients, it present with lung or brain metastasis as the first manifestation of the disease. In the cases of occult primary with metastasis to the lungs, ASPS should also be suspected as one of the possibility.
Fletcher CD. Unni KK, Mertens F. Alveolar soft part sarcoma. Pathology and Genetics of Tumors of Soft tissue and Bone. Lyon: IARC Press; 2002. p. 208‑10. Weiss SW, Goldblum Jr. Malignant soft tissue tumors of uncertain type. In: Enzinger and Weiss’s Soft Tissue Tumor. 4th ed. St Louis: CV Mosby; 2001. p. 1483‑571. Montgomery E. Soft tissue tumor. In: Silverberg Principles and Practice of Surgical Pathology and Cytopathology. 4th ed. Philadelphia: Churchill Livingstone Elsevier; 2006. p. 307‑418. Logrono R, Wojtowycz MM, Wunderlich DW, Warner TF, Kurtycz DF. Fine needle aspiration cytology and core biopsy in the diagnosis of alveolar soft part sarcoma presenting with lung metastases. A case report. Acta Cytol 1999;43:464‑70.
Access this article online Quick Response Code:
Website: www.indianjcancer.com DOI: 10.4103/0019-509X.146775 PMID: *****
Kundu ZS, Kalra R1, Rana P1, Mathur SK1, Sangwan SS, Siwach RC
Long term survival after whole brain radiotherapy for brain metastasis in follicular dendritic cell sarcoma
machine. He improved symptomatically and a subsequent CECT scan showed near complete regression of the space occupying lesions as well edema. Patient has been on a regular follow‑up since then and is largely asymptomatic. His most recent CECT scan of the head (May 2013) is within the normal limits [Figure 2]. The patient is therefore clinically and radiologically free of disease, 7 years after the initial diagnosis and 6 years following radiotherapy for brain metastasis.
A 36‑year‑old male patient presented with epigastric pain, anorexia and weight loss of 1 month duration (July 2006). Physical examination revealed epigastric fullness and a right sided inguinal lymph node measuring 6‑cm in greatest dimension. Fine‑needle aspiration from the inguinal lymph node and a biopsy revealed FDCS. Contrast enhanced computed tomographic scan (CECT scan) of the abdomen revealed multiple enlarged retroperitoneal, mesenteric and right inguinal lymph nodes. Further metastatic work‑up was non‑contributory. Patient was thus started on chemotherapy for disseminated dendritic cell sarcoma of the lymph nodes. He received eight cycles of adriamycin and ifosfamide combination chemotherapy. A subsequent CECT scan showed complete resolution of lymph node masses. The patient was asymptomatic for 1‑year when he presented with headache and vomiting. CECT scan of the head was highly suggestive of metastasis in the frontal region [Figure 1]. After a course of steroids, mannitol and neurosurgery consultation for decompression, the patient was taken up for palliative WBRT. He received 30 Gy in 10 daily fractions by parallel opposed portals on a telecobalt
Discussion
Sir, We present a rare case of brain metastasis in a patient with follicular dendritic cell sarcoma (FDCS), who was treated with whole brain radiotherapy (WBRT) and is alive and free from disease, 7 years after diagnosis.
Indian Journal of Cancer | July–September 2014 | Volume 51 | Issue 3
Dendritic cell sarcoma is a rare neoplasm of unknown etiology first reported in 1978. [1] Since then 462 cases have been reported excluding the present one.[2] Systemic involvement and distant metastasis are more commonly seen in liver and lungs. Brain involvement is extremely rare and has been reported in four patients only (including the present one). FDCS is the most common histological subtype (343 reported cases). It behaves like an intermediate grade sarcoma with a substantial risk of local recurrence (28%) and distant metastasis (27%). [2] For localized disease, a radical excision is the preferred treatment and there is no further benefit with adjuvant radiotherapy and chemotherapy. For disseminated/advanced disease (like the present case), chemotherapy is the only option, but the outcome is generally poor.[2] Brain metastases develop in 20‑40% cancer patients during their course of illness. [3] The prognosis for such patients is generally poor with a median survival of less than 3 months without any treatment. The mainstay of treatment has been corticosteroids for alleviation of 395
Copyright of Indian Journal of Cancer is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Figure 1: Cervical smear showing HPV changes. Few developing granulomas are also seen (arrows). Inset (left) shows epithelioid giant cell. Inset (right) shows well‑developed HPV changes in another area (Pap, ×400)
could be responsible for the development of superimposed tubercular lesion in cervix. A diminished immunity due to local or systemic effects of tumor may lead to reactivation of dormant tuberculosis.[4] Radiation or chemotherapeutic agents administered to cure or prevent relapse of malignancy may also exacerbate pre‑existing tuberculous lesion.[4] In our opinion, despite the rare incidence, tuberculosis of the cervix should be looked for while evaluating a case for cervical carcinoma and vice versa because of possibility of high morbidity and mortality in patients with co‑existent disease. A simple Pap smear examination can be a stepping‑stone to achieve it. Seth Ankit, Kudesia Madhur, Gupta Kusum, Pant Leela1, Mathur Anjali Departments of Pathology, Kasturba Hospital, Daryaganj, 1Hindurao Hospital, MalkaGanj, Delhi, India
Correspondence to:
Dr. Ankit Seth, E-mail: [email protected]
Respiratory symptoms as first manifestation in an occult alveolar soft part sarcoma
Sir, A 23‑years‑old male presented in chest diseases out‑door patient department with complaints of cough and occasional breathlessness for the last 4 weeks. X‑ray chest showed nodular opacities in lower halves of both lungs [Figure 1]. All his hematological investigations including erythrocyte sedimentation rate and sputum examination were normal. CT scan of chest showed multiple nodules in both lungs with possibility of the diagnosis of metastasis [Figure 2]. After suspicion of the multiple metastases in lungs, CT scan of the abdomen and pelvis was performed to find out the possible primary site and/or the metastasis in the abdominal organs. During this investigation, a soft tissue mass in the left gluteal region was detected. There was an ill‑defined heterogeneous mass involving the left gluteal medius and minimus muscle [Figure 3]. MRI of this region showed the heterogeneous mass of size 6.5 × 5.5 × 5.0 cm with Indian Journal of Cancer | July–September 2014 | Volume 51 | Issue 3
a
b
Figure 2: (a) HPV changes in ectocervical epithelium (H and E, ×100), (b) tuberculous cervicitis (H and E, ×100)
References 1. Samantaray S, Parida G, Rout N, Giri SK, Kar R. Cytologic detection of tuberculous cervicitis: A report of 7 cases. Acta Cytol 2009;53:594‑6. 2. Misch KA, Smithies A, Twomey D, O’Sullivan JC, Onuigbo W. Tuberculosis of the cervix: Cytology as an aid to diagnosis. J Clin Pathol 1976;29:313‑6. 3. Castelo‑Branco C, Mallofre C, Torné A, Gratacós E, Iglesias Guiu X. Primary adenocarcinoma of the endometrium associated with genital tuberculosis. A case report. J Reprod Med 1995;40:673‑5. 4. Karnak D, Kayacan O, Beder S. Reactivation of pulmonary tuberculosis in malignancy. Tumori 2002;88:251‑4. Access this article online Quick Response Code:
Website: www.indianjcancer.com DOI: 10.4103/0019-509X.146773 PMID: *****
poor capsulation and increased vascularity [Figure 4]. Tc‑99 methylenediphosphonate (MDP) bone scan showed no skeletal metastasis. Fine needle aspiration cytology showed dyscohesive cells with an eccentrically located nucleus, nuclear pleomorphism, and variable nuclear chromatin. These neoplastic cells showed a single cell population of epithelioid to plasmacytoid morphology [Figure 5]. The core needle biopsy confirmed the diagnosis of alveolar soft part sarcoma and showed a characteristic morphology with large tumoral cells in nests separated by delicate fibrovascular septae creating pseudo‑alveolar pattern confirming the diagnosis of alveolar soft part sarcoma [Figure 6]. The tumor was widely resected and measured 6 × 5 × 5 cm in dimensions. This was poorly circumscribed. The cut surface was grey‑white to grey‑yellow and friable with areas of hemorrhage and necrosis. There were large tortuous vessels in the surrounding normal tissue [Figure 7]. The patient was treated with chemotherapy using doxorubicin and ifosfamide. However, the patient died within 18 months of treatment due to extensive metastasis and bilateral malignant pleural effusion. 393
Letters to Editor
Figure 1: X‑ray chest showing nodular opacities in both lungs
Figure 3: Computed tomography scan abdomen and pelvis detecting mass in left gluteal region
Figure 2: Computed tomography scan thorax confirming multiple nodular opacities in both lungs
Figure 4: Magnetic resonance imaging T‑2 weighted image showing the tumor in left gluteal muscles
Figure 5: Fine needle aspiration cytology showing round to polygonal cells
ASPS is a rare soft tissue tumor commonly involving the extremities of young adults and less commonly the trunk, head and neck, retroperitoneum, lung, mediastinum, stomach, and the female genital tract. This accounts for less than 1% of all soft tissue sarcomas. [1] This usually presents as a slowly‑growing, painless mass, which can remain undetected due to lack of symptoms. Early metastasis is a characteristic feature of this tumor and, in a good number of cases, metastasis to the lungs or brain is the 394
Figure 6: Histopathology showing pseudo‑alveolar pattern
first presentation of the disease.[1‑4] This young male patient being reported also first developed symptoms related to respiratory system, and the primary lesion remained asymptomatic, which could only be detected during the screening CT scan of abdomen and pelvis. During this imaging, the lesion in the gluteal region was detected and proved to be alveolar soft part sarcoma on histopathology. Indian Journal of Cancer | July–September 2014 | Volume 51 | Issue 3
Letters to Editor Departments of Orthopedic, and 1Pathology, Pt BD Sharma PGIMS, Rohtak, Haryana, India
Correspondence to:
Dr. Zile Singh Kundu, E‑mail: [email protected]
References 1. 2. 3. 4. Figure 7: The widely excised specimen
We conclude that ASPS can remain undetected for a long period and can be easily overlooked due to relative lack of symptoms of the primary tumor. Early metastasis is a characteristic feature of this poor prognostic tumor and unfortunately, in large number of patients, it present with lung or brain metastasis as the first manifestation of the disease. In the cases of occult primary with metastasis to the lungs, ASPS should also be suspected as one of the possibility.
Fletcher CD. Unni KK, Mertens F. Alveolar soft part sarcoma. Pathology and Genetics of Tumors of Soft tissue and Bone. Lyon: IARC Press; 2002. p. 208‑10. Weiss SW, Goldblum Jr. Malignant soft tissue tumors of uncertain type. In: Enzinger and Weiss’s Soft Tissue Tumor. 4th ed. St Louis: CV Mosby; 2001. p. 1483‑571. Montgomery E. Soft tissue tumor. In: Silverberg Principles and Practice of Surgical Pathology and Cytopathology. 4th ed. Philadelphia: Churchill Livingstone Elsevier; 2006. p. 307‑418. Logrono R, Wojtowycz MM, Wunderlich DW, Warner TF, Kurtycz DF. Fine needle aspiration cytology and core biopsy in the diagnosis of alveolar soft part sarcoma presenting with lung metastases. A case report. Acta Cytol 1999;43:464‑70.
Access this article online Quick Response Code:
Website: www.indianjcancer.com DOI: 10.4103/0019-509X.146775 PMID: *****
Kundu ZS, Kalra R1, Rana P1, Mathur SK1, Sangwan SS, Siwach RC
Long term survival after whole brain radiotherapy for brain metastasis in follicular dendritic cell sarcoma
machine. He improved symptomatically and a subsequent CECT scan showed near complete regression of the space occupying lesions as well edema. Patient has been on a regular follow‑up since then and is largely asymptomatic. His most recent CECT scan of the head (May 2013) is within the normal limits [Figure 2]. The patient is therefore clinically and radiologically free of disease, 7 years after the initial diagnosis and 6 years following radiotherapy for brain metastasis.
A 36‑year‑old male patient presented with epigastric pain, anorexia and weight loss of 1 month duration (July 2006). Physical examination revealed epigastric fullness and a right sided inguinal lymph node measuring 6‑cm in greatest dimension. Fine‑needle aspiration from the inguinal lymph node and a biopsy revealed FDCS. Contrast enhanced computed tomographic scan (CECT scan) of the abdomen revealed multiple enlarged retroperitoneal, mesenteric and right inguinal lymph nodes. Further metastatic work‑up was non‑contributory. Patient was thus started on chemotherapy for disseminated dendritic cell sarcoma of the lymph nodes. He received eight cycles of adriamycin and ifosfamide combination chemotherapy. A subsequent CECT scan showed complete resolution of lymph node masses. The patient was asymptomatic for 1‑year when he presented with headache and vomiting. CECT scan of the head was highly suggestive of metastasis in the frontal region [Figure 1]. After a course of steroids, mannitol and neurosurgery consultation for decompression, the patient was taken up for palliative WBRT. He received 30 Gy in 10 daily fractions by parallel opposed portals on a telecobalt
Discussion
Sir, We present a rare case of brain metastasis in a patient with follicular dendritic cell sarcoma (FDCS), who was treated with whole brain radiotherapy (WBRT) and is alive and free from disease, 7 years after diagnosis.
Indian Journal of Cancer | July–September 2014 | Volume 51 | Issue 3
Dendritic cell sarcoma is a rare neoplasm of unknown etiology first reported in 1978. [1] Since then 462 cases have been reported excluding the present one.[2] Systemic involvement and distant metastasis are more commonly seen in liver and lungs. Brain involvement is extremely rare and has been reported in four patients only (including the present one). FDCS is the most common histological subtype (343 reported cases). It behaves like an intermediate grade sarcoma with a substantial risk of local recurrence (28%) and distant metastasis (27%). [2] For localized disease, a radical excision is the preferred treatment and there is no further benefit with adjuvant radiotherapy and chemotherapy. For disseminated/advanced disease (like the present case), chemotherapy is the only option, but the outcome is generally poor.[2] Brain metastases develop in 20‑40% cancer patients during their course of illness. [3] The prognosis for such patients is generally poor with a median survival of less than 3 months without any treatment. The mainstay of treatment has been corticosteroids for alleviation of 395
Copyright of Indian Journal of Cancer is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
