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Year : 2013  |  Volume : 9  |  Issue : 3  |  Page : 511-513

Sacral pain as the initial symptom in primary Hodgkin's lymphoma of bone

Department of Internal Medicine, Mohammed VI Teaching Hospital, Marrakech, Morocco

Date of Web Publication8-Oct-2013

Correspondence Address:
Fatima Zahra Ha-ou-nou
Department of Internal Medicine, Mohammed VI Teaching Hospital, Marrakech- 40000
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.119365

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 > Abstract 

Progressive painless enlargement of peripheral lymph nodes is the most common presentation of Hodgkin's lymphoma. The bone involvement is seen particularly in the later stages, but very rarely in early stages of the disease. Primary osseous Hodgkin lymphoma is exceedingly rare. Here, we report a case of Primary osseous Hodgkin lymphoma in a 35-year-old male presenting with osteolytic lesions of iliac and sacral bone without other symptoms. Histopathology examination revealed Hodgkin's disease. All other investigations (including computed tomography of the chest, abdomen and pelvis, bone marrow aspiration) were negative for occult disease. Polychemotherapy was started and the symptoms resolved. The case report highlights the importance of clinical suspicion of primary Hodgkin lymphoma of the bone, especially those involving the pelvis bones.

Keywords: Bones, Hodgkin lymphoma, pelvis

How to cite this article:
Ha-ou-nou FZ, Benjilali L, Essaadouni L. Sacral pain as the initial symptom in primary Hodgkin's lymphoma of bone. J Can Res Ther 2013;9:511-3

How to cite this URL:
Ha-ou-nou FZ, Benjilali L, Essaadouni L. Sacral pain as the initial symptom in primary Hodgkin's lymphoma of bone. J Can Res Ther [serial online] 2013 [cited 2022 Nov 30];9:511-3. Available from: https://www.cancerjournal.net/text.asp?2013/9/3/511/119365

 > Introduction Top

Hodgkin's lymphoma (HL) is a lymphoproliferative neoplasm of lymph node comprising multinucleated Reed Sternberg cells within an inflammatory milieu. [1]

It usually presents with lymphadenopathy. However, under some circumstances, those tumors arise from any tissues other than the lymph nodes, simulating other neoplastic or infectious diseases. The common extra nodal sites of involvement are the gastro intestinal tract and pulmonary system. Skeletal lesion at presentation accounts for only 1-4%. [2] Primary osseous Hodgkin lymphoma (POHL) is exceedingly rare. [3] To our knowledge only a few cases have been reported in the literature.

Here we describe an additional case of POHL in 35-year-old man.

 > Case Report Top

A 35 year old male patient presented with sacral pain for 4 months. There was no fever, night sweats or weight loss and no history of bleeding, respiratory, urinary or gastro intestinal symptoms. The physical examination was only revealed pain on palpation of the sacral region. There was no lymphadenopathy, or any mass palpated on the body.

X-ray and computerized tomography scan of the pelvis showed osteolytic lesions in the iliac wings and sacrum [Figure 1].
Figure 1: Computerized tomography scan imaging of the pelvis demonstrate lytic lesions of Ilium and sacrum

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Laboratory values showed an elevated C- reactive protein (48mg/l) and erythrocytes sedimentation rate (117mm in the first hour) with hypercalcaemia (117mg/l). Complete blood count, creatinine, blood urea nitrogen and lactate dehydrogenase were normal.

A neoplastic or infectious osseous disease, such as tuberculosis, was first suggested. Chest X-ray, search of tuberculosis, prostatic ultrasound and prostate-specific antigen were normal. Biopsy performed from the iliac crest revealed reactive cell infiltration and mononuclear Reed-Sternberg cells. In immunohistochemical examination, Reed-Sternberg cells showed positive staining for CD15 and CD30 cells, negative for CD3, CD5, CD10 and CD20 cells. Histopathological and immunohistochemical features of iliac bone were concordant with Hodgkin's lymphoma, nodular sclerosis variant [Figure 2] and [Figure 3]. A computed tomography scan of the chest and the abdomen showed no pathologic findings, particularly non lymphadenopathy could be observed. Cellular bone marrow aspirate and biopsy showed no evidence of lymphoma infiltrate. POHL was diagnosed. The patient underwent poly-chemotherapy with ABVD (doxorubicin, bleomycin, vinblastine, and dacarbazine).
Figure 2: Histology from Bone biopsy, Illustrates mixed inflammatory cells, lacunar cells and Hodgkin cells

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Figure 3: Immunostaining for CD15 shows perinuclear positivity in the large atypical cells

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 > Discussion Top

As well known, most of the HL involves the lymph nodes. Skeletal involvement is a rare event in Hodgkin's disease. [2] It occurs in approximately 10% to 20% of the cases, particularly in advanced stage of disease but very rarely at the time of presentation. [4] POHL is seldom encountered. A review of the literature revealed 54 cases of HL with bone involvement at the time of diagnosis, in which only 16 patients with POHL were identified. [5],[6],[7]

POHL is defined as a lymphoma that is confined to the bone without evidence of concurrent organ or lymph node involvement within six months from initial diagnosis. [8] Regarding published cases, it commonly affects the middle-aged to elderly population, with a median age of 36.3 years. The most common presentation of patients with POHL is bone pain, and less-frequently bone's mass, pathological fracture and neurological deficit due to nerve compression. The various sites of involvement in decreasing order of frequency are long bones (femur, humerus), dorsolumber spine, pelvic bones, sternum, ribs, scapula and clavicle.

Radiologically, HL of bone shows osteolysis, osteosclerosis, or mixed osteosclerosis/osteolysis. Lytic lesions are more common than sclerotic lesions. [5] These aspects are not path-breaking, differential diagnoses included osteomyelitis, non-Hodgkin lymphoma, metastasis, leukemia, and primary bone sarcoma.

Because both clinical and radiological features of POHL aren't specific, the correct diagnosis is only reached after extensive and repeated investigations and review of the histology. Histopathologic diagnosis rests on the identification of the Reed-Sternberg cell or their variants in the appropriate non-neoplastic background; it is straightforward by using tightly focused immunohistochemical examination. Mixed cellularity and nodular sclerosis are commonly observed in the POHL with a preponderance of the first type. [8]

Like other localizations, the therapeutic strategies from POHL vary from surgery, polychemotherapy and radiotherapy. Some cases showed a good prognosis with local radiotherapy alone. [8] In recent years, with combined modality treatments (radio- and chemotherapy) the long term prognosis of patients with osseous HD appears good. [7],[9] The use of surgery is restricted to obtaining open biopsies or treating mechanical or neurological complications. [4]

 > Conclusion Top

Although extremely rare, the primary osseous Hodgkin's lymphoma should be remember in the differential diagnosis of lytic lesions, especially involving the pelvis bones and vertebrae.The practical interest is to make early diagnosis because POHL have more favorable prognosis compared with other malignant bone lesions.

 > Acknowledgement Top

We wish to thank orthopedic surgeon, Dr Boubker Essadki, for performing surgical bone biopsies to obtain lymphoma diagnosis.

 > References Top

1.Piccaluga PP, Agostinelli C, Gazzola A, Tripodo C, Bacci F, Sabattini E, et al. Pathobiology of Hodgkin lymphoma. Adv Hematol 2011:920898.  Back to cited text no. 1
2.Guermazi A, Brice P, de Kerviler EE, Fermé C, Hennequin C, Meignin V, et al. Extranodal Hodgkin disease: Spectrum of disease. Radiographics 2001;21:161-79.  Back to cited text no. 2
3.Bhagavathi S, Fu K. Primary bone lymphoma. Arch Pathol Lab Med 2009;133:1868-71.  Back to cited text no. 3
4.Biswas A, Puri T, Goyal S, Haresh KP, Gupta R, Julka PK, et al. Osseous Hodgkin's lymphoma-review of literature and report of an unusual case presenting as a large ulcerofungating sternal mass. Bone 2008;43:636-40.  Back to cited text no. 4
5.Köseoðlu RD, Senayli A, Biçakçi U, Onuk-Filiz N, Sezer T, Celkan T. Osseous presentation of Hodgkin's disease: A case report and review of the literature.Turk J Pediatr 2007;49:218-22.  Back to cited text no. 5
6.Langley CR, Garrett SJ, Urand J, Kohler J, Clarke NM. Primary multifocal osseous Hodgkin's lymphoma. World J Surg Oncol 2008;6:34.  Back to cited text no. 6
7.Oshikawa G, Arai A, Sasaki K, Ichinohasama R, Miura O. [Primary multifocal osseous Hodgkin lymphoma]. Rinsho Ketsueki 2009;50:92-6.  Back to cited text no. 7
8.Ostrowski ML, Inwards CY, Strickler JG, Witzig TE, Wenger DE, Unni KK. Osseous Hodgkin disease. Cancer 1999;85:1166-78.  Back to cited text no. 8
9.Gebert C, Hardes J, Ahrens H, Buerger H, Winkelmann W, Gosheger G. Primary multifocal osseous Hodgkin disease: A case report and review of the literature. J Cancer Res ClinOncol 2005;131:163-8.  Back to cited text no. 9


  [Figure 1], [Figure 2], [Figure 3]

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