Leg lymphedema caused by iliopectineal bursitis associated with destruction of a rheumatoid hip joint: A case report
Identifieur interne : 002197 ( Pmc/Curation ); précédent : 002196; suivant : 002198Leg lymphedema caused by iliopectineal bursitis associated with destruction of a rheumatoid hip joint: A case report
Auteurs : Gen Kuroyanagi ; Kunio Yamada ; Tsukasa Imaizumi ; Jun Mizutani ; Ikuo Wada ; Osamu Kozawa ; Haruhiko Tokuda [Japon] ; Takanobu OtsukaSource :
- Experimental and Therapeutic Medicine [ 1792-0981 ] ; 2013.
Abstract
The present study describes a case of leg lymphedema due to iliopectineal bursitis associated with rheumatoid arthritis (RA), which was satisfactorily controlled by surgery and combination therapy with methotrexate (MTX) and tacrolimus. A 68-year-old male, who had a six-year history of RA, developed an iliopectineal bursa associated with destruction of the hip joint. The mass gradually increased in size, and there was swelling in his right lower extremity. The patient was subsequently hospitalized with increasing right hip pain and leg edema. A colorless transparent lymph fluid leaked from his leg, and leg lymphedema was thus diagnosed. The patient also had a 20-year history of myelodysplastic syndrome. Therefore, the extensive or total resection of the bursa was considered to be too invasive, so a partial bursal excision was performed via an anterior approach. Following the partial bursal excision, total hip arthroplasty (THA) was performed using the Hardinge approach. The leg lymphedema disappeared following the surgery, and the iliopectineal bursa was no longer enlarged. MTX and tacrolimus were postoperatively administered to strictly control the RA. The RA was subsequently well controlled, without any increases in the levels of inflammatory markers, such as C-reactive protein and matrix metalloproteinase-3. This case demonstrated that iliopectineal bursitis was resolved following THA, without complete excision of the intrapelvic bursa, and that strict RA control led to a good clinical course without recurrent inflammation of the bursa. Similar procedures may be beneficial in other patients contraindicated for resection of the entire bursa.
Url:
DOI: 10.3892/etm.2013.1243
PubMed: 24137283
PubMed Central: 3797314
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Gen Kuroyanagi<affiliation><nlm:aff id="af1-etm-06-04-0887">Department of Orthopedic Surgery, Nagoya City University Graduate School of Medical Sciences, Nagoya, Aichi 467-8601;</nlm:aff>
<wicri:noCountry code="subfield">Aichi 467-8601</wicri:noCountry>
</affiliation>
<affiliation><nlm:aff id="af2-etm-06-04-0887">Department of Pharmacology, Gifu University Graduate School of Medicine, Gifu 501-1194;</nlm:aff>
<wicri:noCountry code="subfield">Gifu 501-1194</wicri:noCountry>
</affiliation>
<affiliation><nlm:aff id="af2-etm-06-04-0887">Department of Pharmacology, Gifu University Graduate School of Medicine, Gifu 501-1194;</nlm:aff>
<wicri:noCountry code="subfield">Gifu 501-1194</wicri:noCountry>
</affiliation>
<affiliation><nlm:aff id="af3-etm-06-04-0887">Department of Orthopedic Surgery, Komaki City Hospital, Komaki, Aichi 485-8520;</nlm:aff>
<wicri:noCountry code="subfield">Aichi 485-8520</wicri:noCountry>
</affiliation>
<affiliation><nlm:aff id="af3-etm-06-04-0887">Department of Orthopedic Surgery, Komaki City Hospital, Komaki, Aichi 485-8520;</nlm:aff>
<wicri:noCountry code="subfield">Aichi 485-8520</wicri:noCountry>
</affiliation>
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<affiliation><nlm:aff id="af2-etm-06-04-0887">Department of Pharmacology, Gifu University Graduate School of Medicine, Gifu 501-1194;</nlm:aff>
<wicri:noCountry code="subfield">Gifu 501-1194</wicri:noCountry>
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<front><div type="abstract" xml:lang="en"><p>The present study describes a case of leg lymphedema due to iliopectineal bursitis associated with rheumatoid arthritis (RA), which was satisfactorily controlled by surgery and combination therapy with methotrexate (MTX) and tacrolimus. A 68-year-old male, who had a six-year history of RA, developed an iliopectineal bursa associated with destruction of the hip joint. The mass gradually increased in size, and there was swelling in his right lower extremity. The patient was subsequently hospitalized with increasing right hip pain and leg edema. A colorless transparent lymph fluid leaked from his leg, and leg lymphedema was thus diagnosed. The patient also had a 20-year history of myelodysplastic syndrome. Therefore, the extensive or total resection of the bursa was considered to be too invasive, so a partial bursal excision was performed via an anterior approach. Following the partial bursal excision, total hip arthroplasty (THA) was performed using the Hardinge approach. The leg lymphedema disappeared following the surgery, and the iliopectineal bursa was no longer enlarged. MTX and tacrolimus were postoperatively administered to strictly control the RA. The RA was subsequently well controlled, without any increases in the levels of inflammatory markers, such as C-reactive protein and matrix metalloproteinase-3. This case demonstrated that iliopectineal bursitis was resolved following THA, without complete excision of the intrapelvic bursa, and that strict RA control led to a good clinical course without recurrent inflammation of the bursa. Similar procedures may be beneficial in other patients contraindicated for resection of the entire bursa.</p>
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<article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject>
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<title-group><article-title>Leg lymphedema caused by iliopectineal bursitis associated with destruction of a rheumatoid hip joint: A case report</article-title>
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<contrib-group><contrib contrib-type="author"><name><surname>KUROYANAGI</surname>
<given-names>GEN</given-names>
</name>
<xref ref-type="aff" rid="af1-etm-06-04-0887"><sup>1</sup>
</xref>
<xref ref-type="aff" rid="af2-etm-06-04-0887"><sup>2</sup>
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<xref ref-type="corresp" rid="c1-etm-06-04-0887"></xref>
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<contrib contrib-type="author"><name><surname>YAMADA</surname>
<given-names>KUNIO</given-names>
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<xref ref-type="aff" rid="af3-etm-06-04-0887"><sup>3</sup>
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<contrib contrib-type="author"><name><surname>IMAIZUMI</surname>
<given-names>TSUKASA</given-names>
</name>
<xref ref-type="aff" rid="af3-etm-06-04-0887"><sup>3</sup>
</xref>
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<contrib contrib-type="author"><name><surname>MIZUTANI</surname>
<given-names>JUN</given-names>
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<xref ref-type="aff" rid="af1-etm-06-04-0887"><sup>1</sup>
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<contrib contrib-type="author"><name><surname>WADA</surname>
<given-names>IKUO</given-names>
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<xref ref-type="aff" rid="af1-etm-06-04-0887"><sup>1</sup>
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<contrib contrib-type="author"><name><surname>KOZAWA</surname>
<given-names>OSAMU</given-names>
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<xref ref-type="aff" rid="af2-etm-06-04-0887"><sup>2</sup>
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<contrib contrib-type="author"><name><surname>TOKUDA</surname>
<given-names>HARUHIKO</given-names>
</name>
<xref ref-type="aff" rid="af4-etm-06-04-0887"><sup>4</sup>
</xref>
</contrib>
<contrib contrib-type="author"><name><surname>OTSUKA</surname>
<given-names>TAKANOBU</given-names>
</name>
<xref ref-type="aff" rid="af1-etm-06-04-0887"><sup>1</sup>
</xref>
</contrib>
</contrib-group>
<aff id="af1-etm-06-04-0887"><label>1</label>
Department of Orthopedic Surgery, Nagoya City University Graduate School of Medical Sciences, Nagoya, Aichi 467-8601;</aff>
<aff id="af2-etm-06-04-0887"><label>2</label>
Department of Pharmacology, Gifu University Graduate School of Medicine, Gifu 501-1194;</aff>
<aff id="af3-etm-06-04-0887"><label>3</label>
Department of Orthopedic Surgery, Komaki City Hospital, Komaki, Aichi 485-8520;</aff>
<aff id="af4-etm-06-04-0887"><label>4</label>
Department of Clinical Laboratory, National Center for Geriatrics and Gerontology, Obu, Aichi 474-8511,<country>Japan</country>
</aff>
<author-notes><corresp id="c1-etm-06-04-0887">Correspondence to: Dr Gen Kuroyanagi, Department of Orthopedic Surgery, Nagoya City University Graduate School of Medical Sciences, 1 Kawasumi, Mizuho-cho, Mizuho-ku, Nagoya, Aichi 467-8601, Japan, E-mail: <email>kokuryugen@yahoo.co.jp</email>
</corresp>
</author-notes>
<pub-date pub-type="ppub"><month>10</month>
<year>2013</year>
</pub-date>
<pub-date pub-type="epub"><day>02</day>
<month>8</month>
<year>2013</year>
</pub-date>
<pub-date pub-type="pmc-release"><day>02</day>
<month>8</month>
<year>2013</year>
</pub-date>
<pmc-comment> PMC Release delay is 0 months and 0 days and was based on the
. </pmc-comment>
<volume>6</volume>
<issue>4</issue>
<fpage>887</fpage>
<lpage>890</lpage>
<history><date date-type="received"><day>04</day>
<month>4</month>
<year>2013</year>
</date>
<date date-type="accepted"><day>25</day>
<month>7</month>
<year>2013</year>
</date>
</history>
<permissions><copyright-statement>Copyright © 2013, Spandidos Publications</copyright-statement>
<copyright-year>2013</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/3.0"><license-p>This is an open-access article licensed under a Creative Commons Attribution-NonCommercial 3.0 Unported License. The article may be redistributed, reproduced, and reused for non-commercial purposes, provided the original source is properly cited.</license-p>
</license>
</permissions>
<abstract><p>The present study describes a case of leg lymphedema due to iliopectineal bursitis associated with rheumatoid arthritis (RA), which was satisfactorily controlled by surgery and combination therapy with methotrexate (MTX) and tacrolimus. A 68-year-old male, who had a six-year history of RA, developed an iliopectineal bursa associated with destruction of the hip joint. The mass gradually increased in size, and there was swelling in his right lower extremity. The patient was subsequently hospitalized with increasing right hip pain and leg edema. A colorless transparent lymph fluid leaked from his leg, and leg lymphedema was thus diagnosed. The patient also had a 20-year history of myelodysplastic syndrome. Therefore, the extensive or total resection of the bursa was considered to be too invasive, so a partial bursal excision was performed via an anterior approach. Following the partial bursal excision, total hip arthroplasty (THA) was performed using the Hardinge approach. The leg lymphedema disappeared following the surgery, and the iliopectineal bursa was no longer enlarged. MTX and tacrolimus were postoperatively administered to strictly control the RA. The RA was subsequently well controlled, without any increases in the levels of inflammatory markers, such as C-reactive protein and matrix metalloproteinase-3. This case demonstrated that iliopectineal bursitis was resolved following THA, without complete excision of the intrapelvic bursa, and that strict RA control led to a good clinical course without recurrent inflammation of the bursa. Similar procedures may be beneficial in other patients contraindicated for resection of the entire bursa.</p>
</abstract>
<kwd-group><kwd>iliopectineal bursa</kwd>
<kwd>rheumatoid arthritis</kwd>
<kwd>total hip arthroplasty</kwd>
<kwd>rheumatoid hip joint</kwd>
<kwd>leg lymphedema</kwd>
</kwd-group>
</article-meta>
</front>
<floats-group><fig id="f1-etm-06-04-0887" orientation="portrait" position="float"><label>Figure 1.</label>
<caption><p>(A) Computed tomography (CT) showed an enlarged mass anterior to the right hip joint (white arrow). (B) Magnetic resonance imaging (MRI) showing an enlarged mass anterior to the right hip joint. An axial T2-weighted MRI image showed that there was a connection between the mass and the right hip joint (white arrow). (C) MRI venography demonstrated that the right femoral vein was displaced medially by the mass (white arrow).</p>
</caption>
<graphic xlink:href="ETM-06-04-0887-g00"></graphic>
</fig>
<fig id="f2-etm-06-04-0887" orientation="portrait" position="float"><label>Figure 2.</label>
<caption><p>Histological appearance of the synovial fluid in the contents of the bursa (hematoxylin and eosin staining; magnification, ×100). Bone and cartilage debris and fibrinoid necrosis depositions were apparent (white arrow).</p>
</caption>
<graphic xlink:href="ETM-06-04-0887-g01"></graphic>
</fig>
<table-wrap id="t1-etm-06-04-0887" orientation="portrait" position="float"><label>Table I.</label>
<caption><p>Laboratory data.</p>
</caption>
<table frame="hsides" rules="groups"><thead><tr><th align="left" valign="middle" rowspan="1" colspan="1">Parameters</th>
<th align="center" valign="middle" rowspan="1" colspan="1">At the time of hospitalization</th>
<th align="center" valign="middle" rowspan="1" colspan="1">Eighteen months after surgery</th>
<th align="center" valign="middle" rowspan="1" colspan="1">Normal range</th>
</tr>
</thead>
<tbody><tr><td align="left" valign="top" rowspan="1" colspan="1">Complete blood counts</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> WBC (/<italic>μ</italic>
l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">6800</td>
<td align="center" valign="top" rowspan="1" colspan="1">6200</td>
<td align="center" valign="top" rowspan="1" colspan="1">3500–9000</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Segs (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">85.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">91.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Stabs (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Lymphocytes (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">11.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">7.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Monocytes (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">3.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">2.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Eosinophils (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Basophils (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">1.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Blast (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> RBC (/<italic>μ</italic>
l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">410×10<sup>4</sup>
</td>
<td align="center" valign="top" rowspan="1" colspan="1">394×10<sup>4</sup>
</td>
<td align="center" valign="top" rowspan="1" colspan="1">410–530×10<sup>4</sup>
</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Hb (g/dl)</td>
<td align="center" valign="top" rowspan="1" colspan="1">13.2</td>
<td align="center" valign="top" rowspan="1" colspan="1">13.1</td>
<td align="center" valign="top" rowspan="1" colspan="1">12.4–17.2</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Hct (%)</td>
<td align="center" valign="top" rowspan="1" colspan="1">39.6</td>
<td align="center" valign="top" rowspan="1" colspan="1">39.3</td>
<td align="center" valign="top" rowspan="1" colspan="1">38.0–54.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Plt (/<italic>μ</italic>
l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">23.8×10<sup>4</sup>
</td>
<td align="center" valign="top" rowspan="1" colspan="1">21.3×10<sup>4</sup>
</td>
<td align="center" valign="top" rowspan="1" colspan="1">14.0–35.0×10<sup>4</sup>
</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1">Blood Chemistry</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Total protein (g/dl)</td>
<td align="center" valign="top" rowspan="1" colspan="1">6.4</td>
<td align="center" valign="top" rowspan="1" colspan="1">6.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">6.7–8.3</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Albumin (g/dl)</td>
<td align="center" valign="top" rowspan="1" colspan="1">3.3</td>
<td align="center" valign="top" rowspan="1" colspan="1">3.7</td>
<td align="center" valign="top" rowspan="1" colspan="1">4.0–5.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> AST (IU/l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">34.1</td>
<td align="center" valign="top" rowspan="1" colspan="1">31.5</td>
<td align="center" valign="top" rowspan="1" colspan="1">13.0–33.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> ALT (IU/l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">30.2</td>
<td align="center" valign="top" rowspan="1" colspan="1">29.5</td>
<td align="center" valign="top" rowspan="1" colspan="1">6.0–30.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Urinary nitrogen (mg/dl)</td>
<td align="center" valign="top" rowspan="1" colspan="1">22.1</td>
<td align="center" valign="top" rowspan="1" colspan="1">11.6</td>
<td align="center" valign="top" rowspan="1" colspan="1">8.0–22.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Creatinine (mg/dl)</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.67</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.87</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.60–1.10</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Serum sodium (mEq/l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">139.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">140.6</td>
<td align="center" valign="top" rowspan="1" colspan="1">138.0–146.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Serum potassium (mEq/l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">4.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">3.9</td>
<td align="center" valign="top" rowspan="1" colspan="1">3.6–4.9</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> Serum chloride (mEq/l)</td>
<td align="center" valign="top" rowspan="1" colspan="1">104.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">107.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">99.0–109.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1">Immunology</td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
<td align="center" valign="top" rowspan="1" colspan="1"></td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> CRP (mg/dl)</td>
<td align="center" valign="top" rowspan="1" colspan="1">11.0</td>
<td align="center" valign="top" rowspan="1" colspan="1">1.7</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0–0.3</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> RF (IU/ml)</td>
<td align="center" valign="top" rowspan="1" colspan="1">106.4</td>
<td align="center" valign="top" rowspan="1" colspan="1">22.7</td>
<td align="center" valign="top" rowspan="1" colspan="1">0.0–15.0</td>
</tr>
<tr><td align="left" valign="top" rowspan="1" colspan="1"> MMP-3 (ng/ml)</td>
<td align="center" valign="top" rowspan="1" colspan="1">208.8</td>
<td align="center" valign="top" rowspan="1" colspan="1">234.9</td>
<td align="center" valign="top" rowspan="1" colspan="1">36.9–121.0</td>
</tr>
</tbody>
</table>
<table-wrap-foot><fn id="tfn1-etm-06-04-0887"><p>WBC, white blood cell; RBC, red blood cell; Hb, hemoglobin; Hct, hematocrit; Plt, platelet; AST, L-aspartate aminotransferase; ALT, L-alanine aminotransferase; CRP, C-reactive protein; RF, rheumatoid factor; MMP-3, matrix metalloproteinase-3.</p>
</fn>
</table-wrap-foot>
</table-wrap>
</floats-group>
</pmc>
</record>
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