Successful treatment of bacillary angiomatosis with oral doxycycline in an HIV-infected child with skin lesions mimicking Kaposi sarcoma
Identifieur interne : 002327 ( Pmc/Corpus ); précédent : 002326; suivant : 002328Successful treatment of bacillary angiomatosis with oral doxycycline in an HIV-infected child with skin lesions mimicking Kaposi sarcoma
Auteurs : Olaf Rodriguez ; Liane R. Campbell ; Jason M. Bacha ; Carrie L. KovarikSource :
- JAAD Case Reports [ 2352-5126 ] ; 2016.
Url:
DOI: 10.1016/j.jdcr.2015.12.002
PubMed: 27051835
PubMed Central: 4809473
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Successful treatment of bacillary angiomatosis with oral doxycycline in an HIV-infected child with skin lesions mimicking Kaposi sarcoma</title><author><name sortKey="Rodriguez, Olaf" sort="Rodriguez, Olaf" uniqKey="Rodriguez O" first="Olaf" last="Rodriguez">Olaf Rodriguez</name><affiliation><nlm:aff id="aff1">Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania</nlm:aff></affiliation></author><author><name sortKey="Campbell, Liane R" sort="Campbell, Liane R" uniqKey="Campbell L" first="Liane R." last="Campbell">Liane R. Campbell</name><affiliation><nlm:aff id="aff2">Baylor College of Medicine Children's Foundation–Tanzania, Pediatrics, Mbeya, Tanzania</nlm:aff></affiliation></author><author><name sortKey="Bacha, Jason M" sort="Bacha, Jason M" uniqKey="Bacha J" first="Jason M." last="Bacha">Jason M. Bacha</name><affiliation><nlm:aff id="aff2">Baylor College of Medicine Children's Foundation–Tanzania, Pediatrics, Mbeya, Tanzania</nlm:aff></affiliation></author><author><name sortKey="Kovarik, Carrie L" sort="Kovarik, Carrie L" uniqKey="Kovarik C" first="Carrie L." last="Kovarik">Carrie L. Kovarik</name><affiliation><nlm:aff id="aff3">Department of Dermatology, University of Pennsylvania, Philadelphia, Pennsylvania</nlm:aff></affiliation><affiliation><nlm:aff id="aff4">Department of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">27051835</idno><idno type="pmc">4809473</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4809473</idno><idno type="RBID">PMC:4809473</idno><idno type="doi">10.1016/j.jdcr.2015.12.002</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">002327</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002327</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Successful treatment of bacillary angiomatosis with oral doxycycline in an HIV-infected child with skin lesions mimicking Kaposi sarcoma</title><author><name sortKey="Rodriguez, Olaf" sort="Rodriguez, Olaf" uniqKey="Rodriguez O" first="Olaf" last="Rodriguez">Olaf Rodriguez</name><affiliation><nlm:aff id="aff1">Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania</nlm:aff></affiliation></author><author><name sortKey="Campbell, Liane R" sort="Campbell, Liane R" uniqKey="Campbell L" first="Liane R." last="Campbell">Liane R. Campbell</name><affiliation><nlm:aff id="aff2">Baylor College of Medicine Children's Foundation–Tanzania, Pediatrics, Mbeya, Tanzania</nlm:aff></affiliation></author><author><name sortKey="Bacha, Jason M" sort="Bacha, Jason M" uniqKey="Bacha J" first="Jason M." last="Bacha">Jason M. Bacha</name><affiliation><nlm:aff id="aff2">Baylor College of Medicine Children's Foundation–Tanzania, Pediatrics, Mbeya, Tanzania</nlm:aff></affiliation></author><author><name sortKey="Kovarik, Carrie L" sort="Kovarik, Carrie L" uniqKey="Kovarik C" first="Carrie L." last="Kovarik">Carrie L. Kovarik</name><affiliation><nlm:aff id="aff3">Department of Dermatology, University of Pennsylvania, Philadelphia, Pennsylvania</nlm:aff></affiliation><affiliation><nlm:aff id="aff4">Department of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania</nlm:aff></affiliation></author></analytic><series><title level="j">JAAD Case Reports</title><idno type="eISSN">2352-5126</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Mohle Boetani, J C" uniqKey="Mohle Boetani J">J.C. Mohle-Boetani</name></author><author><name sortKey="Koehler, J E" uniqKey="Koehler J">J.E. Koehler</name></author><author><name sortKey="Berger, T G" uniqKey="Berger T">T.G. Berger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Batsakis, J G" uniqKey="Batsakis J">J.G. Batsakis</name></author><author><name sortKey="Ro, J Y" uniqKey="Ro J">J.Y. Ro</name></author><author><name sortKey="Frauenhoffer, E E" uniqKey="Frauenhoffer E">E.E. Frauenhoffer</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Koehler, J E" uniqKey="Koehler J">J.E. Koehler</name></author><author><name sortKey="Leboit, P E" uniqKey="Leboit P">P.E. LeBoit</name></author><author><name sortKey="Egbert, B M" uniqKey="Egbert B">B.M. Egbert</name></author><author><name sortKey="Berger, T G" uniqKey="Berger T">T.G. Berger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Schwartz, R A" uniqKey="Schwartz R">R.A. Schwartz</name></author><author><name sortKey="Nychay, S G" uniqKey="Nychay S">S.G. Nychay</name></author><author><name sortKey="Janniger, C K" uniqKey="Janniger C">C.K. Janniger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mayama, S" uniqKey="Mayama S">S. Mayama</name></author><author><name sortKey="Cuevas, L E" uniqKey="Cuevas L">L.E. Cuevas</name></author><author><name sortKey="Sheldon, J" uniqKey="Sheldon J">J. Sheldon</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dedicoat, M" uniqKey="Dedicoat M">M. Dedicoat</name></author><author><name sortKey="Newton, R" uniqKey="Newton R">R. Newton</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mutalima, N" uniqKey="Mutalima N">N. Mutalima</name></author><author><name sortKey="Molyneux, E" uniqKey="Molyneux E">E. Molyneux</name></author><author><name sortKey="Johnston, W" uniqKey="Johnston W">W. Johnston</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Wanat, K A" uniqKey="Wanat K">K.A. Wanat</name></author><author><name sortKey="Reid, E" uniqKey="Reid E">E. Reid</name></author><author><name sortKey="Kamiyango, W" uniqKey="Kamiyango W">W. Kamiyango</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Guerra, L G" uniqKey="Guerra L">L.G. Guerra</name></author><author><name sortKey="Neira, C J" uniqKey="Neira C">C.J. Neira</name></author><author><name sortKey="Boman, D" uniqKey="Boman D">D. Boman</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">JAAD Case Rep</journal-id><journal-id journal-id-type="iso-abbrev">JAAD Case Rep</journal-id><journal-title-group><journal-title>JAAD Case Reports</journal-title></journal-title-group><issn pub-type="epub">2352-5126</issn><publisher><publisher-name>Elsevier</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">27051835</article-id><article-id pub-id-type="pmc">4809473</article-id><article-id pub-id-type="publisher-id">S2352-5126(15)00202-7</article-id><article-id pub-id-type="doi">10.1016/j.jdcr.2015.12.002</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Successful treatment of bacillary angiomatosis with oral doxycycline in an HIV-infected child with skin lesions mimicking Kaposi sarcoma</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Rodriguez</surname><given-names>Olaf</given-names></name><degrees>BS</degrees><xref rid="aff1" ref-type="aff">a</xref></contrib><contrib contrib-type="author"><name><surname>Campbell</surname><given-names>Liane R.</given-names></name><degrees>MD</degrees><xref rid="aff2" ref-type="aff">b</xref></contrib><contrib contrib-type="author"><name><surname>Bacha</surname><given-names>Jason M.</given-names></name><degrees>MD</degrees><xref rid="aff2" ref-type="aff">b</xref></contrib><contrib contrib-type="author"><name><surname>Kovarik</surname><given-names>Carrie L.</given-names></name><degrees>MD</degrees><email>carrie.kovarik@uphs.upenn.edu</email><xref rid="aff3" ref-type="aff">c</xref><xref rid="aff4" ref-type="aff">d</xref><xref rid="cor1" ref-type="corresp">∗</xref></contrib></contrib-group><aff id="aff1"><label>a</label>Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania</aff><aff id="aff2"><label>b</label>Baylor College of Medicine Children's Foundation–Tanzania, Pediatrics, Mbeya, Tanzania</aff><aff id="aff3"><label>c</label>Department of Dermatology, University of Pennsylvania, Philadelphia, Pennsylvania</aff><aff id="aff4"><label>d</label>Department of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania</aff><author-notes><corresp id="cor1"><label>∗</label>Correspondence to: Carrie L. Kovarik, MD, Department of Dermatology, University of Pennsylvania, 3600 Spruce Street, 2 Maloney Building, Philadelphia, Pennsylvania 19104.Department of DermatologyUniversity of Pennsylvania3600 Spruce Street, 2 Maloney BuildingPhiladelphiaPennsylvania19104 <email>carrie.kovarik@uphs.upenn.edu</email></corresp></author-notes><pub-date pub-type="pmc-release"><day>04</day><month>2</month><year>2016</year></pub-date><pmc-comment> PMC Release delay is 0 months and 0 days and was based on .</pmc-comment>
<pub-date pub-type="collection"><month>1</month><year>2016</year></pub-date><pub-date pub-type="epub"><day>04</day><month>2</month><year>2016</year></pub-date><volume>2</volume><issue>1</issue><fpage>77</fpage><lpage>79</lpage><permissions><copyright-statement>© 2015 by the American Academy of Dermatology, Inc. Published by Elsevier, Inc.</copyright-statement><copyright-year>2015</copyright-year><copyright-holder>American Academy of Dermatology, Inc.</copyright-holder><license license-type="CC BY-NC-ND" xlink:href="http://creativecommons.org/licenses/by-nc-nd/4.0/"><license-p>This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).</license-p></license></permissions><kwd-group><title>Key words</title><kwd>bacillary angiomatosis</kwd><kwd>bacteria</kwd><kwd>global health</kwd><kwd>HIV</kwd><kwd>infection</kwd><kwd>pediatrics</kwd></kwd-group><kwd-group><title>Abbreviations used</title><kwd>ART, antiretroviral therapy</kwd><kwd>BA, bacillary angiomatosis</kwd><kwd>HHV-8, human herpes virus 8</kwd><kwd>KS, Kaposi sarcoma</kwd></kwd-group></article-meta></front><body><sec id="sec1"><title>Case report</title><p>A 12-year-old boy presented for treatment of widespread papules and fungating, ulcerating nodules (<xref rid="fig1" ref-type="fig">Fig 1</xref>). Seven months before referral, HIV was diagnosed, and he started antiretroviral therapy (ART), with a baseline CD4 count of 76 cells per microliter (5%). Per history, the rash began 1 month after ART initiation and progressively worsened in the 6 months after ART therapy started. The patient was referred to the Baylor Tanzania Center of Excellence in Mbeya, Tanzania because of concern of possible Kaposi sarcoma (KS).</p><p>A skin biopsy was performed. The patient underwent chest radiography, abdominal ultrasound scan, and liver/renal function tests, the results of which were normal. A complete blood count was notable for severe anemia (hemoglobin level of 4.0 g/dL) requiring blood transfusion. The clinical diagnosis of bacillary angiomatosis (BA) was favored, and the patient was prescribed doxycycline, 100 mg twice daily. Although KS was also in the clinical differential diagnosis, chemotherapy was withheld pending biopsy results. Histologically, the lesion biopsy result was consistent with BA, and bacilli were observed with Warthin-Starry staining.</p><p>At the 1-week follow-up appointment, a dramatic improvement in the appearance of the lesions was noted (<xref rid="fig2" ref-type="fig">Fig 2</xref>). The patient continued doxycycline with continued improvement over 7 additional weeks, with near complete resolution of the sores and lesions (<xref rid="fig3" ref-type="fig">Fig 3</xref>).</p></sec><sec id="sec2"><title>Discussion</title><p>BA is a rare multisystem bacterial infectious vasculoproliferative disorder caused by <italic>Bartonella henselae</italic> and <italic>Bartonella quintana</italic> and is most commonly seen in HIV-infected individuals with CD4 counts less than 200 cells per microliter.<xref rid="bib1" ref-type="bibr"><sup>1</sup></xref> BA commonly presents in the skin but may affect almost any visceral organ, including liver, spleen, or nasal cavity.<xref rid="bib2" ref-type="bibr"><sup>2</sup></xref> Although there is no consensus treatment for BA, clinicians experienced in treating this condition recommend therapy with oral erythromycin or doxycycline for 8 to 12 weeks to avoid relapses.<xref rid="bib3" ref-type="bibr"><sup>3</sup></xref></p><p>KS is a rare angioproliferative opportunistic infection caused by human herpes virus 8 (HHV-8) also seen in HIV-infected patients with low CD4 counts. The overlapping constellation of cutaneous manifestations of BA and KS make them clinically indistinguishable from each other when presenting as single or multiple violaceous papules, nodules, plaques or tumors.<xref rid="bib4" ref-type="bibr"><sup>4</sup></xref> Occasionally, clinical clues can lead toward one diagnosis or the other. KS may cause prominent lymphedema of the legs and oral lesions. BA may have a very rapid onset with swift growth of lesions, which may occur with immune reconstitution inflammatory syndrome, further complicating the difficulty in distinguishing these entities without pathology. However, KS can also develop rapidly in severely immunocompromised patients and during immune reconstitution, and BA can have oral lesions that mimic KS. In addition, other HIV-associated dermatologic conditions, such as disseminated mycobacterial or fungal infections can have similar presentations to those of KS or BA.</p><p>A study on the seroprevalence of HHV-8 in a general population of Ugandan children suggests that infection with the virus often occurs during early childhood and reaches a level of approximately 50% before puberty.<xref rid="bib5" ref-type="bibr"><sup>5</sup></xref> Tanzania, like other East African countries, has a high HHV-8 seropositivity rate, with one study estimating the prevalence of evidence of HHV-8 infection to be 66% among adults.<xref rid="bib6" ref-type="bibr"><sup>6</sup></xref> In Malawian children, HIV infection has been significantly associated with KS with an estimated odds ratio of 93.5, emerging as one of the most common pediatric cancers in sub-Saharan Africa in the context of the HIV epidemic.<xref rid="bib7" ref-type="bibr"><sup>7</sup></xref></p><p>Given the frequency of KS in this clinical scenario and the aggressive nature of KS, particularly in children, health care providers in many regions of Africa are often correct when they render a clinical diagnosis of KS and begin chemotherapy, whereas other potential diagnoses such as BA may not be considered. The differentiation of BA and KS histologically may still be difficult in early lesions, particularly if HHV-8 immunostaining or Warthin-Starry staining are not available. BA and KS can also present simultaneously, further complicating the clinical diagnosis, as antibiotic therapy may resolve the BA but not treat the KS and associated systemic symptoms. Conversely, BA lesions in a patient being empirically treated with chemotherapy for KS may initially respond because of the vascular nature of BA lesions, but the lesions would likely not completely resolve, creating a clinical quandary.<xref rid="bib8" ref-type="bibr"><sup>8</sup></xref></p><p>A skin biopsy reviewed by a dermatopathologist with special stains is necessary to definitively differentiate between BA and KS; however, this is not often available in the settings in which these patients present. In those cases in which a patient cannot be definitively diagnosed, and the clinical differential diagnosis includes both BA and KS, we propose that initial empiric treatment with erythromycin or doxycycline may be a viable option. Although a study assessing the efficacy of empiric erythromycin or doxycycline therapy in cases such as this has not yet been performed, both antibiotics are often readily available, even in resource constrained settings. If the patient does have BA, this therapy typically leads to complete resolution of the BA with minimal negative side effects and may result in a response in as little as 7 days, as in the case of our patient. The rapid clearance of BA with doxycycline observed by our patient is not unusual and has been previously reported.<xref rid="bib9" ref-type="bibr"><sup>9</sup></xref> Ultimately, if the patient does not respond to empiric erythromycin or doxycycline, the clinician may confidently proceed with treatment for KS or other conditions.</p></sec></body><back><ref-list><title>References</title><ref id="bib1"><label>1</label><element-citation publication-type="journal" id="sref1"><person-group person-group-type="author"><name><surname>Mohle-Boetani</surname><given-names>J.C.</given-names></name><name><surname>Koehler</surname><given-names>J.E.</given-names></name><name><surname>Berger</surname><given-names>T.G.</given-names></name></person-group><article-title>Bacillary angiomatosis and bacillary peliosis in patients infected with human immunodeficiency virus: clinical characteristics in a case-control study</article-title><source>Clin Infect Dis</source><volume>22</volume><issue>5</issue><year>1996</year><fpage>794</fpage><lpage>800</lpage><pub-id pub-id-type="pmid">8722933</pub-id></element-citation></ref><ref id="bib2"><label>2</label><element-citation publication-type="journal" id="sref2"><person-group person-group-type="author"><name><surname>Batsakis</surname><given-names>J.G.</given-names></name><name><surname>Ro</surname><given-names>J.Y.</given-names></name><name><surname>Frauenhoffer</surname><given-names>E.E.</given-names></name></person-group><article-title>Bacillary angiomatosis</article-title><source>Ann Otol Rhinol Laryngol</source><volume>104</volume><issue>8</issue><year>1995</year><fpage>668</fpage><lpage>672</lpage><pub-id pub-id-type="pmid">7543738</pub-id></element-citation></ref><ref id="bib3"><label>3</label><element-citation publication-type="journal" id="sref3"><person-group person-group-type="author"><name><surname>Koehler</surname><given-names>J.E.</given-names></name><name><surname>LeBoit</surname><given-names>P.E.</given-names></name><name><surname>Egbert</surname><given-names>B.M.</given-names></name><name><surname>Berger</surname><given-names>T.G.</given-names></name></person-group><article-title>Cutaneous vascular lesions and disseminated cat-scratch disease in patients with the acquired immunodeficiency syndrome (AIDS) and AIDS-related complex</article-title><source>Ann Intern Med</source><volume>109</volume><year>1988</year><fpage>449</fpage><lpage>455</lpage><pub-id pub-id-type="pmid">3415105</pub-id></element-citation></ref><ref id="bib4"><label>4</label><element-citation publication-type="journal" id="sref4"><person-group person-group-type="author"><name><surname>Schwartz</surname><given-names>R.A.</given-names></name><name><surname>Nychay</surname><given-names>S.G.</given-names></name><name><surname>Janniger</surname><given-names>C.K.</given-names></name></person-group><article-title>Bacillary angiomatosis: presentation of six patients, some with unusual features</article-title><source>Br J Dermatol</source><volume>136</volume><issue>1</issue><year>1997</year><fpage>60</fpage><lpage>65</lpage><pub-id pub-id-type="pmid">9039296</pub-id></element-citation></ref><ref id="bib5"><label>5</label><element-citation publication-type="journal" id="sref5"><person-group person-group-type="author"><name><surname>Mayama</surname><given-names>S.</given-names></name><name><surname>Cuevas</surname><given-names>L.E.</given-names></name><name><surname>Sheldon</surname><given-names>J.</given-names></name></person-group><article-title>Prevalence and transmission of Kaposi's sarcoma-associated herpesvirus (human herpesvirus 8) in Ugandan children and adolescents</article-title><source>Int J Cancer</source><volume>77</volume><issue>6</issue><year>1998</year><fpage>817</fpage><lpage>820</lpage><pub-id pub-id-type="pmid">9714046</pub-id></element-citation></ref><ref id="bib6"><label>6</label><element-citation publication-type="journal" id="sref6"><person-group person-group-type="author"><name><surname>Dedicoat</surname><given-names>M.</given-names></name><name><surname>Newton</surname><given-names>R.</given-names></name></person-group><article-title>Review of the distribution of Kaposi’s sarcoma-associated herpesvirus (KSHV) in Africa in relation to the incidence of Kaposi’s sarcoma</article-title><source>Br J Cancer</source><volume>88</volume><year>2003</year><fpage>1</fpage><lpage>3</lpage><pub-id pub-id-type="pmid">12556950</pub-id></element-citation></ref><ref id="bib7"><label>7</label><element-citation publication-type="journal" id="sref7"><person-group person-group-type="author"><name><surname>Mutalima</surname><given-names>N.</given-names></name><name><surname>Molyneux</surname><given-names>E.</given-names></name><name><surname>Johnston</surname><given-names>W.</given-names></name></person-group><article-title>Impact of infection with human immunodeficiency virus-1 (HIV) on the risk of cancer among children in Malawi - 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This case shows BA presenting with widespread papules and fungating, ulcerating nodules mimicking KS.</p></caption><graphic xlink:href="gr1"></graphic></fig><fig id="fig2"><label>Fig 2</label><caption><p>A dramatic improvement in the appearance of the cutaneous lesions was noted within 1 week of treatment with oral doxyxycline.</p></caption><graphic xlink:href="gr2"></graphic></fig><fig id="fig3"><label>Fig 3</label><caption><p>The patient's skin showed near complete resolution of the sores and lesions after 8 weeks of treatment with oral doxycycline.</p></caption><graphic xlink:href="gr3"></graphic></fig></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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