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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Porencephaly in a fennec fox (<italic>Vulpes zerda</italic>)</title><author><name sortKey="Yamazaki, Mutsumi" sort="Yamazaki, Mutsumi" uniqKey="Yamazaki M" first="Mutsumi" last="Yamazaki">Mutsumi Yamazaki</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author><author><name sortKey="Yoshimoto, Saeko" sort="Yoshimoto, Saeko" uniqKey="Yoshimoto S" first="Saeko" last="Yoshimoto">Saeko Yoshimoto</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author><author><name sortKey="Ishikawa, Tomoko" sort="Ishikawa, Tomoko" uniqKey="Ishikawa T" first="Tomoko" last="Ishikawa">Tomoko Ishikawa</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author><author><name sortKey="Une, Yumi" sort="Une, Yumi" uniqKey="Une Y" first="Yumi" last="Une">Yumi Une</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">27523321</idno><idno type="pmc">5138435</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5138435</idno><idno type="RBID">PMC:5138435</idno><idno type="doi">10.1292/jvms.16-0121</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">000040</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000040</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Porencephaly in a fennec fox (<italic>Vulpes zerda</italic>)</title><author><name sortKey="Yamazaki, Mutsumi" sort="Yamazaki, Mutsumi" uniqKey="Yamazaki M" first="Mutsumi" last="Yamazaki">Mutsumi Yamazaki</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author><author><name sortKey="Yoshimoto, Saeko" sort="Yoshimoto, Saeko" uniqKey="Yoshimoto S" first="Saeko" last="Yoshimoto">Saeko Yoshimoto</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author><author><name sortKey="Ishikawa, Tomoko" sort="Ishikawa, Tomoko" uniqKey="Ishikawa T" first="Tomoko" last="Ishikawa">Tomoko Ishikawa</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author><author><name sortKey="Une, Yumi" sort="Une, Yumi" uniqKey="Une Y" first="Yumi" last="Une">Yumi Une</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author></analytic><series><title level="j">The Journal of Veterinary Medical Science</title><idno type="ISSN">0916-7250</idno><idno type="eISSN">1347-7439</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>A postmortem examination revealed a large brain cavity in the right cerebral hemisphere
of a 9-year-old male fennec (<italic>Vulpes zerda</italic>). The cavity was filled with
cerebrospinal fluid and extended to the right lateral ventricle. Swelling and displacement
of the right hippocampal area were also observed. Histologic examination revealed no
evidence of previous infarct lesions, hemorrhage, inflammation or invasive tumor cells.
Observation of the defective part suggested a local circulatory disorder during the fetal
stage, although the cause was not detected. No neurological symptoms that could enable a
provisional diagnosis were observed during the course of his life. This is the first
report of asymptomatic porencephaly in a fennec fox.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Cantile, C" uniqKey="Cantile C">C. Cantile</name></author><author><name sortKey="Chianini, F" uniqKey="Chianini F">F. Chianini</name></author><author><name sortKey="Arispici, M" uniqKey="Arispici M">M. Arispici</name></author><author><name sortKey="Fatzer, R" uniqKey="Fatzer R">R. Fatzer</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Davies, E S" uniqKey="Davies E">E. S. Davies</name></author><author><name sortKey="Volk, H A" uniqKey="Volk H">H. A. Volk</name></author><author><name sortKey="Behr, S" uniqKey="Behr S">S. Behr</name></author><author><name sortKey="Summers, B" uniqKey="Summers B">B. Summers</name></author><author><name sortKey="De Lahunta, A" uniqKey="De Lahunta A">A. de Lahunta</name></author><author><name sortKey="Syme, H" uniqKey="Syme H">H. 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<front><journal-meta><journal-id journal-id-type="nlm-ta">J Vet Med Sci</journal-id><journal-id journal-id-type="iso-abbrev">J. Vet. Med. Sci</journal-id><journal-id journal-id-type="publisher-id">JVMS</journal-id><journal-title-group><journal-title>The Journal of Veterinary Medical Science</journal-title></journal-title-group><issn pub-type="ppub">0916-7250</issn><issn pub-type="epub">1347-7439</issn><publisher><publisher-name>The Japanese Society of Veterinary Science</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">27523321</article-id><article-id pub-id-type="pmc">5138435</article-id><article-id pub-id-type="publisher-id">16-0121</article-id><article-id pub-id-type="doi">10.1292/jvms.16-0121</article-id><article-categories><subj-group subj-group-type="heading"><subject>Wildlife Science</subject><subj-group><subject>Note</subject></subj-group></subj-group></article-categories><title-group><article-title>Porencephaly in a fennec fox (<italic>Vulpes zerda</italic>)</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>YAMAZAKI</surname><given-names>Mutsumi</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>YOSHIMOTO</surname><given-names>Saeko</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>ISHIKAWA</surname><given-names>Tomoko</given-names></name><xref ref-type="aff" rid="aff2"><sup>2</sup></xref></contrib><contrib contrib-type="author"><name><surname>UNE</surname><given-names>Yumi</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref><xref rid="cor1" ref-type="corresp"><sup>*</sup></xref></contrib><aff id="aff1"><label>1)</label>Laboratory of Veterinary Pathology, Azabu University, 1–17–71 Fuchinobe, Chuo-ku, Sagamihara, Kanagawa 252–5201, Japan</aff><aff id="aff2"><label>2)</label>Hamura Zoo, 4122 Hane, Hamura, Tokyo 205–0012, Japan</aff></contrib-group><author-notes><corresp id="cor1"><label>*</label>Correspondence to: Une, Y., Laboratory of Veterinary
Pathology, School of Veterinary Medicine, Azabu University, 1–17–71 Fuchinobe, Chuo-ku,
Sagamihara, Kanagawa 252–5201, Japan. e-mail: <email xlink:href="une@azabu-u.ac.jp">une@azabu-u.ac.jp</email></corresp></author-notes><pub-date pub-type="epub"><day>11</day><month>8</month><year>2016</year></pub-date><pub-date pub-type="ppub"><month>11</month><year>2016</year></pub-date><volume>78</volume><issue>11</issue><fpage>1749</fpage><lpage>1751</lpage><history><date date-type="received"><day>07</day><month>3</month><year>2016</year></date><date date-type="accepted"><day>28</day><month>7</month><year>2016</year></date></history><permissions><copyright-statement>©2016 The Japanese Society of Veterinary Science</copyright-statement><copyright-year>2016</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-nd/4.0/"><license-p>This is an open-access article distributed under the terms of the Creative
Commons Attribution Non-Commercial No Derivatives (by-nc-nd) License. </license-p></license></permissions><abstract><p>A postmortem examination revealed a large brain cavity in the right cerebral hemisphere
of a 9-year-old male fennec (<italic>Vulpes zerda</italic>). The cavity was filled with
cerebrospinal fluid and extended to the right lateral ventricle. Swelling and displacement
of the right hippocampal area were also observed. Histologic examination revealed no
evidence of previous infarct lesions, hemorrhage, inflammation or invasive tumor cells.
Observation of the defective part suggested a local circulatory disorder during the fetal
stage, although the cause was not detected. No neurological symptoms that could enable a
provisional diagnosis were observed during the course of his life. This is the first
report of asymptomatic porencephaly in a fennec fox.</p></abstract><kwd-group><kwd>asymptomatic</kwd><kwd>cortex</kwd><kwd>fennec fox</kwd><kwd>porencephaly</kwd><kwd>spontaneousness</kwd></kwd-group></article-meta></front><body><p>Porencephaly is a rare morphological abnormality of the brain, characterized by cerebral
cavities or cysts, and sometimes spreads to the ventricular system or subarachnoid space
[<xref rid="r2" ref-type="bibr">2</xref>, <xref rid="r4" ref-type="bibr">4</xref>, <xref rid="r11" ref-type="bibr">11</xref>, <xref rid="r17" ref-type="bibr">17</xref>]. Unlike
hydranencephaly, the brain defect in porencephaly is limited to the cerebral hemisphere [<xref rid="r12" ref-type="bibr">12</xref>]. In humans, porencephaly commonly manifests in infants
as a congenital disorder [<xref rid="r4" ref-type="bibr">4</xref>], but in animals, it often
occurs in livestock (e.g., cattle, sheep and goats) where prenatal virus infections may
represent a predisposing factor [<xref rid="r3" ref-type="bibr">3</xref>, <xref rid="r5" ref-type="bibr">5</xref>, <xref rid="r6" ref-type="bibr">6</xref>, <xref rid="r12" ref-type="bibr">12</xref>, <xref rid="r16" ref-type="bibr">16</xref>]. In dogs and
cats, porencephaly occurs sporadically, and the cause remains unknown [<xref rid="r1" ref-type="bibr">1</xref>, <xref rid="r2" ref-type="bibr">2</xref>, <xref rid="r9" ref-type="bibr">9</xref>, <xref rid="r10" ref-type="bibr">10</xref>, <xref rid="r11" ref-type="bibr">11</xref>, <xref rid="r17" ref-type="bibr">17</xref>].</p><p>Generally, anatomical brain anomalies are often associated with seizures; however,
porencephaly can be an incidental finding in asymptomatic animals as well. Here, we report a
case of a 9-year-old male fennec fox that suddenly presented with anorexia, unsteady gait and
jaundice of the visible mucous membranes. He was treated with a liver tonic, prednisone and
ampicillin, resulting in alleviation of symptoms. Unfortunately, after one week, signs of
jaundice re-appeared, accompanied by loss of appetite. He died a month later and was found to
have lost more than 200 g of body weight before death. On postmortem examination, jaundice of
the oral mucosa and subcutaneous tissue was observed. Swelling of the superficial cervical,
axillary, inguinal, mesenteric, pancreaticoduodenal and anterior mediastinal lymph nodes was
significant. Also seen were right ventricular dilatation, myocardial degeneration and
cicatrices of the external surfaces in both kidneys. The liver and spleen were also swollen,
and the parenchyma of the liver was fragile. Gross examination of the head revealed asymmetry
of the skull and a large cavity in the right cerebral cortex. The maxilla had a small dent on
the right side and a bulge on the lateral side of it. In the brain, the frontal lobe and only
part of the parietal lobe along the central groove were left, whereas most of the right
cortical cortex was replaced by a cavity filled with cerebrospinal fluid (CSF) (<xref ref-type="fig" rid="fig_001">Figs. 1</xref><fig orientation="portrait" fig-type="figure" id="fig_001" position="float"><label>Fig. 1.</label><caption><p>The cavity in the affected region of the brain: Most of the right cortical cortex was
replaced by a cavity filled with cerebrospinal fluid (CSF). The cavity decreased in
size due to outflow of CSF.</p></caption><graphic xlink:href="jvms-78-1749-g001"></graphic></fig> and <xref ref-type="fig" rid="fig_002">2</xref><fig orientation="portrait" fig-type="figure" id="fig_002" position="float"><label>Fig. 2.</label><caption><p>Excised brain: The frontal region and a part of the parietal lobe along the central
groove remained. A part of the corpus callous and the brainstem can be seen. The cavity
decreased in sign due to defluxion of CSF.</p></caption><graphic xlink:href="jvms-78-1749-g002"></graphic></fig>). The cerebellar vermis also showed asymmetry. On the formalin fixed brain cut
sections, the cavity extended to the dilated right lateral ventricle. Swelling and
displacement of the right hippocampus were also observed (<xref ref-type="fig" rid="fig_003">Fig. 3</xref><fig orientation="portrait" fig-type="figure" id="fig_003" position="float"><label>Fig. 3.</label><caption><p>A serial frontal plane section of the brain: The cavity extends to the right lateral
ventricle (asterisk), which may have resulted in swelling and displacement of the right
hippocampal aspect (arrowhead).</p></caption><graphic xlink:href="jvms-78-1749-g003"></graphic></fig>).</p><p>For histologic analysis, major organs were fixed in a 10% phosphate-buffered formalin
solution. The brain was cut into 5-<italic>µ</italic>m-thick sections from the frontal plane,
embedded in paraffin, and stained with hematoxylin and eosin (HE) according to standard
procedures. The brain sections were also stained with Kluver-Barrera (KB).
Immunohistochemistry (IHC) staining was also performed on paraffin wax sections of other
organs, such as lymph nodes. The sections were first incubated with primary antibodies to CD3
(Dako, Carpinteria, CA, U.S.A.) and CD20 (Thermo Fisher Scientific, Waltham, MA, U.S.A.),
followed by peroxidase-conjugated secondary antibody (Nichirei, Tokyo, Japan). The IHC signal
was visualized using diaminobenzidine. Histologic examination of a cross section of the
thalamus and cerebral peduncle revealed atrophy of the choroid plexus in the right lateral
ventricle and marked edema in the neuropil beneath the cyst, including the right hippocampus
(<xref ref-type="fig" rid="fig_004">Fig. 4</xref><fig orientation="portrait" fig-type="figure" id="fig_004" position="float"><label>Fig. 4.</label><caption><p>Hematoxylin and eosin image of a cross-section of the thalamus and cerebral peduncle:
Atrophy of the choroid plexus in the right lateral ventricle is observed, along with
marked edema in the neuropil beneath the cyst. The cyst wall is contiguous with the
arachnoid (arrow).</p></caption><graphic xlink:href="jvms-78-1749-g004"></graphic></fig>); myelinoclasis was also detected in the same area by KB staining.
However, we did not observe any abnormal nerve cells, neuronal necrosis, fibrosis or aberrant
structure of the cortex layer. We also confirmed the cyst wall as a membraneous structure
contiguous with the arachnoid (<xref ref-type="fig" rid="fig_004">Fig. 4</xref>). The swelling
of mesenteric and other lymph nodes appeared to indicate evidence of lymphocytic infiltration.
Tumor cells were also seen in the capsule and adipose tissue around the lymph nodes. The tumor
cells appeared round or ellipsoid in shape with an eosinophilic cytoplasm, an atypical nucleus
and clear multiple nucleoli. Many mitotic and apoptotic cells were also detected in the tumor
tissue as well. Moreover, tumor cells were observed in the parenchyma of the lung, heart,
liver, kidneys and testicles. These tumor cells were phenotypically positive for CD3 and
negative for CD20 antibodies. Histologic examination also revealed that the cicatrices in both
kidneys were fibrotic.</p><p>With respect to this case, the cavity was present in the right cerebral hemisphere and
extended to the right lateral ventricle and into the subarachnoid space. In addition, no
abnormal nerve cells or unusual cortex layer was seen. Moreover, the animal did not show any
neurological manifestations during his lifetime. Because of these reasons, we diagnosed this
case as a porencephaly. It is worth noting that hydranencephaly and schizencephaly are
differential diagnosis for porecephaly. However, hydranencephaly is characterized by the
complete or almost complete absence of the cerebral hemispheres, leaving only membranous sacs
filled with CSF and enclosed by leptomeninges [<xref rid="r12" ref-type="bibr">12</xref>]. By
contrast, schizencephaly is characterized by full thickness clefts spanning the wall of the
cerebral hemispheres that are lined and surrounded by polymicrogyric cortex [<xref rid="r13" ref-type="bibr">13</xref>].</p><p>Porencephaly is more commonly seen in livestock animals, but it is rarely reported in dogs
and cats. In exotic animals, porecephaly is also rare, although a few cases have been reported
in cynomolgous monkeys [<xref rid="r7" ref-type="bibr">7</xref>, <xref rid="r14" ref-type="bibr">14</xref>]. Therefore, this article is the first to report a case of porencephaly
in a fennec fox. Although hippocampal atrophy was previously observed in porencephaly of
humans and dogs [<xref rid="r8" ref-type="bibr">8</xref>, <xref rid="r9" ref-type="bibr">9</xref>], only advanced hydropic degeneration of the cortex and hippocampal neuropils
beneath the cyst was found in this case. Furthermore, necrosis and fibrosis of neurons did not
occur in these areas, suggesting that the hydropic degeneration may be an acute response.
Remarkably, this fennec did not show any signs of seizure or neurologic manifestation over the
course of his life, and these results indicated that the animal was well-adapted. In canine,
brain anomalies like porencephaly have been shown to have some correlation with seizures
[<xref rid="r2" ref-type="bibr">2</xref>, <xref rid="r10" ref-type="bibr">10</xref>, <xref rid="r15" ref-type="bibr">15</xref>], giving us reason to believe that this may be an
asymptomatic case of the abnormality.</p><p>The risk factors for porencephaly include ischemia, hemorrhage, inflammation, infection,
trauma and abnormal development of CNS during the prenatal period. In humans, virus infection
or vascular insufficiency has been implicated as major causes of secondary porencephaly. While
a mutation in the procollagen type IV α 1 gene (Col4a1) was reported in mice with porencephaly
[<xref rid="r4" ref-type="bibr">4</xref>], a similar genetic correlation has not been noted
in other animals [<xref rid="r10" ref-type="bibr">10</xref>]. In many humans and animals, the
actual cause is unclear; therefore, a study of a large number of cases is useful for
elucidation of the cause of porencephaly.</p><p>In this report, any cause of secondary porencephaly including invasive tumor cells was not
detected in the brain. There was also no clinical sign of seizure or neurologic manifestation
and malformation of the skull that was also detected in this fennec, leading to the
speculation that this case of porencephaly may be a congenital one. The defective regions (the
right occipital lobe and part of the temporal lobe) are predominantly supplied by the right
middle cerebral artery. Thus, the cause may be a local circulatory disorder in the fetal
stage. However, we could not establish the cause of porencephaly. The cause of death could
have been related to severe multiple organ failure due to the multicentric and disseminated
T-cell lymphoma.</p></body><back><ack><p>We wish to thank Dr. Takayuki Mineshige of Azabu University for his
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