case report and literature review Fetiform teratomain an ... · No neural tissue including spinal...
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Fetiform teratoma in an Italian-Friesian calf: case report and literature review G. CUTTONE a , F. LAUS b , G. ROSSI b , L. TIBALDI a , E. MAZZI a , V. CUTERI b , G. CATONE b a Practitioner, Mantova, Italy b School of Biosciences and Veterinary Medicine, University of Camerino BACKGROUND Fetiform teratoma is a rare form of highly developed mature teratoma that includes one or more components resembling a malformed fetus 1 . Most authors agree that fetiform terato- mas are highly developed mature teratomas; the natural hi- story of fetus in fetu, however, is controversial 1 . Fetus in fetu has often been interpreted as a fetus growing with or within its twin. As such, this interpretation assumes a special com- plication of twinning, one of several grouped under the term parasitic twin. However, classification of similar con- genital malformations is difficult because too few cases ha- ve been reported in humans and animals to provide the ba- sis for generalization. In the present paper, we describe the first case of highly dif- ferentiated extragonadal fetiform teratoma with cranial con- nection resembling a case of craniopagus parasiticus in an Italian-Friesian calf, successfully treated by surgery. CASE PRESENTATION A 35-day-old male Italian-Friesian calf weighing 55 kg was referred because of a mass on the fronto-nasal region. The delivery was without complications and the calf appeared in good condition with otherwise appropriate development of the musculoskeletal system. The asymmetrical mass was covered with hair and had well- defined margins. The long axis measured 15 cm and the short axis 10 cm. Two lateral structures of similar size and conformation were recognized as underdeveloped hind limbs, while at the center of the mass a small tail was present (Fig. 1A). Palpation revealed that the mass was not strictly adherent to the underlying tissues while bone structures were clearly pal- pable in the central area. Latero-lateral (Fig. 1B) and cranio-ventral X-ray projection revealed the presence of three bony structures: two with va- guely triangular shape and one with a more oval shape, iden- tified as the pelvic portions of the parasitic twin. Complete blood count (CBC) and the main haematochemi- cal parameters proved to be in the normal ranges. Aliquots of serum were tested for Neospora caninum and Chlamydia spp. by indirect immunofluorescence antibody tests (IFAT) and for Bovine Viral Diarrhea Virus (BVDV) and Bovine herpe- G. Cuttone et al. Large Animal Review 2016; 22: 187-189 187 N Autore per la corrispondenza: Fulvio Laus ([email protected]). SUMMARY Introduction - Fetiform teratoma is a rare form of teratoma in animals and people that resembles a malformed fetus. This paper describes the first case of highly differentiated extragonadal fetiform teratoma with cranial connection in an Italian- Friesian calf. Case presentation - A 35-day-old male Italian-Friesian calf weighing 55 kg was referred because of a mass localized in the fron- to-nasal region. The mass contained two lateral structures of similar size and conformation that were recognized as underde- veloped hind limbs, while at its center there was a small tail. The mass was surgically excised and sent to the pathologist for examination. Gross examination identified two femur-like rudimentary limbs and a sketch of bone located in between, morphologically referable to a rudimentary coxae-like bone. Some mucinous cysts, a virtual body cavity showing adipose and muscular tissues, some cartilaginous nuclei and a coelomatic body cavity were also noted. Histological examination showed differentiation into skin with dermal appendages, hair, adipose tissue, cartilage, bone, lymphoid tissue, neurovascular bundles, and a rudimentary tail. No neural tissue including spinal cord, brain matter, or gonadal differentiation was seen. On the basis of these findings, the mass was diagnosed as a highly differentiated extragonadal fetiform teratoma. Conclusion - Fetiform teratoma should be included among differential diagnoses in cases of neonatal malformation in bovi- ne. Analyzing the available literature, the Friesian genetic strain seem to be predisposed to fetal malformation, but a systema- tic reporting of cases is needed, in order to investigate further the epidemiological, etiological, pathophysiological and thera- peutic aspect of this kind of congenital disease. KEY WORDS Theriogenology, calves, malformation, fetiform teratoma.
Transcript of case report and literature review Fetiform teratomain an ... · No neural tissue including spinal...
Fetiform teratoma in an Italian-Friesian calf:case report and literature review
G. CUTTONEa, F. LAUSb, G. ROSSIb, L. TIBALDIa, E. MAZZIa, V. CUTERIb, G. CATONEb
a Practitioner, Mantova, Italyb School of Biosciences and Veterinary Medicine, University of Camerino
BACKGROUND
Fetiform teratoma is a rare form of highly developed matureteratoma that includes one or more components resemblinga malformed fetus1. Most authors agree that fetiform terato-mas are highly developed mature teratomas; the natural hi-story of fetus in fetu, however, is controversial1. Fetus in fetuhas often been interpreted as a fetus growing with or withinits twin. As such, this interpretation assumes a special com-plication of twinning, one of several grouped under theterm parasitic twin. However, classification of similar con-genital malformations is difficult because too few cases ha-ve been reported in humans and animals to provide the ba-sis for generalization.In the present paper, we describe the first case of highly dif-ferentiated extragonadal fetiform teratoma with cranial con-nection resembling a case of craniopagus parasiticus in anItalian-Friesian calf, successfully treated by surgery.
CASE PRESENTATION
A 35-day-old male Italian-Friesian calf weighing 55 kg wasreferred because of a mass on the fronto-nasal region. Thedelivery was without complications and the calf appeared ingood condition with otherwise appropriate development ofthe musculoskeletal system.The asymmetrical mass was covered with hair and had well-defined margins. The long axis measured 15 cm and theshort axis 10 cm. Two lateral structures of similar size andconformation were recognized as underdeveloped hindlimbs, while at the center of the mass a small tail was present(Fig. 1A). Palpation revealed that the mass was not strictly adherent tothe underlying tissues while bone structures were clearly pal-pable in the central area.Latero-lateral (Fig. 1B) and cranio-ventral X-ray projectionrevealed the presence of three bony structures: two with va-guely triangular shape and one with a more oval shape, iden-tified as the pelvic portions of the parasitic twin.Complete blood count (CBC) and the main haematochemi-cal parameters proved to be in the normal ranges. Aliquots ofserum were tested for Neospora caninum and Chlamydia spp.by indirect immunofluorescence antibody tests (IFAT) andfor Bovine Viral Diarrhea Virus (BVDV) and Bovine herpe-
G. Cuttone et al. Large Animal Review 2016; 22: 187-189 187
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Autore per la corrispondenza:Fulvio Laus ([email protected]).
SUMMARYIntroduction - Fetiform teratoma is a rare form of teratoma in animals and people that resembles a malformed fetus. Thispaper describes the first case of highly differentiated extragonadal fetiform teratoma with cranial connection in an Italian-Friesian calf.Case presentation - A 35-day-old male Italian-Friesian calf weighing 55 kg was referred because of a mass localized in the fron-to-nasal region. The mass contained two lateral structures of similar size and conformation that were recognized as underde-veloped hind limbs, while at its center there was a small tail. The mass was surgically excised and sent to the pathologist forexamination. Gross examination identified two femur-like rudimentary limbs and a sketch of bone located in between,morphologically referable to a rudimentary coxae-like bone. Some mucinous cysts, a virtual body cavity showing adipose andmuscular tissues, some cartilaginous nuclei and a coelomatic body cavity were also noted. Histological examination showeddifferentiation into skin with dermal appendages, hair, adipose tissue, cartilage, bone, lymphoid tissue, neurovascular bundles,and a rudimentary tail. No neural tissue including spinal cord, brain matter, or gonadal differentiation was seen. On the basisof these findings, the mass was diagnosed as a highly differentiated extragonadal fetiform teratoma.Conclusion - Fetiform teratoma should be included among differential diagnoses in cases of neonatal malformation in bovi-ne. Analyzing the available literature, the Friesian genetic strain seem to be predisposed to fetal malformation, but a systema-tic reporting of cases is needed, in order to investigate further the epidemiological, etiological, pathophysiological and thera-peutic aspect of this kind of congenital disease.
KEY WORDSTheriogenology, calves, malformation, fetiform teratoma.
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188 Fetiform teratoma in an Italian-Friesian calf: case report and literature review
svirus-1 (BoHV-1) by enzyme-linked immunosorbent assay(ELISA). RT-PCR and Nested PCR were used to test the bulkmilk for BVDV and BoHV-1, respectively. All tests on serumand milk gave negative results.Cardiac auscultation, electrocardiography, thoracic and ab-dominal ultrasonography did not reveal any abnormality.The mass was surgically excised and sent to the pathologistfor examination (Fig. 2).The calf was discharged 11 days after surgery and eightmonths later was still in good condition as a normal subject.On gross examination, the well-circumscribed mass excisedfrom the cranial region showed on bisection two rudimen-tary limbs, each containing an incomplete long bone resem-
bling a femur, and a sketch of bone located between the twoappendices, morphologically referable to a rudimentarycoxae-like bone (Fig. 3). Inside the excised mass, some smallcysts filled with mucinous material were also seen, protru-ding within a virtual body cavity, whose cut section showedadipose and muscular tissues, and some cartilaginous nucleithat resemble other sketches of bone delimitating a coelo-matic body cavity. Multiple cuts did not reveal any axial ske-leton or cephalic differentiation. Multiple gross sections,confirmed by the histological examination of the differentportions of the mass, showed differentiation into skin withdermal appendages, hair, adipose tissue, cartilage, bone,lymphoid tissue, neurovascular bundles, and rudimentary
Figure 1 - A) Clinical appearance of the mass. Note the underdeveloped hind limbs (arrowheads) and the small tail (arrow). B) Latero-late-ral X-ray projection of the head. The central structure (arrows) was identified as the pelvic portions of the fetiform teratoma.
Figure 2 - Intraoperative picture of the surgery: A) before and B) after removal of the bony mass.
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INFORMAZIONI:Segreteria di Redazione - [email protected]
tail. No neural tissue including spinal cord, brain matter, orgonadal differentiation was seen. On the basis of these fin-dings, the mass was diagnosed as a highly differentiated ex-tragonadal fetiform teratoma.
DISCUSSION
Teratomas are embryonal neoplasms composed of tissue de-rived from all three germ layers. They can be extragonadal orgonadal and arise from primordial germ cells that may beco-me stranded during their migration, coming to rest at extra-gonadal sites. Fetiform teratomas should not be confusedwith fetus in fetu, which is invariably associated with anen-cephaly and achardia2,3,4; difference in the origin of the twohas been described in the literature3. Unlike classical terato-mas, fetiform teratomas have complex tissue differentia-tion/organization and organoid differentiation. Usually thecaudal development is better than the cephalic one, as in thepresent case, which entirely lacks cephalic differentiation.Limb formation is seen more often, while visceral organ tis-sue and skeletal muscle are inconspicuous or absent, as inthis case.In our case, on the basis of his tissue differentiation and theabsence of a head or central and peripheral nervous system,the fetus-like structure may be classified as fetiform teratoma,
a rare form of mature teratoma that include one or morecomponents resembling a malformed fetus. This teratomadiffers from “fetus in fetu” because it appears to contain com-plete organ systems, even major body parts such as the torso,tail, and limbs. Fetus in fetu differs from fetiform teratoma inhaving an apparent spine1.In our case, surgery was performed successfully and no otherabnormalities were detected on the autosite. Although we cannot establish a breed predisposition, it is in-teresting to note that most (4 out of 5) cases of parasitictwins reported in bovine have occurred in the Friesian gene-tic strain5,6,7,8,9.There is a dearth of epidemiologic, clinical and pathologicalinformation about these congenital malformations becausethe heterogeneous terminology can cause confusion and al-so because abnormalities tend to be underreported. Syste-matic reporting of cases of fetal malformation should be en-couraged, in order to provide the basis for further investiga-tion of the epidemiological, etiological, pathophysiologicaland therapeutic aspects of this kind of congenital disease.
ACKNOWLEDGEMENTS
We thank Sheila Beatty who provided language editing services.
References
1. Kuno N., Kadomatsu K., Nakamura M., Miwa-Fakuchi T., HirabayashiN., Ishizuka T. (2004) Mature ovarian cystic teratoma with a highly dif-ferentiated homunculus: a case report. Birth Defects Res A Clin Mol Te-ratol, 70:40-46.
2. Spencer R. (1992) Conjoined twins: theoretical embryologic basis. Te-ratology, 6:591-602.
3. Greenberg J.A., Clancy T.E. (2008) Fetiform teratoma (humunculus).Rev Obstet Gynecol, 1:95-96.
4. Weiss J.R., Burgess J.R., Kaplan K.J. (2006) Fetiform teratoma (homun-culus). Arch Pathol Lab Med, 130:1552-1556.
5. Abt D.A., Croshaw J.E., Hare W.C.D. (1962) Monocephalus dipygus pa-rasiticus and other anomalies in a calf. J Am Vet Med Assoc, 141:1068-1072.
6. Gordon A.S.M., Lowe R.J. (1973) A bovine double monster: clinicalanatomical, and embryological considerations. Vet Rec, 93:67.
7. Hiraga T., Dennis S.M. (1989) Congenital duplication. Vet Clin NorthAm Food Anim Pract, 9:145-161.
8. Leipold H.W., Dennis S.M., Huston K. (1972) Embryonic duplicationin cattle. Cornell Veterinarian, 62:575-580.
9. Turner C.W. (1936) Pygopagus Parasitic Bovine Twins Involving the Ud-der. Department of Dairy Husbandry, University of Missouri, ColumbiaMissouri Agricultural Experiment Station, Journal Series No. 410.
Figure 3 - Sketch of bone, found inside the excised mass,morphologically referable to a rudimentary coxae-like bone.
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