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Gastric teratoma in infants
Gastric Teratoma in Infants Joaquin Azpiroz C., MD, Mexico City, Mexico Eleazar Valle M., MD, Mexico City, Mexico Arnold0 Herberth F., MD, Monterrey, Mexico Vicente Carrilto A., MD, Mexico City, Mexico Rosa Vaca G., MD, Mexico City. Mexico
Teratomas are true congenital tumors composed of abnormal tissues foreign to the area in which they are found. These tumors can be either benign or malignant depending on their structure and behavior. Teratomas are uncommon in children and occur more frequently in median locations; however, their occurrence in the abdominal viscera is not usual [I-3]. Only twenty-three cases of gastric teratomas in pediatric patients have been reported [4-81. Because of their rarity, we believe it of interest to report an additional case and to discuss the clinical and therapeutic characteristics of these tumors. Case Report The patient (GTM), an eight month old male infant, was admitted to La Raza Medical Center because of hematemesis and melena. His parents had noticed progressive enlargement of the abdomen over a three month period. Three days prior to his admission the infant vomited coffee-ground material on four occasions and passed three tarry stools within twenty-four hours. Physical examination revealed a well developed, undernourished, pale infant weighing 5,940 gm. Temperature was 37.3”C. The abdomen was markedly distended, particularly on the left side, where a firm, nonlobulated mass could be palpated. This mass which measured about 8 cm in diameter extended from the mesogastrium to the left upper and lower quadrants of the abdo-
From the Departments of Pediatric Surgery and Pathology, La Raza Mediccl Center, I.M.S.S.. Mexico City, Mexico. Reprint requests should be addressed to Dr Joaquh Azpiroz, Amores 830. Mexico 12, D.F.
vefume 129, Seplember
1974
men and the renal fossa. Findings were otherwise normal. Laboratory studies indicated a hemoglobin of 8.2 gm/lOO ml, hematocrit of 31 per cent, white blood cell count of 12,800 per mm3 with 31 per cent segmented forms, 65 per cent lymphocytes, and 2 per cent monocytes. The erythrocytes demonstrated marked anisocytosis and polychromatophilia. Sedimentation rate was 12 mm/hr, and platelet count was 408,000 per mm:‘. Urine was normal. Blood values were as follows: glucose, 111 mg/lOO ml; blood urea nitrogen, 16.2 mg/lOO ml; creatinine, 1 mg/lOO ml; total cholesterol, 120 mg/lOO ml; total protein, 7.1 gm with 3.9 gm/lOO ml albumin and 3.2 gm/lOO ml globulin. The alkaline phosphatase was 4.9 units. Lactic dehydrogenase was 780 units. Direct bilirubin was 0.4 mg/lOO ml and indirect was 0.06 mg/lOO ml. Urinary excretion of catecholamines was 1.4/24 hr. Prothrombin time was 100 per cent at 11 seconds. X-ray studies, including intravenous pyelography, gastrointestinal series, and x-ray films of the chest, skull, and skeleton, showed a large mass in the left upper quadrant producing upward displacement of the stomach and right lateral displacement of the loops of small bowel. The infant was given blood transfusion in preparation for laparotomy. On the tenth hospital day, surgical exploration was performed through a supraumbilical transverse incision. An ovoid mass originating in the posterior wall of the stomach was found. This mass was pinkish gray, soft, and well encapsulated and was adherent to the transverse colon, pancreas, and duodenum. The mass was freed from these structures by dissection, but since it could not be separated from the stomach, a section from the posterior wall of this organ was removed along with the tumor. A Stamm gastrostomy was also performed.
429
Azpiroz et al
Figure 1. Cut surface with spongy appearance due to the presence of numerous cysts of varying size.
F&ure 3. Fibrous connective tissue and cysts covered with epithelium and cartilage are evident. On pathologic study the specimen consisted of an ovoid tumor that measured 11 by 7.5 by 6.5 cm, weighed 290 gm, and was soft in consistency. The capsule was 3 mm in thickness and the mass contained pinkish white soft spongy tissue. (Figure 1.) Within this tissue, there were some fibrous grayish bands crossing in several directions. Histologic study revealed structures derived from the different germ layers, particularly cartilage, bone, and glandular formations of columnar epithelium resembling small intestine. (Figure 2.) Other structures included lymphatic follicles, nervous tissue and nervous plexus, and stratified squamous epithelium. No cellular atypias were found and most of the different tissues were well differentiated. The tumor was considered a benign solid gastric teratoma. (Figures 3,4, and 5.) Postoperatively the patient was maintained on continuous intravenous fluids and open drainage of the gastrostomy tube. Normal intestinal peristalsis started on the fourth postoperative day. Oral feedings .were given on the sixth day after surgery. The gastrostomy tube was kept in the stomach for a total of ten days, and the
patient was discharged from the hospital on the twentieth postoperative day. (Table I.)
430
Figure 2. of Photomicrograph gastric watt. Along the muscle layers are cystic formations covered by columnar epitheiium.
Ftgure 4. Nervous tissue.
Comments Teratomas are true neoplasms composed of multiple tissues foreign to the site in which they occur. Literally, teratoma is defined as a malformation and tumor at the same time. The most common sites of occurrence are the sacrococcygeal region, retroperitoneal space, and gonads, but they may appear in other places [l-3,7,8]. Since there are very few reports on teratomas of the stomach, we may conclude that they are rare. A review of the literature indicated that the first case in a pediatric patient was reported by Polony in 1936 [12]. The second registered case was that of Selman [13] in 1943 who described a teratoma arising from the anterior wall of the stomach. A total of twenty-three cases of gastric teratoma in pediatric patients have been reported. Age. Gastric teratomas usually present early in life. Most of the cases in pediatric patients were diagnosed within the first year of life, with the exception of Polony’s case in 1936 and Chaterjee’s in
The Amerkan Journal of Surgery
Gastric
TABLE I
1964 which were discovered in children six and two years of age, respectively. Ten of the reported cases occurred in newborns. ,Teratomas of the stomach rarely occur in adults [4,5,7,8,15]. Sex. It is interesting that all gastric teratomas have occurred in male patients and that teratomas at all other locations have a higher incidence in female patients. Clinical Features. In reviewing the twenty-three reported cases and our own, we found that the main clinical features were abdominal distention and a palpable abdominal mass, being present in twenty patients. Other accompanying manifestations included gastrointestinal bleeding in five patients, vomiting in five, respiratory distress in two, and an abdominal mass in three newborns causing difficult delivery. Fever and abdominal tenderness were present on one occasion each. Radiologic Findings. Radiologic study frequently reveals large calcifications resembling bone and teeth, in contrast to those seen in neuroblastoma or Wilms’ tumor which are very small and of light density. Only thirteen of the reported cases had evidence of calcification on x-ray films. Excretory urography usually shows nothing abnormal and a gastrointestinal series may show deformity of the stomach from extrinsic compression [7,8,15]. Gastric teratomas may arise in any portion of the stomach, fundus, body, or antrum. They may be exo- or endogastric and may vary in size. There have been thirteen exogastric tumors, two were endogastric, and five were both exo- and endogastric; in four the type was not reported. In pediatric patients it is difficult to be aware of the possibility of gastric tumors because of their low incidence. In making a differential diagnosis one should consider meconium peritonitis, neuro-
Volume128,Septemberl974
Infants in
Reported Cases of Teratoma of the Stomach in Pediatric Patients Author
Figure 5. Connective and fibroid tissue and llpoid ceils are Sean.
Teratoma
Polony [ 721 Selman 1731 Large et al (6) Handelsman, Reinhoff, Ward 1771 Reggianini [4] Lodzinski [7] Sarinana, Silva, and Esparza [9] Cooray [14] Ravitch (71 Wagenmann Paul 1741 Kelley (41 Chaterjee [ 71 Panabokke [7] Verhaak [ 751 Lee (81 lkeda [8] Atwell, Claireaux, and Nixon [IO] Meadow (51 Cozzi [7] De Angelis (41 Lebreuil et al (81
Year
1936
and
1943 1952 1955
1955 1956 1958
1959 1960 1961 1962 1963 1964 1964 1964 1964 1966 1967 1968 1968 1969 1969
Patient’s Sex and Age M, 6yr M, 3mo M, 7mo
Outcome Recovered
M,4mo
Recovered Recovered Recovered
M, 1 mo M, 5 days M, 1 mo
Recovered Recovered Recovered
M, 1 mo
Died Recovered
M, 10mo M, 19 days M,9mo M, 1 day M, 2 yr M, 2 mo M, 15 days M, 1 day M) ...
... Recovered Recovered Recovered Died Recovered Recovered
...
M, 1 day
Recovered
M, M, M, M,
Recovered Recovered Recovered Recovered
1 day 3mo 1 day 1 day
blastoma, Wilms’ tumor, gastric duplications, and retroperitoneal teratoma. On the basis of the cases reported, gastric teratomas are benign, with a good prognosis and no reported recurrences after resection. Treatment. Treatment of these tumors is surgical excision with partial gastrectomy or resection of the wall in which the tumor arises. In one instance total gastrectomy was performed and the procedure was well tolerated 141.Complications reported after these operations are not frequent and they may consist of anemia and failure to thrive. Of the twenty-four patients reported, only three died. Summary
Gastric teratoma is a rare condition, particularly in pediatric patients. Only twenty-three cases have previously been reported in the literature. A case of gastric teratoma in an eight month old male infant is presented. The main clinical features were progressive enlargement of the abdomen, hematemesis, and melena. At laparotomy, an ovoid mass originating in the posterior wall of the stomach was found.
431
Azpiroz
et al
Histologic findings consisted of structures derived from the different germ layers, particularly cartilage, bone, and glandular formations resembling small intestine. The clinical and pathologic findings in these cases are reviewed and therapeutic considerations are presented. References 1. Willis RA: Teratomas. Washington, DC, The Armed Forces Institute of Pathology, 195 1, p 9. 2. Dargeon H: Tumors of Childhood. New York, Hoeber, 1960. 3. Salas MM: Las neoplasias en 10s nines. MBxico, DF, MBdicas del Hosp. Inf. de Mex, 1968. 4. De Angelis VR: Gastric teratoma in a newborn infant: total gastrectomy with survival. Surgery 66: 794, 1969. 5. Meadow SR: Gastric teratoma. Postgrad Med J 44: 183, 1968.
432
6. Large HL et al: Gastric tridermal teratoma in infancy. JAMA 28: 824, 1952. 7. Cozri F et al: Teratoma dello stomaco in eta pediatrica: presentazione di un case clinic0 e rassegna della literatura. Arch /ta/ Pediatr 26: 240, 1968. 8. Lebreuil G et al: Teratome gastrique chez un nouveau-n& Arch Anat PatholParis 17: 47, 1969. 9. Sarihana C, Silva CA, Esparza H: Teratoma tridermico del estomago en un ni;o de 45 dias de edad tratado con exito por gastrectomia partial. /30/ Med Hasp Inf Mex 15: 529, 1958. 10. Atwell JD, Claireaux AE, Nixon HH: Teratoma of the stomach in the newborn. J Pediatr Surg 2: 197, 1967. 11. Handelsman JC, Reinhoff WF, Ward GE: Benign teratoma of the stomach in an infant. Am J Dis ChiM90: 196, 1955. 12. Polony M: Tgratome exogastrique. Mem Acad Chir 62: 622, 1936. 13. Selman AN: Complex tridermal teratoma of the stomach. Case report. Am J Surg 59: 567, 1943. 14. Paul M, Cooray GH, Wickremasinghe SY: Teratoma of the stomach. Br J Surg 50: 154, 1982. 15. Aubrespy P, Derlon S: TBratome gastrique chez un nouveau-n& Mem Acad Chir 96: 528, 1970.
The American Journal of Surgery
Teratomas are true congenital tumors composed of abnormal tissues foreign to the area in which they are found. These tumors can be either benign or malignant depending on their structure and behavior. Teratomas are uncommon in children and occur more frequently in median locations; however, their occurrence in the abdominal viscera is not usual [I-3]. Only twenty-three cases of gastric teratomas in pediatric patients have been reported [4-81. Because of their rarity, we believe it of interest to report an additional case and to discuss the clinical and therapeutic characteristics of these tumors. Case Report The patient (GTM), an eight month old male infant, was admitted to La Raza Medical Center because of hematemesis and melena. His parents had noticed progressive enlargement of the abdomen over a three month period. Three days prior to his admission the infant vomited coffee-ground material on four occasions and passed three tarry stools within twenty-four hours. Physical examination revealed a well developed, undernourished, pale infant weighing 5,940 gm. Temperature was 37.3”C. The abdomen was markedly distended, particularly on the left side, where a firm, nonlobulated mass could be palpated. This mass which measured about 8 cm in diameter extended from the mesogastrium to the left upper and lower quadrants of the abdo-
From the Departments of Pediatric Surgery and Pathology, La Raza Mediccl Center, I.M.S.S.. Mexico City, Mexico. Reprint requests should be addressed to Dr Joaquh Azpiroz, Amores 830. Mexico 12, D.F.
vefume 129, Seplember
1974
men and the renal fossa. Findings were otherwise normal. Laboratory studies indicated a hemoglobin of 8.2 gm/lOO ml, hematocrit of 31 per cent, white blood cell count of 12,800 per mm3 with 31 per cent segmented forms, 65 per cent lymphocytes, and 2 per cent monocytes. The erythrocytes demonstrated marked anisocytosis and polychromatophilia. Sedimentation rate was 12 mm/hr, and platelet count was 408,000 per mm:‘. Urine was normal. Blood values were as follows: glucose, 111 mg/lOO ml; blood urea nitrogen, 16.2 mg/lOO ml; creatinine, 1 mg/lOO ml; total cholesterol, 120 mg/lOO ml; total protein, 7.1 gm with 3.9 gm/lOO ml albumin and 3.2 gm/lOO ml globulin. The alkaline phosphatase was 4.9 units. Lactic dehydrogenase was 780 units. Direct bilirubin was 0.4 mg/lOO ml and indirect was 0.06 mg/lOO ml. Urinary excretion of catecholamines was 1.4/24 hr. Prothrombin time was 100 per cent at 11 seconds. X-ray studies, including intravenous pyelography, gastrointestinal series, and x-ray films of the chest, skull, and skeleton, showed a large mass in the left upper quadrant producing upward displacement of the stomach and right lateral displacement of the loops of small bowel. The infant was given blood transfusion in preparation for laparotomy. On the tenth hospital day, surgical exploration was performed through a supraumbilical transverse incision. An ovoid mass originating in the posterior wall of the stomach was found. This mass was pinkish gray, soft, and well encapsulated and was adherent to the transverse colon, pancreas, and duodenum. The mass was freed from these structures by dissection, but since it could not be separated from the stomach, a section from the posterior wall of this organ was removed along with the tumor. A Stamm gastrostomy was also performed.
429
Azpiroz et al
Figure 1. Cut surface with spongy appearance due to the presence of numerous cysts of varying size.
F&ure 3. Fibrous connective tissue and cysts covered with epithelium and cartilage are evident. On pathologic study the specimen consisted of an ovoid tumor that measured 11 by 7.5 by 6.5 cm, weighed 290 gm, and was soft in consistency. The capsule was 3 mm in thickness and the mass contained pinkish white soft spongy tissue. (Figure 1.) Within this tissue, there were some fibrous grayish bands crossing in several directions. Histologic study revealed structures derived from the different germ layers, particularly cartilage, bone, and glandular formations of columnar epithelium resembling small intestine. (Figure 2.) Other structures included lymphatic follicles, nervous tissue and nervous plexus, and stratified squamous epithelium. No cellular atypias were found and most of the different tissues were well differentiated. The tumor was considered a benign solid gastric teratoma. (Figures 3,4, and 5.) Postoperatively the patient was maintained on continuous intravenous fluids and open drainage of the gastrostomy tube. Normal intestinal peristalsis started on the fourth postoperative day. Oral feedings .were given on the sixth day after surgery. The gastrostomy tube was kept in the stomach for a total of ten days, and the
patient was discharged from the hospital on the twentieth postoperative day. (Table I.)
430
Figure 2. of Photomicrograph gastric watt. Along the muscle layers are cystic formations covered by columnar epitheiium.
Ftgure 4. Nervous tissue.
Comments Teratomas are true neoplasms composed of multiple tissues foreign to the site in which they occur. Literally, teratoma is defined as a malformation and tumor at the same time. The most common sites of occurrence are the sacrococcygeal region, retroperitoneal space, and gonads, but they may appear in other places [l-3,7,8]. Since there are very few reports on teratomas of the stomach, we may conclude that they are rare. A review of the literature indicated that the first case in a pediatric patient was reported by Polony in 1936 [12]. The second registered case was that of Selman [13] in 1943 who described a teratoma arising from the anterior wall of the stomach. A total of twenty-three cases of gastric teratoma in pediatric patients have been reported. Age. Gastric teratomas usually present early in life. Most of the cases in pediatric patients were diagnosed within the first year of life, with the exception of Polony’s case in 1936 and Chaterjee’s in
The Amerkan Journal of Surgery
Gastric
TABLE I
1964 which were discovered in children six and two years of age, respectively. Ten of the reported cases occurred in newborns. ,Teratomas of the stomach rarely occur in adults [4,5,7,8,15]. Sex. It is interesting that all gastric teratomas have occurred in male patients and that teratomas at all other locations have a higher incidence in female patients. Clinical Features. In reviewing the twenty-three reported cases and our own, we found that the main clinical features were abdominal distention and a palpable abdominal mass, being present in twenty patients. Other accompanying manifestations included gastrointestinal bleeding in five patients, vomiting in five, respiratory distress in two, and an abdominal mass in three newborns causing difficult delivery. Fever and abdominal tenderness were present on one occasion each. Radiologic Findings. Radiologic study frequently reveals large calcifications resembling bone and teeth, in contrast to those seen in neuroblastoma or Wilms’ tumor which are very small and of light density. Only thirteen of the reported cases had evidence of calcification on x-ray films. Excretory urography usually shows nothing abnormal and a gastrointestinal series may show deformity of the stomach from extrinsic compression [7,8,15]. Gastric teratomas may arise in any portion of the stomach, fundus, body, or antrum. They may be exo- or endogastric and may vary in size. There have been thirteen exogastric tumors, two were endogastric, and five were both exo- and endogastric; in four the type was not reported. In pediatric patients it is difficult to be aware of the possibility of gastric tumors because of their low incidence. In making a differential diagnosis one should consider meconium peritonitis, neuro-
Volume128,Septemberl974
Infants in
Reported Cases of Teratoma of the Stomach in Pediatric Patients Author
Figure 5. Connective and fibroid tissue and llpoid ceils are Sean.
Teratoma
Polony [ 721 Selman 1731 Large et al (6) Handelsman, Reinhoff, Ward 1771 Reggianini [4] Lodzinski [7] Sarinana, Silva, and Esparza [9] Cooray [14] Ravitch (71 Wagenmann Paul 1741 Kelley (41 Chaterjee [ 71 Panabokke [7] Verhaak [ 751 Lee (81 lkeda [8] Atwell, Claireaux, and Nixon [IO] Meadow (51 Cozzi [7] De Angelis (41 Lebreuil et al (81
Year
1936
and
1943 1952 1955
1955 1956 1958
1959 1960 1961 1962 1963 1964 1964 1964 1964 1966 1967 1968 1968 1969 1969
Patient’s Sex and Age M, 6yr M, 3mo M, 7mo
Outcome Recovered
M,4mo
Recovered Recovered Recovered
M, 1 mo M, 5 days M, 1 mo
Recovered Recovered Recovered
M, 1 mo
Died Recovered
M, 10mo M, 19 days M,9mo M, 1 day M, 2 yr M, 2 mo M, 15 days M, 1 day M) ...
... Recovered Recovered Recovered Died Recovered Recovered
...
M, 1 day
Recovered
M, M, M, M,
Recovered Recovered Recovered Recovered
1 day 3mo 1 day 1 day
blastoma, Wilms’ tumor, gastric duplications, and retroperitoneal teratoma. On the basis of the cases reported, gastric teratomas are benign, with a good prognosis and no reported recurrences after resection. Treatment. Treatment of these tumors is surgical excision with partial gastrectomy or resection of the wall in which the tumor arises. In one instance total gastrectomy was performed and the procedure was well tolerated 141.Complications reported after these operations are not frequent and they may consist of anemia and failure to thrive. Of the twenty-four patients reported, only three died. Summary
Gastric teratoma is a rare condition, particularly in pediatric patients. Only twenty-three cases have previously been reported in the literature. A case of gastric teratoma in an eight month old male infant is presented. The main clinical features were progressive enlargement of the abdomen, hematemesis, and melena. At laparotomy, an ovoid mass originating in the posterior wall of the stomach was found.
431
Azpiroz
et al
Histologic findings consisted of structures derived from the different germ layers, particularly cartilage, bone, and glandular formations resembling small intestine. The clinical and pathologic findings in these cases are reviewed and therapeutic considerations are presented. References 1. Willis RA: Teratomas. Washington, DC, The Armed Forces Institute of Pathology, 195 1, p 9. 2. Dargeon H: Tumors of Childhood. New York, Hoeber, 1960. 3. Salas MM: Las neoplasias en 10s nines. MBxico, DF, MBdicas del Hosp. Inf. de Mex, 1968. 4. De Angelis VR: Gastric teratoma in a newborn infant: total gastrectomy with survival. Surgery 66: 794, 1969. 5. Meadow SR: Gastric teratoma. Postgrad Med J 44: 183, 1968.
432
6. Large HL et al: Gastric tridermal teratoma in infancy. JAMA 28: 824, 1952. 7. Cozri F et al: Teratoma dello stomaco in eta pediatrica: presentazione di un case clinic0 e rassegna della literatura. Arch /ta/ Pediatr 26: 240, 1968. 8. Lebreuil G et al: Teratome gastrique chez un nouveau-n& Arch Anat PatholParis 17: 47, 1969. 9. Sarihana C, Silva CA, Esparza H: Teratoma tridermico del estomago en un ni;o de 45 dias de edad tratado con exito por gastrectomia partial. /30/ Med Hasp Inf Mex 15: 529, 1958. 10. Atwell JD, Claireaux AE, Nixon HH: Teratoma of the stomach in the newborn. J Pediatr Surg 2: 197, 1967. 11. Handelsman JC, Reinhoff WF, Ward GE: Benign teratoma of the stomach in an infant. Am J Dis ChiM90: 196, 1955. 12. Polony M: Tgratome exogastrique. Mem Acad Chir 62: 622, 1936. 13. Selman AN: Complex tridermal teratoma of the stomach. Case report. Am J Surg 59: 567, 1943. 14. Paul M, Cooray GH, Wickremasinghe SY: Teratoma of the stomach. Br J Surg 50: 154, 1982. 15. Aubrespy P, Derlon S: TBratome gastrique chez un nouveau-n& Mem Acad Chir 96: 528, 1970.
The American Journal of Surgery