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How to differentiate congenital pancreatic cysts and cystic pancreatic teratomas?

  • Nasser Bustangi
  • Tiphaine Raia-Barjat
  • Mousa Mobarki
  • Alix Clemenson
  • François Varlet
  • Aurelien ScalabreEmail author
ORIGINAL ARTICLE
  • 55 Downloads

Abstract

Purpose

The differential diagnosis of congenital pancreatic cysts and mature cystic pancreatic teratomas is challenging due to the lack of reliable diagnostic tests or pathognomonic imaging findings. This study highlights the differences between these extremely rare entities with few published case reports.

Methods

Based on our recent experience with two cases, we conducted an extensive review of literature including all reported cases of congenital pancreatic cysts and cystic pancreatic teratomas in children.

Results

We report two cases of large cystic abdominal mass detected by prenatal ultrasonography. Both neonates were asymptomatic after birth and were operated on by laparoscopy at the age of 1 year and 3 months, respectively. Histopathology demonstrated a congenital pancreatic cyst in the first case and a mature cystic teratoma in the second case. The post-operative course was uneventful for both children. We describe the clinical and imaging data of the 33 congenital pancreatic cysts and 10 mature cystic teratomas of the pancreas previously reported in the literature, and discuss diagnosis criteria and treatment strategies.

Conclusion

Congenital pancreatic cysts and pancreatic teratomas are rare lesions in children. Clinical presentation is not specific and these lesions are often asymptomatic. Although associated malformations are evocative of congenital pancreatic cyst, the final diagnosis can only be confirmed by histopathological examination. Available treatments include ultrasound-guided puncture, enucleation, distal pancreatectomy, cystogastrostomy, and cysto-jejunostomy by the Roux-en-Y technique. Laparoscopic excision can be performed shortly after birth and is our preferred option.

Keywords

Congenital pancreatic cyst Pancreatic cystic teratoma Dermoid cyst Prenatal diagnosis Laparoscopy 

Introduction

Dermoid cysts of the pancreas, also called mature cystic teratomas, are benign extragonadal germ cell cysts derived from all three germinal layers. They are lined by stratified squamous epithelium and usually contain a variety of tissues. On the other hand, congenital pancreatic cysts are lesions lined by a cuboidal epithelium with a deeper layer of acinar tissue. Both lesions are extremely rare entities with few published case reports. Prenatal diagnosis is challenging due to the lack of reliable diagnostic tests or pathognomonic imaging findings. Common differential diagnoses include other cystic lesions of the upper abdomen such as intestinal duplication and choledocal cyst. The largest pancreatic cysts may be confused with choledocal, mesenteric, adrenal, omental, hepatic, splenic, renal, ovarian, or urachal cysts [1]. The diagnosis of abdominal cystic pulmonary sequestration can also be discussed.

The exact etiology of congenital pancreatic cyst is unclear. They may occur as a result of developmental anomalies of the pancreatic ductal system and result from sequestration of primitive pancreatic ducts [2]. Cysts that develop secondarily to obstruction of the ductal system are sometimes called retention cysts [2, 3]. Pancreatic cystic dysplasia can also be encountered in a variety of congenital syndromes including von Hippel–Lindau and Ivemark II syndromes [4, 5, 6, 7, 8]. In this case, the pancreas usually contains multiple cystic lesions associated with other congenital malformations. Mature cystic teratoma is a congenital developmental abnormality of germ cell origin that may be derived from any of the three germinal layers (ectoderm, mesoderm, and endoderm). These tumors are commonly detected in ovary, but can also occur in testes, cranium, mediastinum, retroperitoneum, and sacrococcygeal regions [9]. Pancreas is the rarest site of presentation and very few cases of pancreatic dermoid cysts were reported in children [10, 11, 12].

We report two cases of large cystic abdominal lesions detected by prenatal ultrasonography. Both children were operated on by laparoscopy with uneventful follow-up. Histopathology demonstrated a congenital pancreatic cyst in the first case and a mature cystic teratoma in the second case. We conducted a systematic literature review and identified 33 congenital pancreatic cysts reported in children and 11 pancreatic dermoid cysts. Diagnosis criteria and treatment strategies are discussed.

Case 1

A 44-year-old women gravida 3 para 2 was referred at 22 weeks of gestation, because a 12 mm abdominal cyst was detected by prenatal ultrasonography. This cyst was located below the stomach, in contact with the inferior vena cava. It was regular and delimited by a thin wall. No other anomaly was detected. Follow-up ultrasounds demonstrated normal fetal growth. A male neonate was born at 38 weeks of gestation by vaginal delivery, weighting 3980 g. He was asymptomatic and we underwent clinical and radiological follow-up. Postnatal ultrasonography showed a progressive growth of the cyst with a diameter of 19 mm after birth and 36 mm 3 months later. Its size remained stable afterward. An MRI was performed at the age of 3 months. The cyst showed high signal intensity on T2-weighted images. There was no biliary dilatation and the gallbladder was normal. The boy remained asymptomatic and the mass was not palpable. Routine blood biochemistry profiles showed an increase of lipase at 127 U/L at 6 months. Surgical treatment was undergone by laparoscopy at the age of 1 year. The cyst was originating from the back of the pancreatic head and was approached by Kocher maneuver. A few mL of clear fluid were aspirated from the cyst, containing 259,160 U/L lipase. A large posterior portion of the cyst wall was excised with the whole mucosa. The post-operative period was uneventful. The boy was discharged 2 days after surgery. Histopathologic evaluation revealed the diagnosis of congenital pancreatic cyst, as the cyst wall was lined by a single layer of cuboidal epithelium. The patient was totally asymptomatic with no sign of recurrence on abdominal ultrasonography after 2 months of follow-up.

Case 2

A 30-year-old woman gravida 1 para 2 was referred at 20 weeks of gestation, because prenatal ultrasonography showed a double bubble sign. A duodenal atresia was evocated. No other malformation was seen and karyotype was 46, XX. The end of pregnancy was uneventful and a female neonate was born by vaginal delivery at 38 weeks of gestation weighing 3120 g. She was completely asymptomatic and there was no palpable abdominal mass. No jaundice or stool color changes were observed. Plain abdominal X-ray excluded the diagnosis of duodenal atresia. Ultrasonography confirmed the persistence of the cystic abdominal mass and showed a well-defined wall without calcifications. It was 3 cm diameter, located in the right hypochondria in contact with the hepatic hilum. As the patient was completely asymptomatic, she was discharged and MRI was programmed 3 weeks later. It revealed the persistence of the cystic mass measuring 35 × 16 × 22 mm, located in the retroperitoneal area just below the hepatic hilum and in front of the inferior vena cava. Diagnosis of duodenal duplication was suspected. Although unlikely, the diagnosis of choledochal cyst was also mentioned. Surgery was performed at 3 months of age by laparoscopic approach (Fig. 1). The retroperitoneal mass was in direct contact with the posterior wall of the pancreatic head. The cyst was opened and contained a whitish sebaceous secretion with debris. The mucosa was completely resected with the extrapancreatic part of the cyst wall. The patient was discharged 3 day post-operative after removal of the abdominal drainage. Lipase level remained normal in the post-operative period. Histologic examination revealed a cyst lined by stratified squamous epithelium with overlying lamellar keratins. The underlying connective tissues were composed of collagen fibers containing multiple tissues (cartilage, lymphoid nodules, pancreatic tissue, colonic epithelium, and scattered neural tissue), suggesting a mature teratoma. No immature foci were identified. At 33 months, the girl was doing well and alpha-fetoprotein (AFP) and US were normal.
Fig. 1

Laparoscopic excision of a mature pancreatic teratoma (asterisk) in a 3-month-old girl. a Aspect of the teratoma located behind the duodenum, b opening of the cystic teratoma, c resection of the extrapancreatic part of the cyst wall, d resection of the mucosa

To review the literature on these topics, articles on true congenital pancreatic cysts and pancreatic teratomas published in English language were identified through a MEDLINE computerized search. Additional sources were identified through cross-referencing.

Clinical, imaging findings, treatment, and outcomes of 33 other congenital pancreatic cysts reported in the literature are summarized in Table 1 [1, 2, 4, 7, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30, 31, 32, 33, 34, 35, 36]. Eleven of them were diagnosed prenatally [1, 13, 14, 15, 16, 17, 18, 19, 20, 36]. Clinical, imaging findings, treatment, and outcomes of 11 pancreatic teratomas in children are summarized in Table 2 [12, 37, 38, 39, 40, 41, 42, 43, 44, 45].
Table 1

Clinical, imaging findings, treatment, and outcomes of 34 cases of congenital pancreatic cysts

References

Age at diagnosis

Gender

Associated malformation

Symptoms

Maximum diameter

Location

Age at time of treatment

Treatment

Follow-up

McPherson [21]

6 months

Male

None

Abdominal distension and vomiting

16 cm

Pancreas body

6 months

Laparotomy, distal pancreatectomy

Uneventful after 2 weeks

Miles [22]

2 days

Male

None

Abdominal distension

23 cm

Pancreas head and body

8 days

Laparotomy, complete resection

Uneventful after 3 years

Pilot 1964 [23]

NA

NA

NA

NA

NA

NA

NA

NA

NA

Gundersen [24]

16 months

Male

None

Abdominal distension

35 cm

Pancreas head and body

16 months

Laparotomy, gastrocystostomy

Recurrence 6 months later, treated by laparotomy and complete resection

Olurin [25]

10 months

Female

None

Abdominal distension and vomiting

NA

NA

NA

Laparotomy, complete resection

NA

Olurin [25]

3 years

Male

None

Abdominal distension and vomiting

NA

NA

NA

Laparotomy, gastrocystostomy

NA

Mares [26]

20 months

Female

None

Abdominal distension

NA

Pancreas head, multilocular

20 months

Laparostomy, cystoduodenostomy

Uneventfull after 18 months

Baker [13]

Antenatal

Female

None

None

3 cm

Pancreas tail

2 months

Laparotomy, distal pancreatectomy

Uneventful after 17 months

Fleet [14]

Antenatal

Male

None

None

5 cm

Pancreas body

4 days

Laparotomy, cysto-jejunostomy by the Roux-en-Y technique

Uneventful after 2 weeks

Auringer [27]

4 months

Female

None

None

7 cm

Pancreas tail

4 months

Laparotomy, complete resection

NR

Shieh [28]

14 years

Female

None

Pancreatitis

6.1 cm

Pancreas tail

14 years

Laparotomy, distal pancreatectomy

Uneventful

Daher [15]

Antenatal second trimester

Female

None

None

5.5 cm

Pancreas tail

Neonatal

Laparotomy, distal pancreatectomy

Uneventful after 1 week

Fremond [16]

Antenatal, second trimester

male

Omphalocele, Beckwith–Wiedemann syndrome

None

7.5 cm

Pancreas head

7 months

Laparotomy, cysto-jejunostomy by the Roux-en-Y technique

Uneventful after 2 years,

Agarwala [29]

6 years

NA

NA

NA

NA

NA

NA

NA

NA

Kebapci [17]

Antenatal, second trimester

Female

None

Abdominal distension

13 cm

Pancreas tail

7 days

Laparotomy, distal pancreatectomy

Uneventfull, 5 days

Sepulveda [36]

Antenatal, second trimester

Male

Right hemihypertrophy

Abdominal distension

12 cm

Pancreas head

8 days

Antenatal drainage at 30 weeks of gestation, laparotomy, complete resection

Metabolic and hepatic dysfunction after surgery, asymptomatic at 1 year of age

Liao [18]

Antenatal 33 weeks of gestation

NA

NA

NA

NA

Pancreas tail

10 days

NA

NA

Boulanger [4]

11 years

Female

None

None

NA

Pancreas head

11 years

Laparoscopy converted to laparotomy, resection of the mucosa and part of the cyst wall

NA

Matta [30]

4 months

Female

None

Abdominal distension, vomiting

13.5 cm

Pancreas head and body

4 months

Laparotomy, cysto-jejunostomy by the Roux-en-Y technique.

Uneventful after 15 months

Kazez [31]

4 months

Male

None

Abdominal distension

9.5 cm

Pancreas tail

NA

Laparotomy, distal pancreatectomy

Uneventful after 6 months

Chung [19]

Antenatal 22 weeks of gestation

Female

None

Abdominal distension

5.8 cm

NA

3 days

Laparotomy, complete resection

Uneventfull after 7 days

Choi [48]

Antenatal

Female

None

None

5.6 cm

Pancreas tail

Neonatal

Laparotomy, distal pancreatectomy

Uneventful

Boulanger [32]

7 weeks

NA

None

Abdominal distension

NA

Pancreas head and body

7 weeks

Laparotomy, complete resection

Uneventful after 16 months

Castellani [20]

Antenatal

Female

None

Abdominal distension, pain, and anorexia

NA

Pancreas body

10 days

Two ultrasound-guided punctures, then laparotomy for cysto-jejunostomy by the Roux-en-Y technique

Uneventful after 30 days

Gerscovich [1]

Antenatal 26 weeks of gestation

Female

None

Abdominal distension

10 cm

Pancreas tail

11 days

Laparotomy

NA

Chahed [7]

1 month

Male

Ivemark II syndrome with situs inversus, asplenia, heart disease, dysplasic liver

Abdominal distension and vomiting

7 cm

NA

1 month

Laparotomy, cystoduodenostomy

Died of heart failure at two and a half months of age

Al Salem [33]

18 months

Male

None

Abdominal distension

9 cm

Pancreas body

18 months

Laparotomy, complete resection

Uneventful after 8 days

Al Salem [33]

4 months

Female

None

Abdominal distension and vomiting

12 cm

Pancreas head and body

4 months

Laparotomy, cysto-jejunostomy by the Roux-en-Y technique.

Uneventful after 2 weeks

Nasher [34]

18 months

Male

None

Abdominal distension

NA

NA

18 months

Laparotomy, complete resection

NA

Warnock [35]

5 weeks

Female

None

NA

NA

NA

NA

Ultrasound-guided punctures, laparoscopic resection, and a second surgery after relapse

NA

Bawazir [2]

4 months

Female

None

Abdominal distension and vomiting

12 cm

Pancreas head and body

4 months

Laparotomy, cysto-jejunostomy by the Roux-en-Y technique.

Uneventful after 2 weeks

Bawazir [2]

16 months

Male

None

Abdominal distension

9 cm

Pancreas body

16 months

Laparotomy, complete resection

Uneventful after 8 days

Bawazir [2]

4 years

Female

None

Abdominal distension

NA

Pancreas head

4 years

Laparotomy for a Wilms tumor, cysto-jejunostomy during the same procedure

NA

Present case no. 1

Antenatal

Male

None

None

3.6 cm

Pancreas head

1 year

Laparoscopic enucleation

Uneventful after 2 months

NA not available

Table 2

Clinical, imaging findings, treatment, and outcomes of 11 cases of pancreatic teratomas in children

References

Age at diagnosis

Gender

Associated malformation

Symptoms

Maximum diameter

Location

Age at time of treatment

Treatment

Follow-up

DeCourcy [37]

2 years

Female

None

Vomiting epigastric mass

5 cm

Pancreas body

2 years

Laparotomy, complete resection

NA

Bittner [38]

2 years

Female

None

Liver failure epigastric mass

NA

Pancreas head

2 years

Laparotomy, complete resection

Uneventful after 6 months

Iovchev [39]

8 years

Male

None

Vomiting fever abdominal pain abdominal mass

NA

Pancreas body

8 years

External drainage

Uneventful after 7 months

Pomosov [40]

6 years

Male

None

Vomiting fever abdominal pain abdominal mass

NA

Pancreas tail

6 years

Laparotomy, distal pancreatectomy

Uneventful after 6 months

Komarov [41]

4 years

Female

None

Vomiting pain epigastric mass

NA

NA

4 years

External drainage

NA

Assawamatiyanont [42]

11 years

Female

None

Abdominal mass

9 cm

Pancreas body

11 years

Laparotomy, complete resection

Uneventful

Das [12]

4 months

Female

None

Abdominal mass

10 cm

Pancreas body and tail

4 months

Laparotomy, complete resection

Uneventful

Yu [43]

2 years

Male

None

Epigastric mass

11 cm

Pancreas head

2 years

Laparotomy, complete resection

NA

Kela [44]

5 months

Male

None

Abdominal pain central mass

8 cm

Pancreas tail

5 months

Laparotomy, complete resection

Immature teratoma

Chemotherapy

Lost to follow-up after the 1st chemotherapy

Wang [45]

12 months

Female

None

Vomiting Central mass

18 cm

Pancreas body

12 months

Laparotomy, complete resection

Recurrence 19 months after

Distal pancreatectomy

Well 2 years later

Present case no. 2

Antenatal 20 weeks of gestation

Female

None

Prenatal diagnosis no palpable mass

3.5 cm

Pancreas head

3 months

Laparoscopy, complete resection

Uneventful after 30 months

NA not available

Discussion

Congenital pancreatic cysts and teratoma are extremely rare diagnoses in children. Including the two present cases, we identified 34 cases of congenital pancreatic cysts and 11 cases of pancreatic teratomas reported in the pediatric population. 11 congenital pancreatic cysts were detected by prenatal ultrasonography. As illustrated by case no. 2, a large abdominal cyst can also correspond to a pancreatic teratoma. Differential diagnosis between these two entities is challenging, as no clinical or radiological sign is pathognomonic.

Congenital pancreatic cysts are revealed by the presence of a large abdominal cyst in the upper part of the abdomen, with no identified parenchymal tissue or calcification. The cyst wall is usually thin, but can be difficult to differentiate from the duodenum wall, making pancreatic cyst difficult to differentiate from enteric cysts. On the other hand, pancreatic teratomas often contain calcifications considered as pathognomonic. The presence of fat/fluid levels, or air/fluid level is another precious sign. However, pancreatic teratoma can appear exclusively cystic on ultrasound and, therefore, be difficult to differentiate from a congenital pancreatic cyst.

Differential diagnoses include duodenal duplication, duodenal atresia, lymphangioma, mesenteric, adrenal, omental, hepatic, splenic, renal, ovarian, and urachal cysts. It may be difficult to localize the site of origin of largest cysts. Ultrasonography must, therefore, detail which intra-abdominal organs are in contact with the cyst. The biliary tract must be described to eliminate the diagnosis of choledocal cyst. Abdominal computed tomography and magnetic resonance imaging can also provide additional information regarding the aspect and localization of the cyst, which is important in planning the operative procedure.

Congenital pancreatic cyst can also be associated with other congenital malformations. Two cases were associated with Beckwith–Wiedemann syndrome [16] and hemihypertrophy [36]. A few cases of pancreatic dysplasia associated with von Hippel–Lindau and Ivemark II syndrome were reported [6, 7, 8]. In 1979, Hopper et al. reported a case of pancreatic dysplasia associated with asphyxiating thoracic dysplasia and short limbs. The infant survived for only 9 h [46]. Balci et al. also reported three cases of sib fetuses with situs inversus totalis, renal and pancreatic dysplasia, bowing of the lower limbs and clavicles, severe intrauterine growth retardation, and oligohydramnios [5]. Pregnancy was terminated in all cases.

We did not identify any case of pancreatic teratoma associated with congenital malformations.

After birth, small pancreatic cysts are usually asymptomatic. In the present case (case no. 1), the cyst size increased throughout the first year of life, while the patient remained asymptomatic. Abdominal distension and vomiting seems to appear when the diameter of the cyst is over 6 cm in infants.

There is no reliable biological marker that can help for the diagnosis of congenital pancreatic cyst or pancreatic teratoma. Immature teratoma can be associated with elevated α-fetoprotein levels, but this is not reliable in infancy. Moreover, Kela et al. reported the only case of immature teratoma in a 5-month-old child and α-fetoprotein level was normal [44]. Conversely, Asai et al. presented a case of retroperitoneal immature teratoma diagnosed at 33 weeks of gestation, with elevated postnatal α-fetoprotein level [47]. Amylase and lipase levels in serum are usually normal, unless the pancreatic duct is obstructed by the cyst [28, 41]. The fluid of congenital pancreatic cyst can contain extremely high levels of pancreatic enzymes [2, 20], as in the present case no. 1, although it was not communicating with the pancreatic duct. Congenital pancreatic cyst containing normal levels of amylase and lipase was also reported [15].

Surgical treatment of pancreatic cyst is usually favored, although there is no consensus regarding the timing of treatment. In the first case, we chose to wait the age of 1 year, as the infant was asymptomatic and to facilitate the laparoscopic approach. The progressive increase of the cyst diameter suggests that congenital pancreatic cyst should be treated before obstruction of the biliary and pancreatic ducts. Surgical removal is also the standard treatment of dermoid cysts. Different treatment strategies were reported. Ultrasound-guided puncture of the cyst has been tempted, even prenatally, but was not sufficient [20, 36]. Complete resection or enucleation should be performed whenever possible, with maximum preservation of the pancreatic tissue [2, 4, 12, 19, 22, 25, 27, 32, 33, 35, 36, 37, 42, 43, 44, 45]. Distal pancreatectomy is possible when the cyst is located into the pancreatic tail [13, 15, 17, 21, 28, 31, 40, 48]. When the cyst is in the pancreatic head, complete removal is not always possible. The less invasive option in this situation is to perform a gastrocystostomy or duodenocystostomy [2, 7, 24, 25, 26]. Alternatively, other surgeons choose to perform a cysto-jejunostomy by the Roux-en-Y technique with good results [2, 3, 14, 16, 20, 30, 33]. The present cases show that laparoscopic removal is a viable treatment option for congenital pancreatic cysts and pancreatic teratomas in infants, with minimal post-operative pain, minimal scaring, and short hospital stay [4, 35].

The final diagnosis can only be confirmed by histopathological examination. True pancreatic cysts contain epithelial tissue not seen in pseudo-pancreatic cyst developing after trauma or infection [27]. These cysts are lined by a single layer of cuboidal epithelium with a deeper layer of acinar tissue [3]. Cystic teratomas are lined by stratified squamous epithelium and connective tissues composed of collagen fibers, and containing multiple tissues from more than one of the three primitive germ cell layers. They usually contain a combination of cystic and solid elements, including teeth, hair, cartilage and dermal appendages such as hair follicles, sweat glands, and abundant sebaceous material. One case of immature dermoid cyst of the pancreas was reported, containing microfoci of immature tissue [44].

We recommend long-term clinical and radiological follow-up, with dosage of α-fetoprotein levels in case of pancreatic teratoma.

Conclusion

Congenital pancreatic cysts and pancreatic teratomas are rare lesions in children. They can be diagnosed by prenatal ultrasonography. Although pancreatic teratomas often contain calcifications seen on imaging studies, they can be difficult to differentiate from congenital cysts. Clinical presentation is not specific and these lesions are often asymptomatic. Preoperative evaluation and detailed imaging are the corner stone for surgical planning. Available treatments include ultrasound-guided puncture, enucleation, distal pancreatectomy, cystogastrostomy, and cysto-jejunostomy by the Roux-en-Y technique. Laparoscopic excision can be performed shortly after birth and is the best option in our opinion.

Notes

Funding

None.

Compliance with ethical standards

Conflict of interest

The authors declare that they have no conflict of interest.

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Copyright information

© Springer Nature Singapore Pte Ltd 2019

Authors and Affiliations

  1. 1.Department of Pediatric SurgeryUniversity Hospital of Saint-EtienneSaint-ÉtienneFrance
  2. 2.Department of Pediatric SurgeryKing Abdul-Aziz University HospitalJeddahSaudi Arabia
  3. 3.Department of Gynecology and ObstetricsUniversity Hospital of Saint-EtienneSaint-ÉtienneFrance
  4. 4.Department of PathologyUniversity Hospital of Saint-EtienneSaint-ÉtienneFrance
  5. 5.Saint-Etienne UniversitySaint-ÉtienneFrance

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