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Agenesis of the dorsal pancreas: a rare cause of insulin-dependent diabetes without abdominal pain: Case report.

Abstract Agenesis of the dorsal pancreas is a very rare condition with an unknown pathology and etiology, although it may be associated with autosomal dominant or X-linked dominant inheritance or retinoic acid and hedgehog signaling pathway alterations. This condition usually manifests with abdominal pain or pancreatitis, although some cases are asymptomatic. Approximately 50% of affected patients with this disorder present with hyperglycemia or various other anomalies.
PMID
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Dorsal agenesis of the pancreas - a rare cause of abdominal pain and insulin-dependent diabetes.

Authors

Mayor MeshTerms

Congenital Abnormalities

Keywords
Journal Title medicine
Publication Year Start




PMID- 29489657
OWN - NLM
STAT- MEDLINE
DCOM- 20180305
LR  - 20180305
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 97
IP  - 9
DP  - 2018 Mar
TI  - Agenesis of the dorsal pancreas: a rare cause of insulin-dependent diabetes
      without abdominal pain: Case report.
PG  - e0046
LID - 10.1097/MD.0000000000010046 [doi]
AB  - RATIONALE: Agenesis of the dorsal pancreas is a very rare condition with an
      unknown pathology and etiology, although it may be associated with autosomal
      dominant or X-linked dominant inheritance or retinoic acid and hedgehog signaling
      pathway alterations. This condition usually manifests with abdominal pain or
      pancreatitis, although some cases are asymptomatic. Approximately 50% of affected
      patients with this disorder present with hyperglycemia or various other
      anomalies. PATIENT CONCERNS: We report the case of a 23-year-old Chinese woman
      who visited the Department of Endocrinology and Metabolism with insulin-dependent
      diabetes but no specific symptoms, signs, or other deformities. Severe diabetic
      retinopathy indicated a long period of hyperglycemia. DIAGNOSIS: Agenesis of the 
      dorsal pancreas was observed incidentally during the common diagnosis of
      diabetes, and the diagnosis was established using magnetic resonance imaging,
      diffusion-weighted imaging, and magnetic resonance cholangiopancreatography.
      INTERVENTIONS: Following the diagnosis of diabetes, insulin replacement therapy
      was initiated at a dosage of up to 45 U per day. The patient's blood glucose
      level was monitored, and the insulin dosage was adjusted accordingly. OUTCOMES:
      The patient's blood glucose levels gradually normalized after insulin treatment
      and were subsequently maintained with intensive insulin therapy. Treatment for
      diabetic retinopathy was provided by the Ophthalmology Department. LESSONS:
      Agenesis of the dorsal pancreas should be considered in a young patient diagnosed
      with diabetes who presents with obvious diabetes-related complications (e.g.,
      renal, retinal, or neurological) inconsistent with the course of the disease or a
      history of other congenital anomalies. We recommend the routine use of computed
      tomography or magnetic resonance imaging when examining young patients with
      diabetes.
FAU - Liang, Kaiyong
AU  - Liang K
FAU - Ou, Xiaojuan
AU  - Ou X
FAU - Huang, Xukai
AU  - Huang X
FAU - Lan, Qunfang
AU  - Lan Q
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - 0 (Hypoglycemic Agents)
RN  - 0 (Insulin)
RN  - Pancreas agenesis, dorsal
SB  - AIM
SB  - IM
MH  - Abdominal Pain
MH  - Asymptomatic Diseases
MH  - Cholangiopancreatography, Magnetic Resonance
MH  - *Congenital Abnormalities/diagnostic imaging
MH  - Diabetes Mellitus, Type 1/drug therapy/*etiology
MH  - Diffusion Magnetic Resonance Imaging
MH  - Female
MH  - Humans
MH  - Hyperglycemia/etiology
MH  - Hypoglycemic Agents/therapeutic use
MH  - Insulin/therapeutic use
MH  - Magnetic Resonance Angiography
MH  - Pancreas/*abnormalities/diagnostic imaging
MH  - Young Adult
EDAT- 2018/03/01 06:00
MHDA- 2018/03/06 06:00
CRDT- 2018/03/01 06:00
PHST- 2018/03/01 06:00 [entrez]
PHST- 2018/03/01 06:00 [pubmed]
PHST- 2018/03/06 06:00 [medline]
AID - 10.1097/MD.0000000000010046 [doi]
AID - 00005792-201803020-00018 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2018 Mar;97(9):e0046. doi: 10.1097/MD.0000000000010046.